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keratoacanthoma is a common epithelial tumor of the skin characterized by rapid growth ; histopathologic features similar to those of cutaneous sqamous cell caricoma , and a certain tendency for spontaneous resolution . the favored view is that keratoacanthomas are low grade squamous cell carcinomas , which in many cases will regress . the factors which may play a part include sun exposure , minor trauma to skin , immunosuppression , and genetic factors etc . a 22-year - old male patient presented with multiple reddish raised scaly lesions all over his body associated with scaling over the scalp for one year . the patient was shopkeeper by profession dealing in stationary items , there was no history of smoking or tobacco intake . patient had skin type v , and there was no history of diabetes , tuberculosis , or any other immunosuppressive disease . he had applied various emollients and topical steroids with no lasting relief ; however , there was no history of tar application or phototherapy . clinical examination revealed multiple well - circumscribed erythematous indurated scaly plaques on the scalp , bilateral upper and lower limbs , back , and on buttocks . after hematological and biochemical investigations , x - ray chest and hiv test , the patient was started on oral methotrexate 15 mg weekly . he significantly improved on this dose , and pasi dropped to 11.5 after three months . the dose was decreased to 7.5 mg weekly , and the patient was maintained on this dose and kept under monthly follow - up . four months later , the patient presented with reddish to hyperpigmented raised nodules over the healing lesions of psoriasis . he also reported self resolution of few of the lesions over a period of 6 to 12 weeks without any specific treatment . examination revealed the presence of multiple well defined dome shaped erythematous to hyperpigmented nodules , ranging in size from 5 to 15 mm , over both legs , especially shins , and a few over the arms and forearms [ figure 1 ] . few post inflammatory hyperpigmented macules and atrophic scars were seen at the sites where previous nodules had healed spontaneously . right leg shows presence of multiple well - defined domeshaped erythematous to hyperpigmented nodules , ranging in size from 5 to 15 mm . histopathological examination of the nodules showed a globular growth having a central crater containing keratinous material . the epidermis surrounding the crater was acanthotic and overlapped the crater laterally ( lipping ) . these features were consistent with keratoacanthoma , and a diagnosis of multiple keratoacanthomas was made . the epidermis surrounding the crater was acanthotic and overlapped the crater laterally ( lipping ) . [ h & e , 40x ] the patient was unwilling to opt for any therapy for the keratoacanthomas in view of spontaneous resolution of few of them . the patient was followed up for the next 6 months , during which time keratoacanthomas continued to appear and some resolved leaving behind scarring . keratoacanthoma ( ka ) is a rapidly evolving tumor of the skin , composed of keratinizing squamous cells and often resolving spontaneously . multiple kas may occur as part of the ferguson smith or grzybowski 's syndromes or due to other acquired factors . these include ultraviolet ( uv ) light , viruses , chemical carcinogens , immunosuppression , and trauma . the occurrence of kas , mostly on sun - exposed regions of the skin , supports the role of uv light in their causation . prolonged therapy with puva or uvb has also been seen to result in large number of kas . ghadially et al , in their study on 238 patients , found 25 to have history of injury / skin disease causing epithelial disruption ( psoriasis , lichen planus , discoid lupus erythematosus , rosacea , etc ) . vickers and ghadially reported the first case of multiple kas developing in a patient of psoriasis in 1961 . clendenning et al reported kas appearing in a patient of generalized pustular psoriasis . walder et al proposed that high levels of sun exposure in these patients may serve as an initiating factor . clendenning et al postulated that methotrexate therapy in these patients could cause immunosuppression and hence stimulate growth of kas . annamalai et al proposed that chronic inflammation , tar , and phototherapy were responsible for multiple kas and scc in their patient . we believe the most likely etiologic factors for the multiple kas in our patient could be a genetic susceptibility stimulated by either ( a ) epithelial injury due to psoriasis , the patient had history of psoriasis for more than a year , and psoriasis itself causes epithelial injury and chronic inflammation ; moreover , the lesions of keratoacanthoma appeared only in areas of healing psoriatic plaques ; or by ( b ) immunosuppression due to methotrexate , which in this case seems less likely as methotrexate was given for a short duration ; or by ( c ) isomorphic phenomenon : there are single case reports of this phenomenon in keratoacanthoma . the possibility of isomorphic phenomenon in this case seems unlikely as patient had psoriasis for the last one year and no history of keratoacanthoma lesions during that one year ; or by ( d ) it may be sheer coincidence of two diseases occurring together , which again appears unlikely as the lesions of keratoacanthoma appeared only in areas of healing psoriatic plaques .

a 22 year old male psoriatic patient presented with multiple reddish scaly plaques all over body . after hematological and biochemical investigations the patient was started on oral methotrexate 15 mg weekly . pasi score at the start of treatment was 26.2 . after 3 months pasi dropped to 11.5 , the dose of methotrexate was tapered to 7.5 mg weekly and the patient was maintained on this dose and kept under monthly follow up . four months later , the patient presented with reddish to hyperpigmented raised firm nodules having a central crater over the healing plaques of psoriasis . few lesions showed self resolution over a period of 6 - 12 weeks . histopathology of the lesion confirmed it to be keratoacanthoma . we believe the most likely etiologic factors for the multiple kas in our patient could be a genetic susceptibility stimulated by multiple causes .

although a recent clinical trial found no benefit of carotid artery stenting ( cas ) relative to carotid endarterectomy ( cea ) , cas may still represent a potential alternative treatment for patients with a high surgical risk ( 1 ) . recently , the cas procedure has been optimized through refinement of technical devices including stents and distal protection devices . various different stent designs ( such as open - cell and closed - cell ) and materials exist ; however , the safety of the stent itself is rarely taken into consideration . chang et al . ( 2 ) reported that stent deformation is not uncommon after cas ; among 116 cas cases studied , stent fracture and deformation occurred in 27% . herein , we report a case of delayed cerebral infarction caused by longitudinal folding deformation and in - stent thrombosis following cas using a self - expandable stent with an open - cell design . this report had been approved by the institutional review board , with informed consent given by the patient . cerebral angiography showed more than 80% stenosis of the left common carotid artery ( cca ) ( fig . 1a ) : the diameter of the stenotic segment was less than 1.5 mm whereas unaffected proximal and distal segments of the cca measured 9 and 8 mm in diameter , respectively ( fig . carotid artery stenting of the left cca was performed percutaneously via the right femoral artery with a self - expandable stent with an open - cell design ( protg rx carotid stent system , ev3 , plymouth , mn , usa ; 10 60 mm ) . this lesion was pre - dilated and then post - dilated following placement of the stent . aspirin ( 100 mg / day ) and clopidogrel ( 75 mg / day ) were administered daily for 8 months following stent implantation , and the latter was stopped thereafter . after 4 years , a sudden neurological episode occurred , characterized by the right hand weakness . computed tomography angiography ( cta ) revealed the deformation to comprise longitudinal folding and divide into two separated lumens along the whole segment of the implanted stent ( fig . the left cca angiography confirmed extensive in - stent thrombosis with longitudinal folding deformation of the stent ( fig . 1c , d ) , and clopidogrel ( 75 mg / day ) was administered immediately . carotid stent fracture has been reported with markedly disparate incidences from 2% to 29% , and stent deformation without fracture is also likely to occur frequently with the incidence of 23% ( 2 , 3 , 4 , 5 ) . however the clinical impact of stent fracture or deformation is still unclear . to our knowledge , this is the first case report for the longitudinal folding deformation of the carotid stent combined with delayed cerebral infarction . in create trial of the protg stent ( 6 ) , they did not demonstrate delayed ischemic complication due to stent folding deformation among the 408 cas cases studied . in this case , we used the self - expandable nitinol stent with an open - cell design . generally , the cell size in open - cell stents differs between the ends and the body of a stent . relative to stents of the same design , the used stent is known to have a wider cell size in both ends , which may weaken the radial force from the stent ends , thereby resulting in stent folding deformation . ( 7 ) analyzed carotid stent fracture that occurred mainly in self - expandable nitinol stents , and chang et al . ( 2 ) observed that stent deformation was significantly more common in open cell stents than closed cell stents . this stent was placed in the left cca , which is located in a highly active part of the human body . the carotid artery is recently considered as a possible region of stent fracture because of various neck motion and high - pressure pulsatile flows . carotid stents are subject to external mechanical forces of axial and crush deformation by complex head and neck movement ( 8) . vos et al . ( 9 ) demonstrated that carotid artery could lose its natural flexion , extension , and rotational flexibility after cas . therefore , the stented segment of the artery may be a rigid unit in all head positions , thus resulting in kinking or compression at the junctions between the stent and artery . ( 10 ) reported head rotation initiated neurologic symptoms in patients with carotid stent fracture . other possible causes of stent deformation are vessel angulation of more than 45 and presence of heavy calcification around the stented artery ( 2 , 5 ) . however , in our case , no calcification along the left cca occurred , and the stented artery was a straight configuration without curvature ( fig . we experienced a delayed ischemic complication due to such a stent deformation , which was an uncommon phenomenon . some authors described case reports of late stroke related to stent fractures but not stent deformations ( 10 , 11 ) . however , delayed neurological symptoms are known to develop rarely in cases of stent fractures or deformations . although stent fractures were often associated with restenosis , stent fractures or deformations were independent of late stroke ( 2 , 5 , 7 ) . no established method of managing deformation and collapse of a carotid stent , which may precipitate cerebral infarction through in - stent thrombus and restenosis , exists . in symptomatic cases of stent fracture , various therapeutic interventions including anticoagulant treatment , restenting , stent removal by open surgery , endarterectomy , or a bypass graft may be considered ( 7 ) . in this case , we employed a conservative approach to management with dual antiplatelet therapy ; the patient was successfully treated and remained asymptomatic during 12 months of follow - up after discharge . delayed cerebral infarction due to longitudinal folding deformation of the carotid stent is a very rare complication of cas . although the causes of such a complication remain unclear , attending clinicians should be aware of the possibility of this complication following cas and should inform patients of the prospective risk prior to treatment . delayed cerebral infarction due to longitudinal folding deformation of the carotid stent is a very rare complication of cas . although the causes of such a complication remain unclear , attending clinicians should be aware of the possibility of this complication following cas and should inform patients of the prospective risk prior to treatment .

we report a case of delayed cerebral infarction due to stent longitudinal folding deformation following carotid artery stenting using a self - expandable stent with an open - cell design . the stented segment of the left common carotid artery was divided into two different lumens by this folding deformation , and the separated lumens became restricted with in - stent thrombosis . although no established method of managing this rare complication exists , a conservative approach was taken with administration of anticoagulant and dual antiplatelet therapy . no neurological symptoms were observed during several months of clinical follow - up after discharge .

a 49yearold woman suspected of having a right optic neuritis was referred to the neurology department . she had been diagnosed with systemic lupus erythematosus ( sle ) 10 years earlier , and , at the time of presentation , she was receiving azathioprine 50 mg three times a day , prednisone 5 mg daily , and belimumab 640 milligrams intravenously every 4 weeks . four months earlier , during an ophthalmologist screening test for retinal toxicity , an optic coherence tomography ( oct ) had shown a reduction in the thickness of the macular and retinal nerve fiber layer in the right eye . she was recommended to stop treatment with hydroxychloroquine , which the patient had been receiving since the initial diagnosis of sle 10 years before ( 200 mg orally per day ) . an mri showed a highintensity signal in her right optic nerve at that moment ( fig . , followup showed an inferior nasal scotoma in her right eye , not present in previous examinations , and she was referred to the neurology department . a perineural contrast enhancement can be seen on the right optic nerve , suggesting inflammation of the nerve sheath . during the first visit to the neurologist , physical examination did not reveal other pathological findings , so ancillary tests ( visual evoked potentials and both orbital and cerebral mri ) were requested . before the tests could be carried out , the patient developed vertical diplopia on the primary gaze position that was accentuated when looking up . a paralysis of the right eye in upgaze position was found on physical examination then . mri showed signs of inflammation in the right orbit with affected the orbital fat and conditioned a slight proptosis of the eyeball , not visible to the naked eye on physical examination . a mild posterior perineural signal could still be seen in the optic nerve , but also , both the right superior rectus and superior oblique muscles were thickened ( fig . , we found that these alterations in the muscles were already but subtly present in the first mri , long before the diplopia appeared ( fig . visual evoked potentials demonstrated prolonged latencies after visual stimulus , consistent with a moderate demyelinating neuropathy of the right eye . extensive laboratory tests , including blood cell count , renal function , liver function , thyroid function , antiaquaporin4 antibodies , angiotensinconverting enzyme levels , and serologic tests for syphilis , hiv , herpesvirus , and borrelia burgdoferi , were negative or normal . antidsdna levels were 167 ui / ml , and serum complement levels were low , with c3 0.76 g / l and c4 0.07 g / l ; these results were similar to the results of previous blood tests . t2weighted brain and orbital mri . ( a ) coronal view of the mri performed nine months before the patient developed the symptoms . a slight thickening of the superior rectus , which appears hyperintense compared to the contralateral muscle , is patent , as well as mild infiltration of the surrounding fat . ( c ) axial cuts of the same mri showing also slight proptosis of the right eye . hydroxychloroquinerelated toxicity was ruled out due to both clinical and radiologic progression after suspension of the treatment . the patient was diagnosed with orbital inflammatory pseudotumor secondary to sle , and highdose corticosteroid therapy was initiated ( oral metilpredinsolone one gram daily for 5 days ) , followed by slowly decreasing doses of prednisone to 10 mg daily . this therapy achieved to improve the symptoms and diplopia resolved , without permanent damage to the visual field . afterward , immunosuppressive treatment with rituximab was started . the patient has remained asymptomatic ever since , and both antidsdna and complement levels have returned to normal values . neuroophthalmic manifestations , however , are less frequent , with a prevalence of 3.6% in adults ; among these , optic neuritis is the most common form of presentation and also one of the most visually compromising complications of sle 1 . the implication of orbital structures in sle and , specifically , inflammatory pseudotumor is very rare and therefore may go unnoticed 1 , 2 , 3 . it usually presents with proptosis , pain , and diplopia , due to infiltration of the ocular muscles . although increases in the titers of antidsdna antibodies and decreases in the complement levels may accompany disease relapses , this is not true for all sle patients 4 , 5 . in our case , although high levels of antidsdna and low levels of c3 and c4 were found , these results were similar to the results of previous blood tests . visual loss due to inflammatory infiltration of the optic nerve is uncommon ; however , it may be a preceding sign , as in this case . 2 , 6 . response to corticosteroid therapy is excellent in most patients , but sometimes progression of the disease could lead to major damage 2 . although sleassociated retinopathy has been associated with elevated levels of antiphospholipid antibodies 2 , this does not seem to be the case in inflammatory pseudotumor , and due to its low incidence , no risk factors that could help early diagnosis have been identified . retinopathy and maculopathy are widely known complications of hydroxychloroquine treatment , although inflammatory pseudotumor has never been described in this setting . although visual loss may be irreversible and even worsen after suspending treatment with hydroxychloroquine , this is more likely if high doses of hydroxychloroquine are used for more than 5 years or if there is impaired renal or liver function 7 , 8 . although our patient had been treated with hydroxychloroquine for 10 years , the daily doses were below 6 , 5 mg / kg , which are associated with very low risk of retinopathy . besides , she never presented impaired renal or liver function in successive blood tests . taking all this into account suspecting inflammatory pseudotumor and its relation to the sle is imperative to avoid further complications . in this case , the slow progression of the disorder , probably due to simultaneous immunosuppressive therapy and chronic treatment with low doses of oral corticosteroids , as well as the interference of confounding factors ( treatment with hydroxychloroquine ) delayed the diagnosis . however , when the patient developed diplopia a review of the previous mri showed that mild signs had been already present months before the symptoms appeared . therefore , after a new finding appears , going back to previous studies may help to put things into perspective .

key clinical messageorbital inflammatory pseudotumor is a rare complication of systemic lupus erythematosus . it may present a challenge for differential diagnosis , especially in the context of treatment with hydroxychloroquine , although dosage and duration of the treatment may guide us . although high antibody titers can be found , this is not specific .

cerebral arterial air embolism ( caae ) has been reported as a rare complication of medical intervention . we present a case of a 7-month - old girl who was admitted to our emergency department ; post - mortem computed tomography ( ct ) showed a caae . she was known in our hospital because of prematurity ( gestational age , 30 weeks ) and an omphalocele , for which initial non - operative treatment with epithelialisation was instituted , and delayed surgical correction was planned . according to the mother , the child awoke at 3 o clock at night and , as what had happened before , her mother bottle fed her . during feeding the latter was first to arrive and transported the girl and her mother to the emergency department of the academic medical centre amsterdam . they arrived in our hospital approximately 45 min after the 911 call . upon physical examination , a non - responsive child without spontaneous several attempts were made to insert a central venous and arterial catheter , however , to no avail . to gain venous access , an intraosseous infusion ( ioi ) needle was placed in the right tibia . at 5:00 a.m. , she was pronounced deceased . although the clinical history and findings during resuscitation suggested food aspiration , questions regarding the cause of death remained therefore , according to our battered child protocol , the standard radiographs , following the guidelines of the american college of radiography were performed . in addition , a head ct , which was a standard in our hospital in children under the age of 2 years with unexplained trauma / death , was performed within 1h after death . the skeletal radiographs showed no abnormalities , except for the io infusion in the proximal right tibia ( fig . 1 ) . the head ct , however , showed a considerable amount of air within the arterial circulation ( fig . 2 ) . 2ct at the level of the circle of willis showing air within the circle of willis ( open arrow ) anterior cerebral artery ( small arrow ) and peripheral cerebral arteries ( solid arrow ) intraosseous infusion needle correctly positioned within the proximal right tibial metaphysis ct at the level of the circle of willis showing air within the circle of willis ( open arrow ) anterior cerebral artery ( small arrow ) and peripheral cerebral arteries ( solid arrow ) a full judicial autopsy was performed in the netherlands forensic institute , as is mandatory in children with a possible non - natural cause of death . the body showed normal measurements ( length , 64 cm , and 6,900 grams , both p50 ) and a known omphalocele . autopsy showed that the omphalocele contained a large segment of the right liver lobe and the ascending colon including the appendix , with adhesion to the abdominal wall ( fig . inspection of the heart revealed only minor congenital abnormalities , which consisted of a defect in the interatrial septum , fossa ovalis type with deficient flap valve , and slight hypertrabeculation of the ventricles , particularly the right ventricle ( fig . 4 ) . the latter findings , however , were clearly insufficient for a diagnosis of ventricular non - compaction . the trachea was without abnormalities and , in contrast to the clinical history , showed no signs of food aspiration ( fig . additional toxicological , microbiological and biochemical analyses of body fluids and tissues were all inconclusive . 3omphalocele containing a large section of the right liver lobe ( asterisk ) and the ascending colon including the appendix ( arrow)fig . 4view of the atrial septum showing a persistent foramen ovale ( arrow ) and a fenestration ( arrowhead)fig . 5view of the trachea devoid of food remnants , a small amount of saliva is seen ( arrow ) omphalocele containing a large section of the right liver lobe ( asterisk ) and the ascending colon including the appendix ( arrow ) view of the atrial septum showing a persistent foramen ovale ( arrow ) and a fenestration ( arrowhead ) view of the trachea devoid of food remnants , a small amount of saliva is seen ( arrow ) as no distinct cause of death could be established , the cause of death remains undetermined . caae is a rare finding ; it has been described after medical instrumentation in trauma patients , after mechanical ventilation or resuscitation [ 28 ] . in case of vascular access procedures , caae can occur as a result from backflow , a right to left shunt , or shunting through the pulmonary capillary bed [ 911 ] . backflow can be a result of manipulation of central lines ( not necessarily ioi ) and has been described in literature [ 12 , 13 ] . right to left shunting can be caused by an atrial septum defect ( estimated incidence in adult patients 0.20.7 in 1 , 000 ) , a ventricular septum defect ( incidence at birth ranging from 25% ; however , 8590% of these defects will close spontaneously by 1 year of age ) and persistent foramen ovale ( this has been reported to be present in up to 27% of autopsies ) [ 1416 ] . a special category are divers who suffer from decompression sickness after diving accidents [ 17 , 18 ] . in severe cases , caae can develop , which is reported to be the second most common cause of death in divers . recently , an animal study with sheep was performed to test the hypothesis that artifacts caused by postmortem off gassing is at least partly responsible for the presence of gas within the vascular system and tissues of the cadaver after death associated with compressed air diving . none of the control animals showed intravascular air on ct , respectively , 1 and 8 h after death ; only after 24 h after the time of death , relatively small amounts of gas were seen . although it is unknown how well sheep resemble 7-month - old infants , we assume that decay rates will not differ that much and certainly not to the extent that it can explain the amount of air visible on the ct scan . in our case , the only vascular access had been io infusion , which is a widely implemented technique and is part of the standard protocols , such as the advanced paediatric life support textbook . the advantage of io infusion over peripheral vascular access is that it provides a rapid and reliable access to the systemic venous circulation in children [ 2123 ] . it can be used in children of all ages , with the smallest child reported in literature weighing 800 g . this has led to io infusion having almost completely replaced saphenous cut down procedures in critically ill children in emergency situations . it is generally considered to be a safe technique with a reported complication rate of 1% . several complications resulting from the use of io infusion have been reported ; these consisted of extravasation in some cases leading to compartment syndrome ( in rare cases leading to limb amputation ) , osteomyelitis , fracture , skin necrosis and fat embolism [ 24 , 2732 ] . recently , investigators from the berne institute of forensic medicine ( switzerland ) reported on a case of air embolism after resuscitation and ioi in a 4-month - old boy . in their case , resuscitation was considered as the cause for caae ; however , whole - body ct showed air in the lower extremity , which was used for ioi . as in our case , io infusion was the only vascular access . given the patent foramen ovale and atrial septal defect , leading to a right to left shunt , we feel that the only logical explanation for the caae is that air is introduced into the bloodstream via this route . to the best of our knowledge , our case is the second reported case of an air embolism after the use of an io infusion . given the incidence of right - to - left shunts in the general population , this is a serious complication of ioi of which clinicians should be aware .

cerebral arterial air embolism ( caae ) has been reported as a rare complication of medical intervention . there has been one reported case of caae after the use of an intraosseous infusion ( io ) system . we report on a case of caae after tibial io infusion in a 7-month - old girl during resuscitation .

percutaneous kyphoplasty has been proved to be effective in the treatment of osteoporotic compression fractures7,11,12 ) . although the complication rate is low , many complications including cement leakage , emboli to the brain , lung and heart , intraoperative rib fractures , and subsequent fractures of the adjacent or augmented vertebrae have been reported in the recent literature4,14 ) . however , infectious complications after cement augmentation are still rarely reported2,8,13,16,17 ) . tuberculous infections are even more rare , and to the best of our knowledge , only 2 cases of tuberculous spondylitis after cement augmentation have been reported3,10 ) . here we report a case of tuberculous spondylitis after kyphoplasty and its successful management with a review of the literature . a 76-year - old woman visited our clinic for severe low back pain recently aggravated without any predisposing factor . mag- netic resonance imaging ( mri ) scan revealed a benign compression fracture of l1 . blood pressure was maintained within the normal range and blood sugar level was well regulated . three years later , the patient presented to the pain clinic complaining of recurrent low back pain . several days later , the patient returned to our department due to aggravation of low back pain and development of fever . laboratory tests revealed elevated inflammatory markers , erythrocyte sedimentation rate ( esr ) , c - reactive protein ( crp ) and procalcitonin levels . mri scan was conducted and showed new compression fracture at t12 , paravertebral abscess , and spondylitis of t12 and l1 ( fig . we suspected bacterial spondylitis , as there was no typical evidence of tuberculous spondylitis including commencement of infection at the anterior aspect of the vertebral body adjacent to the endplate . the patient did not respond to antibiotic treatment and there was no decrease in inflammatory markers . total corpectomy of t12 and l1 was performed using a transdiaphragmatic retropleural - retroperitoneal approach . then anterior reinforcement with a titanium mesh cage filled with autologous rib graft was performed . the necrotic tissue was biopsied and cultured . however , the bacterial culture was negative . histologic examination of the resected specimen revealed a granulomatous inflammation with caseous necrosis ( fig . specific polymerase chain reaction and acid - fast bacilli stain were subsequently carried out and they confirmed the diagnosis of a tuberculous infection . antibiotics were promptly changed to anti - tuberculous medications including isoniazid , rifampicin , pyrazinamide , and ethambutol . approximately three months after the surgery and anti - tuberculous medications , inflammatory markers decreased to subclinical levels . the anti - tuberculous medications were administered for 10 months , and the patient recovered without any sequelae . there are several reports of episodes of serious local infections after cement augmentation in patients with any history ofsystemic infection13,16 ) . reports of cases of spinal tuberculosis after cement augmentation are extremely rare , with only 2 cases being reported in the literature with pubmed search ( table 1)3,10 ) . although spinal tuberculosis accounts for 2% of all tuberculosis cases , its incidence is increasing in parallel with the growing numbers of immunocompromised patients10 ) . the occurrence of tuberculous spondylitis can be explained in several ways . in case of active pulmonary tuberculosis , hematogenous spreading from the lung to the vertebra is the most probable mechanism10 ) . as cultures of bronchial washings confirmed the diagnosis of tuberculosis , the case reported by ivo can be explained to have been caused by hematogenous spreading10 ) . however , the present case and the case reported by bouvresse showed no evidence of active pulmonary tuberculosis3 ) . since in most of the cases reported in the literature , the patients did not suffer from pulmonary tuberculosis , local reacti- vation of quiescentmycobacteria can be suggested as another probable mechanism3,10 ) . mycobacteria divide asymmetrically , generating a population of cells that grow at different rates , have different sizes , and differ in how susceptible they are to antibiotics , increasing the chances that at least some will survive1 ) . in addition , mycobacteria - infected macrophages at the primary site of infection may migrate and initiate tuberculous spondylitis3 ) . ivo suggested that cement augmentation may act as a trigger for serious tuberculous or nontuberculous infections by unknown mechanisms10 ) . this concept explains why tuberculous and nontuberculous mycobacterial infections arise at the site of injury . according to this concept , cement augmentation can be considered as surgical trauma , and hence tuberculous spondylitis may develop easily . . clinical and radiological features of tuberculous spondylitis and benign com- pression fracture are similar5 ) . however , in our case , it is unclear whether kyphoplasty triggered a local tissue response thereby increasing susceptibility to tuberculous spondylitisor we performed kyphoplasty on an infected vertebra since the time between kyphoplasty and the occurrence of tuberculous spondylitis was more than three years . in the case of tuberculous spondylitis , although mri scan is currently the most accurate imaging study , it still may not differentiate betweentuberculous spondylitis and benign compression fracture in some cases5 ) . the patient in the case reported by ivo initially took broad spectrum antibiotics while our patient underwent conservative treatment for two months10 ) . as the correct diagnosis and specific treatment is essential , a physician should be aware of this disease entity to avoid any delay in diagnosis and treatment . in case of presence of any risk factors for tuberculosis , also , if no pathogen is cultured from tissue specimens of infective spondylitis , performing tuberculosis culture should be considered . active investigation including microbiological and histologic examination is of utmost importance to avoid any delay in correct diagnosis and specific treatment . if medical treatment fails , the surgical resection of the infected vertebrae is recommended with careful consideration of the patient 's general medical condition . if conservative antibiotic therapy fails , resection of the infected bone - cement complex is indicated .

a 76-year - old woman with compression fracture of l1 underwent percutaneous balloon kyphoplasty using polymethyl methacrylate . three years after kyphoplasty of l1 , the patient was readmitted with severe low back pain . magnetic resonance imaging revealed progressive collapse of l1 vertebra and new compression fracture at t12 . there were no signs of infection . as conservative treatment failed , combined surgery consisting of anterior corpectomy of t12 and l1 , interposition of a titanium mesh cage filled with autologous rib graft , and anterior instrumentation of t11-l2 was performed . histologic examination showed granulomatous inflammation surrounding the cement . polymerase chain reaction and culture of the specimen confirmed the diagnosis of tuberculosis . the anti - tuberculous medications were administered for 10 months , and the patient recovered without any sequelae . tuberculous spondylitis should be included in the differential diagnosis of spondylitis after cement augmentation . if conservative antibiotic therapy fails , resection of the infected bone - cement complex is indicated .

it presents as a painless and slowly growing mass and also as exophthalmoses like in orbital masses . this study aims to identify the clinical course , surgical treatment and histopathologic properties of pleomorphic adenoma diagnosed in a patient who had painless exophthalmia with an intraorbital mass . a 62-year - old male patient referred to our clinic due to painless exophthalmia of his right eye for the last 2 years . on neurological examination of the patient , there was no abnormality except for the up - gaze restriction and severe exophthalmia . cranial magnetic resonance imaging ( mri ) revealed an isointense mass on the superolateral part of right orbita in the coronal intersections and it was contrasted homogenously ( fig . 2a and b ) . the existing score of our patient according to rose and wright was + 8 points histological examination of the surgical specimen covered the epithelial and stromal components that formed the glandular structures . figure 1:(a and b ) computerized tomography revealed a homogenously contrasted intraorbital mass in the right eye . figure 2:(a and b ) cranial mri revealed a homogenously contrasted mass on the superolateral part of the right orbita . figure 3:(a and b ) postoperative computerized tomography revealed the replacement of the eye . figure 4:(a ) four times h - e biphasic tumor composed of epithelial and stromal components . ( b ) ten times h - e fibromyxoid stroma and epithelium that form glandular structures . ( a and b ) computerized tomography revealed a homogenously contrasted intraorbital mass in the right eye . ( a and b ) cranial mri revealed a homogenously contrasted mass on the superolateral part of the right orbita . ( a ) four times h - e biphasic tumor composed of epithelial and stromal components . ( b ) ten times h - e fibromyxoid stroma and epithelium that form glandular structures . although the orbital tumors originate primarily from vascular , muscle , cartilage , neural tissues , lacrimal glands and lymphoid structures , they can also originate from the surrounding structures and ( low possibility of ) metastasis . epithelial tumors account for 30% of the lacrimal gland lesions , while 12% of epithelial tumors are pleomorphic adenoma . the term pleomorphic ( benign mixed tumor ) was first defined and used by willis . malignant transformation can occur in 1020% of pleomorphic adenomas of the lacrimal gland [ 5 , 6 ] . seventy - five percent of pleomorphic adenomas transform into pleomorphic adenocarcinoma , while the rest transform into cystic carcinoma . pleomorphic adenoma must be considered for the patients whose clinical course lasts for 12 months . malignancy must be considered for cases with pain and progredien symptoms . in his study , wright reported that 39 of 40 cases diagnosed with primary malignant lacrimal gland tumors had a painful mass . in addition to pain , neoplasm of the lacrimal gland , which destroys the bones , can be interpreted as malignancy . while pleomorphic adenomas do not destroy the bones , malignant neoplasm does [ 9 , 10 ] . although pleomorphic adenomas constitute 49% of all orbital tumors , their morbidity is very high . displacement of the mass without its capsule and malignant transformations following the incisional biopsies prior to the diagnosis of pleomorphic adenoma increase the morbidity . to this end , rose and wright developed a scoring system to evaluate the lacrimal gland masses [ 6 , 9 ] ( table 1 ) . according to this scoring system , if the value obtained from this table is below + 2 , the value may indicate carcinoma and thus incisional biopsy can be performed . the values + 3 and higher may indicate pleomorphic adenoma and thus , incisional biopsy must not be performed . the success of the treatment depends on the displacement of the mass with its capsule . it can also lead to malignant transformation of pleomorphic adenoma . the existing score of our patient according to rose and wright was + 8 points . table 1:management of lacrimal gland masses according to rose and wrightclinicalscore1 + 1duration of acute symptoms<10 months>10 monthspersistent painpresentabsentsensory losspresentabsentradiological1 + 1well - defined round or oval massabsentpresentmoulding of mass to globe or along the lateral orbital wallpresentabsenttumor calcificationpresentabsentinvasion of bonepresentabsentduration of symptoms in relation to tumor sizeshort symptomslong symptoms management of lacrimal gland masses according to rose and wright pleomorphic adenoma of the lacrimal gland must be considered in the presence of long - term painless exophthalmoses . for the patients suspected to have pleomorphic adenoma , incisional biopsy should not be performed in order to prevent the likelihood of a relapse to occur . the displacement of the mass with its capsule can extend the patients ' lives and improve their quality of life .

we present a case of a 62-year - old male patient with pleomorphic adenoma and painless solid mass in his right eye . computerized tomography demonstrated a mass with a diameter of 2.5 cm located in the right lacrimal gland . the mass was removed completely by combined orbitofrontal craniotomy through a transcranial approach . histopathologic examination revealed pleomorphic adenoma of the lacrimal gland . orbital tumors originate primarily from vascular , muscle , cartilage , neural tissues , lacrimal glands and lymphoid structures . five percent of all intraorbital masses originate from the lacrimal gland . pleomorphic adenoma presents as a painless and slowly growing mass and also as exophthalmoses . pleomorphic adenoma has a high morbidity . morbidity increases due to the total displacement of the tumor without its capsule and incisional biopsy for the purpose of diagnosis . the success of the treatment depends on the removal of the tumor with its capsule .

circle hairs ( ch ) represent a body hair growth disorder characterized by asymptomatic presence of hairs with typical circular or spiraliform arrangement , not associated with follicular or inflammatory abnormalities ; they are almost exclusively seen on the trunk and upper legs of male overweight patients , where they are interspersed with normal hairs . clinical , trichoscopic and histopathologic findings of six cases of ch are presented and discussed along with literature review . six caucasian adult males came to our observation for the evaluation of different cutaneous disorders , as described in [ table 1 ] . during physical examination , in all patients was noticed the presence of several ch , among normal hairs , especially on the trunk and thighs [ figures 1a2a ; no signs of inflammation or follicular abnormalities were present . no specific association with other dermatoses and/or systemic diseases has been evidenced in any patient . trichoscopy at 150 magnification [ figures 1b and c , 2b and c , 3a c ] showed in all cases no shaft abnormality and evidenced that some of these ch were located under a thin skin layer corresponding to outer stratum corneum , whereas others were protruding out of skin ; most of ch showed an almost perfect round configuration . after pulling , ch showed a characteristic question mark appearance [ figure 3d ] . in two patients ( patients 1 and 4 ) a biopsy was performed and histopathologic evaluation showed that hair follicle density was normal , with intermediate , miniaturized , anagen and catagen hair follicles and no inflammatory perifollicular infiltrate . moreover , river - bed , dilated , follicle infundibula , with entrapped hair shafts , were observed [ figures 1d2d ] . patients with circle hairs : our experience patient 1 : several dark circle hairs ( ch ) , among normal hairs , on the abdomen ( a ) ; trichoscopy at 150 showing ch protruding from the skin ( b ) or located under a thin skin layer ( c ) ; histopathology showing a river - bed , dilated , follicle infundibulum , with an entrapped hair shaft ( d ) patient 4 : clinical ( a ) , trichoscopic ( b , c ) and histopathologic ( d ) aspect of circle hairs patients 2 ( a ) , 3 ( b ) , 5 ( c ) : trichoscopic aspect of circle hairs ( ch ) . after the initial description of 16 cases by adatto , another seven cases of ch have been clinically , but not histologically , described in the english literature [ table 2 ] . although this condition is rarely reported , it is certainly underestimated , as medical consultation for ch is rare in practice . as in our cases , all reported patients with ch were adult males and the most frequent localization was the back . patients with circle hairs : cases reported in the literature the pathogenesis of ch remains obscure . according to some authors , ch have a small hair diameter that make it difficult to penetrate stratum corneum and , for this reason , the hair grows in a circular tract and in a subcorneal location . other authors do not agree with this hypothesis , claiming that the follicular ostia are continuously open and hence that the hair does not need to perforate the stratum corneum : based on this , they maintain that ch are probably a genetically pre - determined disorder . the possibility that ch correspond to vestigia ( or remnants ) of the mammal undercoat has also been suggested . trichoscopy is a non - invasive technique that has been utilized for the evaluation of different hair disorders . in our cases moreover , it displayed that some ch were located under a thin skin layer , whereas others were protruding from the skin . a possible explanation for this is that the rolling initially takes place below the stratum corneum ; then , with epidermis turnover , the hair protrudes from the skin . , it showed the presence of a river bed dilated follicle infundibulum with entrapped hair shaft . we found no evidence of inflammatory infiltrate and no obstruction at the level of the follicular ostia that might have hampered hair growth , confirming that the follicular ostia are open . whether the dilated infundibulum is the primary cause of ch , or secondary to the subcorneal hair growth , remains to be established and could represent an area of future research . differential diagnosis of ch mainly includes rolled hairs , which are not perfectly circular , but may appear as irregularly coiled within a hyperkeratotic papule and associated with signs of inflammation and , often , with abnormalities of keratinization , such as atopic dermatitis , ichthyosis and palmoplantar keratoderma .

circle hairs ( ch ) represent a body hair growth disorder characterized by asymptomatic presence of hairs with typical circular or spiraliform arrangement , not associated with follicular or inflammatory abnormalities . although this condition is rarely reported , it is probably underestimated , as a medical consultation for ch only is rare in practice . trichoscopic and histopathological findings of ch have never been reported and this article will present and discuss six cases along with literature review .

anterior - segment optical coherence tomography ( as - oct ) is a noncontact , noninvasive method of imaging the anterior ocular segment . cross - sectional images of the tissue obtained using this instrument facilitate analysis of the cornea,1 anterior chamber depth , iris , and lens.2 trabeculectomy blebs3 and scleral flaps4 have also been imaged using as - oct . in addition , a previous study evaluated laser peripheral iridotomy openings using as - oct.5 however , to our knowledge , the current report appears to report the first use of as - oct to visualize what was likely either an incomplete iridotomy or a membrane over an iridotomy . although visualization of the anterior lens capsule through an iridotomy opening by slit - lamp examination is necessary to confirm patency , it may be difficult in some patients with a small iridotomy opening . we report a case of recurrent acute angle - closure glaucoma following a small iridotomy for which patency could not be confirmed by slit - lamp examination . therefore , to confirm the patency , we used as - oct cross - sectional images . intraocular pressure ( iop ) was 42 mmhg in the right eye and 70 mmhg in the left eye . slit - lamp examination showed a cloudy cornea , and gonioscopy showed anterior chamber angle closure in both eyes . the patient was diagnosed with bilateral acute angle - closure glaucoma , and neodymium - doped yttrium aluminum garnet ( nd : yag ) laser iridotomy was performed to eliminate pupillary block in both eyes . after the procedure , iop decreased to a normal level . one month later , however , the patient presented with blurred vision in the right eye . iop was 70 mmhg in the right eye and 23 mmhg in the left eye . slit - lamp examination of the right eye showed a cloudy cornea . despite its small size , the previous laser iridotomy was observed at the 10 oclock position in the iris ( figure 1a ) . following compression , gonioscopy showed that the closure was partially appositional , and more than half of the angle had synechiae . based on the sudden iop elevation associated with a cloudy cornea and blurred vision , we suspected recurrent acute angle - closure glaucoma in this patient , although the initial iridotomy appeared open on slit - lamp examination . to further evaluate the precise condition of the initial laser iridotomy , we referred to cross - sectional images obtained using as - oct ( casia ss-1000 ; tomey corporation , nagoya , japan ) . the images showed a membrane that was not observed by slit - lamp examination , with an anterior bowing configuration at the base of the iridotomy ( figure 1b ) , suggesting increased fluid pressure in the posterior chamber . this observation indicated that pupillary block was the causative mechanism of the increased iop in our patient , and retreatment with nd : yag laser iridotomy was performed at the same site . after the procedure , the iop decreased to 9 mmhg . the anterior lens capsule was observed through the opening by slit - lamp examination ( figure 2a ) . a cross - sectional image showed a completely penetrated iridotomy with widening of the anterior chamber angle and straightening of the iris ( figure 2b ) . in the current case , we believed that recurrent acute angle - closure glaucoma was involved in the pathogenesis , given the sudden iop elevation associated with a cloudy cornea and blurred vision , although the initial laser iridotomy appeared open based on slit - lamp examination . however , we speculated that occlusion of the initial iridotomy occurred at some level of the iridotomy hole that was not directly observable during slit - lamp examination due to the small opening . we performed as - oct to obtain more information about the condition of the iridotomy . the cross - sectional images clearly showed a membrane with an anterior bowing configuration at the base of the iridotomy , suggesting increased fluid pressure in the posterior chamber due to a functional block between the lens and iris . postoperatively , widening of the anterior chamber angle and straightening of the iris occurred and iop decreased . in this case , we thought that the plateau iris configuration might have contributed partially to the pathogenesis ( figure 2c ) . additional laser peripheral iridoplasty will stabilize the iop over a long period . considering that a minimal diameter of 150200 microns is recommended when performing a laser iridotomy,6 the size of the initial iridotomy in our patient was relatively small . however , localized pigment proliferation causes late closure and leads to development of a transparent , thin , fibrous membrane occluding the opening and regeneration of the iris pigment epithelium from the margins of the iridotomy.7 in the current case , the latter mechanism was likely , given that a membrane was clearly present at the bottom of the iridotomy . because the intensity of the membrane was not equivalent to that of the iris stroma although late closure is rare with nd : yag laser iridotomies compared with argon laser iridotomy,8 in our patient the iridotomy closed soon after the initial procedure , despite use of an nd : yag laser . we think that the fibrous membrane developed and regenerated at the base of the iridotomy because of an accelerated wound - healing process in our patient , who was relatively young compared with elderly patients with angle - closure glaucoma , who generally require laser iridotomy . in the current case , an invasive surgery ( ie , goniosynechialysis with or without phacoemulsification ) was avoided by carefully considering the pathogenesis of the iop elevation and evaluating the condition of the iridotomy using as - oct . if the iridotomy was found to be open only by slit - lamp examination , our young patient might have had to undergo that invasive surgery . although visualizing the anterior lens capsule through the iridotomy opening by slit - lamp examination is necessary to confirm the patency , it may be difficult for some patients with a small iridotomy opening . therefore , cross - sectional images obtained using as - oct might be helpful to confirm the precise condition of iridotomy in those patients .

we report the case of a patient with recurrent acute angle - closure glaucoma who had undergone a previous laser iridotomy . because the initial iridotomy was small , patency could not be determined by slit - lamp examination . therefore , anterior - segment optical coherence tomography was used to evaluate the patency . cross - sectional images showed the presence of a membrane with an anterior bowing configuration at the base of the iridotomy , suggesting that recurrent pupillary block was the causative mechanism . a repeat laser iridotomy was performed , with a resultant decrease in the intraocular pressure and widening of the anterior chamber angle . anterior - segment optical coherence tomography may be helpful to confirm the status of a laser iridotomy , especially when the iridotomy is small .

gastric volvulus is a rare condition that comes in two subsets : organoaxial and the less frequent mesenteroaxial volvulus ( 29% ) . an organoaxial volvulus rotates around the long axis of the stomach that bisects from the gastroesophageal junction and the pylorus of the stomach . on the other hand , a mesenteroaxial gastric volvulus most frequently occurs secondary to diaphragmatic defects , but occasionally present even in the absence of predisposing anatomic abnormalities . acute cases of gastric volvulus typically present with the borchardt triad of epigastric pain , retching without emesis and inability to pass a nasogastric tube . chronic cases may have an intermittent presentation of upper abdominal distension , gastroesophageal reflux or intermittent dysphagia . imaging is imperative in the diagnosis of an intermittently presenting volvulus due to the vague symptoms that patients experience . this can present a diagnostic dilemma as conclusive findings in imaging are only present during symptomatic periods and due to the intermittent nature of the disorder , a volvulus may spontaneously resolve before imaging studies are performed . a 22-year - old male presents to the emergency department with several hours of abdominal pain , nausea and vomiting without blood . he has a 2-year history of having episodes of similar symptoms . over the 2-year span , the patient has had a multitude of workups that included esophagogastroduodenoscopys ( egds ) , abdominal x - rays , upper gastrointestinal ( gi ) contrast studies , computed tomography ( ct ) scans and magnetic resonance imagings of the brain and abdomen . physical examination revealed a thin appearing , well - developed male with no acute distress . the laboratory results of the patient were creatinine 1.10 mg / dl ( 0.601.00 ) , serum glucose 124 mg / dl ( 65100 ) , hemoglobin 16.9 l ( 12.016.0 ) and total bilirubin 1.6 mg / dl ( 0.21.0 ) . a gastroenterologist was consulted and an urgent egd was done , showing negative insufflations of the stomach that were characteristic of a volvulus . the diagnosis of mesenteroaxial volvulus was confirmed by single contrast upper gi ( ugi ) imaging ( fig . 1 ) . there was no evidence of a hiatal hernia , diaphragmatic hernia or meckel 's diverticulum on visualization . a 24-french gastrostomy tube was inserted percutaneously into the stomach and inflated . using the gastrostomy tube a point along the greater curve of the stomach , distal to the volvulus , was chosen to suture the stomach to the abdominal wall with permanent sutures . the patient tolerated the surgical procedure well ( fig . 2 ) and was discharged 4 days later . an acute gastric volvulus is a true medical emergency as it can lead to ischemia , necrosis and perforation carrying a mortality rate as high as 3050% [ 1 , 2 ] . a chronic , intermittent volvulus is difficult to diagnose because patients can present with a wide range of symptoms and severity . a symptomatic , chronic gastric volvulus presents with vague symptoms that spontaneously resolve and may include upper abdominal distension , early satiety , water brash , gastroesophageal reflux or intermittent dysphagia . diagnosis is often delayed by the nature of the intermittent pathology , whereby the imaging studies are only abnormal during symptomatic periods . gastric ligament laxity can predispose a patient to excessive rotation of the stomach and intra - abdominal adhesions can act as a hinge for the stomach to rotate around and form a volvulus [ 2 , 5 ] . other risk factors include patients aged over 50-years , asplenism , small and large bowel malformations , pyloric stenosis , colonic distention , rectal atresia , gastric tumor and splenic or left hepatic lobe agenesis [ 3 , 4 ] . x - ray , egd , ct scan and upper gi contrast study all can be used to visualize a volvulus . an x - ray often shows a retrocardiac air bubble or an expanded air fluid level in the chest , while an upper gi contrast study may reveal an upside - down stomach where the pylorus is positioned above the fundus [ 1 , 4 , 5 ] . the preferred treatment for gastric volvulus is repair of the paraesophageal hernia with fundoplication with or without gastropexy . in the absence of diaphragmatic pathology , it is important for physicians to maintain a high clinical suspicion for an intermittent volvulus as the presentation is often vague and subsequent imaging is frequently negative . multiple modalities of imaging that were done on our patient to diagnose the mesenteroaxial volvulus were negative because the volvulus had resolved by the time the imaging studies were conducted . in addition , this patient did not possess any risk factors to indicate a gastric volvulus which made the diagnoses even more difficult . when the diagnosis was confirmed in our patient , prompt surgical treatment was performed to correct the anatomy , prevent potential complications of obstruction and ischemia of the stomach , and relieve the patient of any future discomforts caused by his gastric volvulus .

mesenteroaxial volvulus is a form of gastric volvulus that rotates around the short axis of the stomach . mesenteroaxial volvulus typically presents secondary to an anatomical defect with symptoms that include epigastric pain , retching , dysphagia and early satiety . our patient presented with episodic abdominal pain , nausea and vomiting for 2 years . previous imaging was unremarkable but an esophagogastroduodenoscopy done when the patient most recently presented with abdominal pain revealed a mesenteroaxial volvulus . he underwent a laparoscopic gastrostomy - tube gastropexy and has not had any recurrence of his symptoms to date . this case illustrates the difficulties in diagnosing an intermittent volvulus as untimely imaging of a temporarily unfolded volvulus can delay diagnosis and treatment .

new , large genomic data sets are providing more in - depth insights into the diagnosis and treatment of disease . in the past decade , new and innovative methods have continued to add value to the underlying data and uncover the secrets of the genome . visual data inspection by experienced researchers is an important quality control element in the analytical process . unfortunately , this part of the process is tedious and time consuming , and the increasing volumes of high - throughput sequencing data of various types and platforms are proving to be a major analytical challenge . here , we report a visualization tool that allows researchers to explore their data at a very rapid speed and significantly reduce the burden of reviewing tens and hundreds of thousands of variant calls . areas with systematic read errors can be quickly identified , and inefficient attempts to verify results in noisy regions can be avoided . alview is a fast and portable visualization tool . the core code interfaces with heng li et al s samtools library1 for parsing bam files . the program is written in platform - independent c. peculiarities specific to an operating system are isolated with if defined ( ifdef ) directives ; so , for instance , when microsoft visual c provides alternate support for a portable operating system interface ( posix ) standard function , a handcrafted , native interface work around is supplied . for graphical user interface ( gui ) frameworks , alview uses win32 interface for windows , the gtk2 interface for linux , and bsd unix - based systems and cocoa for apple mac os x. samtools1 is written to posix standards , but different microsoft visual compilers provide various levels of support for these unix style standards . as a result , the source code for third - party libraries that were modified for windows is provided to facilitate compiling and linking alview on windows . the main code for alview , in the file alviewcore.cpp , is written to be portable between operating systems and emphasizes speed of execution . the code can be compiled as a stand - alone executable and must be linked with the zlib2 and samtools1 libraries . sequence reads are processed via custom samtools callback functions arranged in in - memory data structures and represented by an aesthetic , annotated image . the image is then output to the screen as a native graphics object or to the disk as a standard image format file . alview can also be compiled as a webserver daemon that uses the common gateway interface ( cgi)3 standard . the cgi version produces interactive html output and uses dynamic html54 features , including zoom in by selection via a jquery5 library . the cgi webserver alview version loads a list of permitted - to - access bam files from a user - maintained text file ; so custom lists of bam files of interest are easy to generate and use . the source code is free and open to modification so that users and local system operators can implement their own security . the alview cgi webserver version provides modifiable url access , so that , for instance , cells in a spreadsheet can link to viewable results for any sample or location . stand - alone alview accepts parameters that specify bam file name and genomic coordinates . invoking alview in a script can create a slideshow of interesting regions . for example , fields in a single nucleotide polymorphism ( snp ) detection output file can be used to specify a series of calls to alview to generate images for each purported polymorphism or mutation . the burden of reviewing ten and hundreds of thousands of mutation calls can therefore be significantly reduced . the source code is available at github.6 the readme file there points to links for selected executables and complete download packages that include the associated reference genome data . a live webserver version of alview for examining public human cancer short - read datasets is available at https://cgwb.nci.nih.gov/cgi-bin/alview . alview source code and executables for several operating systems are available at the national cancer institute ( nci)/national cancer informatics program s ( ncip s ) github site : https://github.com/ncip/alview . nci retains the copyrights to national cancer institute and associated images , which may not be used in forked projects . alview provides a solid substructure that allows for various types of access to short - read data across different operating systems . figure 1 . demonstrates the various navigation and information buttons available in the web version of alview and shows how selection via mouse provides zoom in capabilities . alview is a trim , fast , precise tool and complements existing programs such as the integrated genomics viewer ( igv),7 bamview,8 and gbrowse 2.0.9 the benefits of alview are extreme speed and a sharp focus on exploring short reads . different implementation philosophies can influence memory usage and performance but provide useful alternative paths to solving similar problems . igv provides much more functionality than alview by supporting many other input file types other than bam sequence read files . igv s java implementation provides write once , run anywhere portability via implementations of the java virtual machine . alview s implementation relies on low - level operating system and native gui toolkit api calls . igv requires registration for download for running off of disk , whereas alview does not . desktop igv may require internet for full , easy , simple operation , whereas alview does not require network connection ( though it may call user - invoked external webpages ) . 7 intel core i52400 cpu at 3.10 ghz and 8 gb ram , restarts of igv v2.3 took from 12 to 18 seconds . restarts of alview took a small fraction of one second . for a small view of a genomic region , the java platform se binary for igv took up 292 mb , while alview took up 11 mb .

the name alview is a contraction of the term alignment viewer . alview is a compiled to native architecture software tool for visualizing the alignment of sequencing data . inputs are files of short - read sequences aligned to a reference genome in the sam / bam format and files containing reference genome data . outputs are visualizations of these aligned short reads . alview is written in portable c with optional graphical user interface ( gui ) code written in c , c++ , and objective - c . the application can run in three different ways : as a web server , as a command line tool , or as a native , gui program . alview is compatible with microsoft windows , linux , and apple os x. it is available as a web demo at https://cgwb.nci.nih.gov/cgi-bin/alview . the source code and windows / mac / linux executables are available via https://github.com/ncip/alview .

adenoid hypertrophy ( ah ) is described as the non - physiological enlargement of the nasopharyngeal tonsils which may lead to nasal obstruction . there are several previously suggested histopathological mechanisms for adult ah , among them chronic nasal inflammation is the most popular one ( 1 ) . rhinoliths develop within the nasal cavity as a result of mineralization of an endogenous or exogenous nidus . rhinoliths cause rhinitis which may lead to complications including nasal polyps , atrophic rhinitis , septal perforation and oroantral fistula ( 2 ) . the aim of this article is to present a case with rhinolithiasis which also has ipsilateral asymmetric adenoid hypertrophy . additionally , previous reports were reviewed for the presence of this clinical entity and possible mechanisms were described . a 24 year old male presented with unilateral nasal obstruction . additionally , the patient described ipsilateral facial pain and persistent purulent rhinorrhea . nasal endoscopic examination revealed purulent secretion and a left sided mass which totally obstructed the nasal cavity . the mass had irregular contours , recovered by granulation tissue and was very hard and immovable on palpation with forceps . nasal endoscopy was impossible on the left side and was normal on the right side with minimal nasopharyngeal adenoid hypertrophy . a computed tomography ( ct ) was performed and a left sided mass in the inferior meatus which has demonstrated central calcification was reported ( figure 1 ) . mild mucosal hypertrophy was present in the left ethmoid and maxillary sinuses , accompanied with ipsilateral asymmetric adenoid hypertrophy ( figure 2 ) . ( r : rinolith , o : orbit , m : maxillary sinus ) asymmetric adenoid hypertrophy , more prominent on the left side . ad : adenoid hypertrophy the patient underwent endonasal endoscopic surgery with the diagnosis of rhinolithiasis . after removal of the rhinolith , adenoid hypertrophy was prominent on the left side ( figure 3 ) . adenoidectomy was subsequently performed and postoperative histopathologic examination revealed lymphoid hyperplasia and chronic inflammatory cell infiltration without any malignant transformation . it : inferior turbinate , ad : adenoid hypertrophy , nx : nasopharynx . within the two year follow - up period , rhinolithiasis is a clinical entity caused by rhinoliths ( 2 ) . malignant or benign tumors of the nasal cavity may also be calcified ( 3 ) . the most important differential diagnoses include hemangioma , osteoma , calcified polyps , in the benign tumor category ; chondrosarcoma and osteosarcoma in the malignant tumor category ; syphilis and tuberculosis in the inflammatory category . ct can provide information that helps to distinguish rhinolith from these entities ( 3,4 ) . ct also provides information about the accompanying diseases and complications including nasal polyps , septal deviation or sinusitis ( 5 ) . rinoliths may be presented as nasal polyps and ct scan is very important in differential diagnosis of these cases ( 5 ) . another valuable diagnostic tool is the nasal endoscopy in the differential diagnosis of rhinolithiasis . in a previous report , ogretmenoglu described the value of endoscopy in the diagnosis of rhinolithiasis ( 6 ) . however paranasal sinus ct scan , especially in axial slices , would be the preferred method of imaging these masses over endoscopy due to its sensitivity and specificity for calcification and foreign bodies , both important features of rhinoliths . additionally nasal endoscopy might not be possible to perform on the affected side in case of such a huge space - occupying mass . endoscopic approach allows the surgeon to intervene accompanying diseases including nasal polyps , sinusitis , septal perforation and septum deviation . nasal saline irrigation under direct vision after removal of the rhinolith , was suggested previously in order to prevent revisions ( 7 ) . since chronic inflammation is a common feature of rhinolithiasis , ah may accompany rhinoliths and interfere with nasal obstruction in case of huge - sized rhinoliths . asymmetric adenoid tissue accompanying rhinolithiasis might be overlooked easily and this may cause the symptoms of the patient to persist after the surgery . a careful postoperative endoscopic examination is also mandatory in patients with rhinolithiasis . however , we have found similar cases in previous reports while reviewing the literature , where asymmetric adenoid hypertrophy was overlooked or underestimated by the authors . in a previous study , a relatively significant asymmetric nasopharyngeal mass was prominent on the axial ct scan of a patient with ipsilateral rhinolith , but the authors did not look any further into this finding ( 8) . in another study , clinically insignificant asymmetric adenoid hypertrophy could be observed on the same side with the rinolith ( 9 ) . the first suggested mechanism is the persistent inflammation and chronic postnasal drip , which cause chronic low - grade inflammation and interfere with the physiologic regression of adenoid tissue . this mechanism was advocated in a previous report as a cause of persistent adenoid tissue in adults ( 1 ) . chronic inflammatory cell infiltration in the adult adenoid tissue also supports this mechanism ( 1 ) . hamdan et al described the higher prevelance of ah in adults with nasal obstruction than adults without nasal obstruction ( 10 ) . the left nasal cavity was totally obstructed in the presented case and lack of airflow in one side might have caused asymmetric ah . another contribution to this asymmetry might be the lying position of the patient for years . contribution of all these mechanisms with different percentages seems to be the most reasonable mechanism for asymmetric ah in this patient . as a conclusion , significant asymmetric adenoid careful endoscopic nasopharyngoscopy as well as paranasal ct scan should be performed before the removal of a rhinolith in order not to overlook asymmetric adenoid tissue . more than one mechanism seems to contribute to the formation or persistance of asymmetric adenoid tissue in the rhinolithiasis patients .

the aim of this article is to present a rhinolithiasis patient with a significant asymmetric adenoid hypertrophy on the same side and to describe possible mechanisms for this clinical entity . careful nasopharyngoscopy after removal of rhinolith is mandatory not to overlook significant adenoid hypertrophy which may interfere with patients symptoms . the role of paranasal ct scan in the diagnosis of an asymmetric adenoid hypertrophy in rhinolithiasis patients is also discussed .

aggregates of c60 or c70 were synthesized and subsequently exposed to methane inside of superfluid helium nanodroplets . the nanodroplets were produced by expanding helium from a stagnation pressure of approximately 2 mpa through a 5 m nozzle , cooled to about 8 k , into vacuum . the average number of helium atoms per droplet formed in the expansion was on the order of 5 10 ; the droplets cool to a temperature of 0.37 k by evaporation . the first cell was filled with low - pressure c60 or c70 ( ses corp . , purity 99.95% ) , which was vaporized from a crucible ; methane at partial pressures ranging from 1 10 to 4 10 pa was present in the second cell . methane complexes grew in the helium droplets upon successive collisions with the dopant molecules . in some experiments , the droplets were doped with ch4 before being doped with c60 ; in another set of experiments , only one pickup region filled with c60 and ch4 was used . after the pickup region , the doped helium droplets passed a region in which they were ionized by electron impact at 70 ev . cations were accelerated into the extraction region of a commercial time - of - flight mass spectrometer equipped with a reflectron ; its mass resolution was about m / m = 1/5000 . we used classical md simulations in combination with force fields constructed from quantum mechanical calculations . in the md simulations , c60 and ch4 the fullerene aggregate was space - fixed at its optimized geometry ; one of the fullerenes carried the charge . the intermolecular forces were represented by analytical atom atom pair potentials . for the fullerene methane force field , density functional calculations with the long - range and dispersion - corrected density functional b97x - d in combination with pople 's 6 - 31g(d , p ) basis set ( 976 points ) were performed . methane was , as expected , slightly deeper bound to c60 than to neutral c60 ; otherwise , the potentials were very similar . methane force field was derived from coupled cluster ccsd calculations with dunning 's correlation - consistent triple- ( cc - pvtz ) basis set ( 816 points).the analytical force fields were retrieved by a nonlinear fitting procedure . the dl_poly_4 simulation package was used for the md simulations ( simulation parameters : t = 4 k , t = 2 fs , overall simulation time 400 ps ) , while for the quantum chemical calculations , the gaussian 09 a.02 program suite was used .

bundles of single - walled nanotubes are promising candidates for storage of hydrogen , methane , and other hydrogen - rich molecules , but experiments are hindered by nonuniformity of the tubes . we overcome the problem by investigating methane adsorption on aggregates of fullerenes containing up to six c60 ; the systems feature adsorption sites similar to those of nanotube bundles . four different types of adsorption sites are distinguished , namely , registered sites above the carbon hexagons and pentagons , groove sites between adjacent fullerenes , dimple sites between three adjacent fullerenes , and exterior sites . the nature and adsorption energies of the sites in c60 aggregates are determined by density functional theory and molecular dynamics ( md ) simulations . excellent agreement between experiment and theory is obtained for the adsorption capacity in these sites .

liposarcomas are the second most common soft tissue sarcoma in adults after malignant fibrous histiocytoma , predominantly occurring in the lower extremity and the retroperitoneum . it constitutes < 1% of all mediastinal malignancies and 9% of all primary mediastinal sarcomas with scarce data in the literature to date [ 1 , 2 ] . in this study , we present a case of primary liposarcoma of the anterior mediastinum abutting and compressing intrathoracic structures in a young female . a 28-year - old asymptomatic obese female presented to the bariatric clinic for weight reduction surgery . during the routine bariatric preoperative evaluation , examination revealed decreased breath sounds bilaterally and dullness to percussion in the anterior chest . on x - ray , widening of the mediastinum with possible intrathoracic further laboratory investigation of common tumor markers , such as abg , afp , hcg , ca 19.9 and cea , were all negative along with chemistry and hepatic panels . ct imaging was performed and revealed a large heterogeneous anterior mediastinal mass with fat and soft tissue . with possible differential including thymolipoma , liposarcoma and germ cell tumors , further imaging of the abdomen , brain and bones ct - guided biopsy revealed a well - differentiated sclerosing variant liposarcoma with cellular - spindled areas . they were negative for sma , cd117 and dog1 ( figs 15 ) . figure 1:initial bariatric preoperative chest x - ray that led us to the diagnosis . . there are scattered tumor giant cells and rare lipoblasts , as well as numerous floret - like multinucleated giant cells ( arrows ) . importantly , silver and chromogenic in situ hybridization was performed for mdm2 and shows diffuse amplification . there are scattered tumor giant cells and rare lipoblasts , as well as numerous floret - like multinucleated giant cells ( arrows ) . importantly , silver and chromogenic in situ hybridization was performed for mdm2 and shows diffuse amplification . the surgical approached was via a median sternotomy and the mass was resected without any complications from the anterior mediastinum and with an intact capsule . the smooth lobulated yellow - red mass weighed 1840 g , and measured 28.0 19.0 8.0 cm . the patient was discharged without any complications and has continued to follow up in clinic without any complaints . although the overall anatomic distribution may be widespread , primary liposarcoma of intrathoracic origin is a very rare entity with few reported cases in the international literature . liposarcoma of the anterior mediastinum is even rarer as it is more commonly found in the posterior mediastinum . most of these masses are diagnosed incidentally or worked up secondary to compression symptoms from intrathoracic structures : dyspnea , tachypnea , wheezing , chest pain , svc compression and voice hoarseness . these tumors are most commonly discovered during chest x - ray with a finding of widened mediastinum . on ct , a low attenuation signal between 50 and 150 hu is indicative of fatty tissue while greater signal intensity indicates soft tissue or tissue necrosis . of course , these indicators can not be used as a diagnosis and differentials for such a mass must include thymolipoma , teratoma , germ cell tumor , lymphoma , herniated peritoneal fat and diaphragmatic hernia . mean age of diagnosis is 43 and it is very rarely documented in children and young adults . these tumors are mesenchymal in origin , and the clinical course and outcome of liposarcoma largely depends on the histopathologic type of liposarcoma as they may be broadly classified into four histologic types : well - differentiated , dedifferentiated , myxoid and pleomorphic . in a 2014 retrospective study of 23 patients conducted in shanghai , well - differentiated and atypical lipomatous variants carried the best prognosis while dedifferentiated and pleomorphic carried the worst . they also found that radical surgery had a better overall survival rate compared with nonradical surgery . for patient receiving nonradical surgery , radiation and chemotherapy did not improve the prognosis or rate of relapse . furthermore , there was no significant difference between overall survival and disease - free survival when comparing parameters such as tumor size , gender and age . based on the limited published data , surgical resection appears to be the best treatment modality and greatly increases the rate of disease - free and overall survival . in difficult cases where complete resection is not possible or difficulty ensues in identifying the margin , en bloc debulking is the best option . certain authors consider that huge masses require preoperative radiotherapy and chemotherapy before debulking surgery or as adjuncts , but they have shown to have low efficacy . recurrence within 6 months is common in deep - seated liposarcomas , but may take as long as 510 years after the initial excision . recurrence is related to the incomplete excision and the amount of tumor tissue left behind at the time of surgery . for difficult resections where the boundary of the tumor tissue is hard to delineate this case describes a young female who was incidentally identified with an anterior mediastinal mass and was promptly treated with radical surgery . eighteen months later , there has been no sign of recurrence ; however , the patient will be closely followed for the next year and there on . although this case was made easier due to the fact that the tumor had a well - differentiated capsule , radical surgery appears to be the best option for all anterior mediastinal tumors .

this case describes the incidental finding and surgical removal of an 1.8-kg liposarcoma in the anterior mediastinum . these tumors are very rare and would normally present with symptoms of intrathoracic compression ; however , this patient was completely asymptomatic . the case presentation and treatment rationale are described along with a brief review of existing literature .

cutaneous lung tissue heterotopia is a rare and peculiar disorder characterized by well - formed , mature lung tissue formation with bronchi , bronchioles , and alveoli in the skin . it looks like a hemangioma due to extensive vasculature , but is composed of mature lung tissue . the only other reported case was a 3-yr - old girl with lung heterotopia on the skin over the scapula ( 1 ) . we describe the clinical , radiological , and pathological properties of a heterotopia of lung tissue in the skin over the anterior suprasternal area of the neck in a newborn infant . this is the first report of mature lung tissue in the skin reported in korea . a male newborn infant ( date of birth july 6 , 2009 ) , weighing 3,480 g at 39 weeks and 2 days of gestation , was delivered by cesarean section due to a prenatally detected huge mass on the anterior aspect of the neck identified by pelvic magnetic resonance imaging at 19 weeks of gestation . he presented with a 4.23.51.0 cm reddish brown hemangioma - like protruding mass connected to the middle of the manubrium of the sternum by a band - like stalk . the surface of the mass was wrinkled , and vessel tissues were visible under the surface . a chest computed tomographic scan revealed that an anterior chest wall mass that was lobulated , hypervascular , and highly enhanced , and contained two arteries and veins which crossed the middle of the manubrium of the sternum ( fig . another 1.12.3 cm highly enhanced mass was located in the anterior mediastinum between the sternum and heart , and was similar to the anterior chest wall mass ( fig . 2b ) . on the 8th postnatal day , he underwent an excision operation of the mass under general anesthesia . the excised mass measured 4.23.51.0 cm , and the cut surface showed a tan to brownish , multi - lobulated , sponge - form lesion in the dermis and subcutis . the specimen was fixed in 10% formalin and routinely processed . on microscopic examination of hematoxylin - eosin stained slides , lobulated lung parenchymal tissue and a dilated bronchus with mature cartilages were seen in the dermis and subcutis ( fig . the lung parenchymal tissue showed small bronchioles lined by columnar cells and alveolar spaces containing alveolar macrophages ( fig . these columnar epithelia and alveolar epithelial cells revealed nuclear positivity on immunohistochemical stain for ttf-1 ( anti - thyroid transcription factor-1 , clone : 8g7g3/1 , glostrup , denmark , dako ) , marker of type ii cell and clara cell of the lung ( fig . the overlying skin and skin appendages were unremarkable , and no other unusual histologic components suggesting teratoma were present . he is one month old now , has no respiratory symptoms , and is being followed at out- patient clinic . however , pathologic examination shows well - formed , mature lung tissue formation in the skin , namely bronchi consisted of cartilage , bronchioles , and alveoli through the dermis and subcutis ( 1 ) . it was first reported in a 3-yr - old girl on the skin over the scapula by singer ( 1 ) . mahler et al . ( 2 ) reported cutaneous ectopic respiratory epithelium , but not respiratory mucosa , on the anterolateral neck , suggesting a branchial cleft remnant . ( 3 ) described cutaneous ectopic respiratory mucosa on the arm , suggesting abnormal limb buds containing sequestrated cells from the branchial cleft . cutaneous lung tissue heterotopia is easily differentiated from pulmonary sequestration , teratoma , bronchogenic cyst , and branchial cleft cyst by histology and the location of the mass . pulmonary sequestration is a nonfunctioning lung tissue supplied by a systemic artery , not connected to bronchi , and containing a mesothelial cell layer ( 4 , 5 ) . teratoma is composed of multiple cell types originating from more than one germ layer , and usually all three layers ( 6 ) . moreno et al . ( 7 ) reported cutaneous cystic teratoma with hair follicles , eccrine sweat glands and ducts , and ciliated respiratory epithelium with goblet cells in the middle of the back in a 5-yr - old girl . the larynx and trachea of the upper respiratory tract typically has ciliated pseudostratified columnar epithelium , so cutaneous bronchogenic cysts are lined by ciliated pseudostratified columnar epithelium with goblet cells and contain mesenchymal components such as smooth muscles and cartilage ( 8) . it is usually found in the pre - sternal area around the sternal notch , but can occur over the scapula ( 9 , 10 ) . branchial cleft cysts contain stratified squamous epithelium or occasionally ciliated pseudostratified columnar epithelium , and have continuity with a sebaceous gland . these cysts prominently have lymphoid nodules , but rarely have smooth muscle or cartilage , and are usually located in the anterolateral aspect of the neck ( 11 ) . bronchogenic cysts and branchial cleft cysts usually include cysts , sinuses , and fistulas , and can be differentiated from lung tissue heterotopias . cutaneous bronchogenic cysts occur from impaired or delayed midline fusion of the mesenchymal bars of the sternum , so the accessory buds from the primitive tracheobronchial tree that originate from the ventral portion of the primitive foregut are sequestrated in the pre - sternal area ( 12 ) . the exact etiology of cutaneous lung tissue heterotopia is not known , and the development of lung tissue in the pre - sternal area in this case report is difficult to explain , but may involve a similar process as pre - sternal cutaneous bronchogenic cysts . ectopic tissues in the skin such as cutaneous lung tissue heterotopias , pulmonary sequestration , bronchogenic cysts , branchial cleft cysts , thyroglossal duct cysts , or teratoma , generally require total excision because of the concern for potential malignancies . in conclusion , this report describes the first case of cutaneous lung tissue heterotopia in korea , as confirmed and differentiated from pulmonary sequestration , bronchogenic cyst , branchial cleft cyst , and teratoma by pathologic findings .

cutaneous lung tissue heterotopia is a very rare disorder where mature lung tissues develop in the skin . this is only the second known report of cutaneous lung tissue heterotopia , with the first by singer et al . in 1998 . a newborn infant had a hemangioma - like , freely movable mass connected to the anterior aspect of the sternal manubrium . pathologic findings showed mature lung tissues with bronchi , bronchioles , and alveoli through the dermis and subcutis , and it was diagnosed as cutaneous lung tissue heterotopia . cutaneous lung tissue heterotopia is hypervascular , so grossly it looks like a hemangioma . it can be differentiated from pulmonary sequestration , teratoma , bronchogenic cyst , and branchial cleft cyst by histology and the location of the mass . we describe the clinical , radiologic , and pathologic findings of a cutaneous lung tissue heterotopia , the first reported in korea .

when she was 4 years old , she experienced blurred vision and was operated for lens dislocation 3 years later . there was a family history of undiagnosed progressive motor disability in her older brother , which finally resulted in seizure and death . her parents had consanguineous marriage . on physical examination , she had fair and wooly hair and mild fixed oromandibular dystonia presenting as fixed smiling . on chest examination , all types of horizontal and vertical eye movements were preserved . at rest , she had dystonic posture in her limbs on both sides , with more severity on the right side her blood cell count , liver function test , thyroid function test , calcium , serum ceruloplasmin , 24-hour urine copper , and serum b12 were within the normal range . serum homocyteine was measured as 200 nmol / ml . her brain magnetic resonance imaging ( mri ) revealed deep watershed infarct in left centrum semiovale ( figure 1 ) . previous mri of lumbosacral vertebrae was suggestive of severe degenerative changes ( figure 2 ) . carotid intima media thickness ( imt ) was within normal range on both sides . the patient was treated with high dose of oral pyridoxine ( 360 mg daily ) and put on methionine - restricted diet . she showed a remarkable recovery from her limb dystonia , although nothing was completely normal . after 3 months and 9 months of treatment , serum hcy decreased to 40 nmol / ml and 26.5 nmol / ml , respectively . homocysteine , an intermediate metabolite of methionine catabolism , can be removed in 2 ways . cystathionine -synthase ( cbs ) is an enzyme that catalyzes hcy irreversibly by the means of b6 as the cofactor . , methionine is rebuilt through remethylation , by means of either methylenetetrahydrofolate reductase or methionine synthase . in the first pathway , hcy is converted into methionine in the presence of coenzyme , methylcobalamine . the reported worldwide incidence of this rare autosomal recessive disorder is between 1 in 50 000 and 1 in 200 000 . the clinical manifestation of cbs deficiency is diversely heterogenous ; however , 4 organs are dominantly affected , namely central nervous system , eye , skeletal , and vascular system . homocysinuria , due to cbs deficiency , usually manifests itself as ocular lens subluxation , which results in severe myopia and iridiodonesis . in a large number of patients , skeletal abnormalities in homocystinuria are a common manifestation similar to what is seen in marfan syndrome . these abnormalities consist of fair wooly hairs , blue eyes , livedo reticularis , limitation of joint mobility , scoliosis , high - arched palate , pes cavus , pectus excavatum or pectus carinatum , and genu valgum and osteoporosis , especially of vertebrae and long bones . central nervous system involvement includes progressive mental retardation , seizure , dystonia , behavioral and personality disorder , and stroke due to thromboembolic syndromes . vascular injury is proposed to be due to endothelial dysfunction , smooth muscle proliferation , extracellular matrix modification , lipoprotein oxidation , and increased thrombin production . endothelial dysfunction is the result of nitric oxide release impairment , thereby disturbing vasodilation and facilitating platelet aggregation . although it had been mentioned in earlier studies that hyperhomocysteinemia provokes intima hypertrophy and resultant increased intima media thickness , some of the recent observation have failed to show this correlation . similarly , in this case , no evidence of increased intima media thickness was detected on carotid doppler ; nevertheless , significant narrowing of internal carotid artery was observed unilaterally . this is in consistent with 2 studies that claimed the plasma level of hcy was higher in carotid artery stenosis , although in the latter , the serum hcy level was not correlated with pulsatility index in the stenoocclusive state of the proximal internal carotid . correlation between hcy and dystonia has been shown in several studies ; muller et al demonstrated that the serum hcy was significantly higher in primary dystonia than in control . while some believe that dystonia is due to microinfarct in the basal ganglia , others assume that neurotoxic effect of hcy can be responsible for developing dystonia . in this case , the brain mri did not reveal any abnormality in basal ganglia , however , having experienced deep white mater infarct would have predisposed this patient to manifest dystonia more vigorously . furthermore , remarkable response of dystonia to high dose of pyridoxine might be suggestive of some reversible effect of hcy , while lowering hcy did not alter arterial stenosis . early diagnosis and treatment of homocystinuria might prevent ectopic lentis and other serious complication including thrombotic events . since in half of the patients with homocystinuria 1% to 5% of cbs activity is normal , high dose of pyridoxine ( b6 ) with a dosage of 200 mg / d , can ameliorate clinical and laboratory signs significantly . even in cases with certain cbs gene mutation and nonresponsive to b6 , high dose of b6 administration ( 500 - 1000 mg daily ) is recommended . in addition to methionine restriction , betaine is the second treatment that lowers plasma hcy through hcy remethylation . finally , folate and cobalamine are other adjunctive treatments that help reducing serum hcy concentration . regarding the normal range of plasma hcy concentration between 5 and 15 nmol / ml , it is recommended that maintaining the hcy concentration to less than 11 nmol / ml will show the best result especially to those being treated shortly after birth .

inherited homocystinuria is a rare autosomal recessive aminoacidopathy which through early diagnosis can prevent its severe neurologic and vascular complications . here we report a 9-year - old girl with homocystinuria , presenting with sequential symptoms of bilateral lens dislocation , skeletal complication , and eventually dystonia from the age of 4 years . laboratory evaluation revealed severe high serum homocysteine level . although pathophysiologically unexplained , evidence of deep white matter watershed infarct along with remarkable ipsilateral carotid stenosis was detected on the contralateral side of the dystonia in the neuroimaging . treatment with high dose of pyridoxine relieved limb and gait dystonia significantly , while carotid stenosis remained unchanged . therefore , homocysteine might have both structural and irreversible effect and functional and reversible impact that could be overcome even in late stages .

a 74-year - old man was admitted due to acute abdominal pain , which had started 3 hours earlier , with a stool problem since the previous month . he had a history of hypertension , cerebral infarction and benign prostatic hyperplasia under medical treatment . physical examination revealed direct and rebound tenderness of the lower quadrants of the abdomen . computed tomography of the abdomen showed multiple air bubbles and wall thickening of the sigmoid colon with fecal materials in the pericolic space . laboratory results were within the normal ranges except for an elevated carbohydrate antigen 19 - 9 ( 88.39 u / ml ) . under the impression of panperitonitis caused by perforation of sigmoid colon cancer serosal surface of sigmoid colon showed a fibrotic torn area , which was considered to be a result of surgical detachment from jejunum . inside was an irregular cauliflower - like mucosal lesion projected into the lumen . on section , sectioning of the jejunum revealed a 3 cm - sized gray - tan and rubbery tumor mainly located in the muscular layer , extending to the perijejunal soft tissue . histologically , the jejunal wall displayed irregularly dilated cystic areas , invasive carcinoma and focal heterotopic pancreatic tissue ( fig . the foci of invasive carcinoma were composed of well - formed glands as well as individual cells and clusters , featuring well to poorly differentiated adenocarcinoma . the tumor was distributed through the muscular layer in a haphazard fashion , and infiltrated into the submucosal layer of jejunum and into the perijejunal soft tissue . the heterotopic pancreatic tissue was composed of ducts and acini without islet , intermingling with the tumor in the muscular layer of the jejunum ( fig . the papillae were composed of mucin - secreting columnar epithelial cells with extensive pyloric metaplasia and variable degrees of cytologic atypia , being consistent with ipmn ( fig . transitional areas from high - grade panin and ipmn to adenocarcinoma were noted ( fig . 5 ) . in the sigmoid colon , adenocarcinoma showing similar patterns to the jejunal tumor was present . immunohistochemically , both jejunal and colonic tumors including ipmn showed positive reactions for cytokeratin 7 , muc5ac and muc6 and negativity for cytokeratin 20 , p53 , and muc2 . by operative , pathologic and immunohistochemical findings , this adenocarcinoma was assumed to have arisen from a jejunal heterotopic pancreas via precancerous conditions ( such as ipmn and panin ) , and invaded directly into the sigmoid colon . a heterotopic pancreas is assumed to be a congenital anomaly , although its origin is not clear . separation from the main pancreatic structure during embryonic rotation and fusion of the dorsal and ventral anlagen2 is the most convincing hypothesis . warthin stated that a heterotopic pancreas is formed from lateral budding of the rudimentary pancreatic ducts as they penetrate the intestinal wall , the mass of pancreatic tissue thus formed being snared off and carried by the longitudinal growth of the intestine , either upward and downward.3 commonly reported locations of the heterotopic pancreas are the stomach ( 25 - 28.2% ) , duodenum ( 17 - 36.3% ) , and jejunum ( 15 - 21.7% ) , which are relatively near to the embryologic origin of a normal pancreas.1 the heterotopic pancreas has rarely been detected in the ileum , colon , spleen , liver , biliary tract , mesentery , lymph node , or rectum.4 - 6 most cases are discovered incidentally during other procedures ; endoscopy , surgery , or autopsy . every pathologic change occurring in an eutopic pancreas can also occur in its heterotopic counterpart , such as chronic pancreatitis , pseudocyst , abscess formation and malignant transformation.7 a malignant change in the heterotopic pancreas is uncommon . it has been reported in about 32 cases.7 appoximately half of these were ductal adenocarcinomas in the gastric heterotopia . the other cases included mucinous cystadenocarcinoma , neuroendocrine carcinoma , anaplastic carcinoma , acinar cell carcinoma , solid pseudopapillary neoplasm , and mixed acinar endocrine carcinoma arising from heterotopic pancreatic tissue in the duodenum , jejunum , spleen , esophagus , ampulla of vater , and mesocolon.7 only five cases of ipmn arising in heterotopic pancreas of stomach ( four cases ) and meckel 's diverticulum ( one case ) have been reported.8 - 12 neoplasms in jejunal heterotopic pancreas are extremely rare . three were reports of ductal adenocarcinoma , and the others were acinar cell carcinoma , borderline mucinous cystic tumor , and solid pseudopapillary tumor.13 - 17 histological determination that a carcinoma has developed from pre - existing heterotopic pancreatic tissue may be difficult . guillou et al.18 proposed minimal diagnostic criteria.18 firstly , the tumor must be found within or close to the heterotopic pancreatic tissue . secondly , the transitional area between pancreatic structures and carcinoma must be observed ( i.e. , duct - cell dysplasia and/or carcinoma in situ ) . finally , the non - neoplastic pancreatic tissue must comprise at least fully developed acini and/or ductal structures . interestingly , our case disclosed the features of varying degrees of panin and ipmn , which are known as precancerous lesions of invasive ductal adenocarcinoma . when both panin and ipmn are present in a single pancreas , differentiation between small ipmn from panin may be difficult . however , the morphological features such as relatively large duct lesion more than 5 mm in diameter , cystically dilated duct - like spaces , intraluminal papillary excrescence with fibrovascular cores in our case strongly favor the diagnosis of ipmn over panin . although muc2 is more frequently expressed in ipmns than in panins , absence of muc2 expression can not rule out an ipmn.19 this rare case showing precancerous lesions associated invasive adenocarcinoma demonstrates that a heterotopic pancreas can develop pathological changes similar to the normal pancreas . in conclusion , although most cases of heterotopic pancreas are asymptomatic and remain undiagnosed until incidentally found , malignant transformations can occur within it . when heterotopic pancreatic tissue is detected ( incidentally or not ) , efforts should be made to find any pathologic change . as precancerous conditions , such as panin and/or ipmn , arising in a heterotopic pancreas may be a warning of an associated invasive cancer , ipmn and/or panin in a heterotopic pancreas should be entirely submitted for histologic examination to search any invasive foci .

a 74-year - old man suffered from jejunal perforation and adhesion to sigmoid colon due to adenocarcinoma associated with intraductal papillary mucinous neoplasm ( ipmn ) arising in a jejunal heterotopic pancreas . the jejunal lesion showed direct extension to the sigmoid colon , which was mistaken as sigmoid colon cancer by surgeons . malignant transformation is a rare complication of a heterotopic pancreas . about half of malignancies in reported cases were ductal adenocarcinoma arising in the stomach , and the jejunal location is extremely rare . furthermore , ipmn is also uncommon finding in a heterotopic pancreas .

the incidence rate ranges from 4.5 to 20% after pneumonectomy and is 0.5% after lobectomy . the risk of developing a bronchopleural fistula is greater when surgery is performed for a malignancy and even more when radiation or chemotherapy is given [ 1 , 2 , 3 ] . until now there are no established guidelines , nor is there a consensus on how to approach the problem . besides attempts to close the fistula , preventive measures , early treatment with antibiotics , drainage of the empyema and aggressive nutritional and rehabilitative support are of major importance . yet , when a patient is inoperable , only endoscopic procedures with the application of sealants can be used to close the fistula . unfortunately , large ( > 8 mm ) or central bronchopleural fistulas are usually not eligible for such endoscopic treatment . recently , fruchter et al . and kramer et al . have described several successful cases where a novel technique has been used to close bronchopleural fistulas . they used an amplatzer septal occluder device , which has originally been developed for the endovascular closure of cardiac septal defects [ 5 , 6 , 7 ] . we present an inoperable patient with a large bronchopleural fistula , arisen after lobectomy for a stage i squamous cell carcinoma . in the absence of other therapeutic options , we decided to apply the novel endoscopic technique , using the amplatzer septal occluder device , in an attempt to close the fistula . we report on a 68-year - old woman with a history of a diffuse , large , b - cell , non - hodgkin lymphoma of the thyroid and stomach ( stage iv b ) . she underwent surgery ( resection of a thyroidal mass ) and was treated with chemotherapy after which complete remission was achieved . at that time , she also underwent tracheal stenting because of recurring aspiration related to a fistula ( postoperative ) between the oesophagus and the trachea . five years later , she was diagnosed with a squamous lung carcinoma in the right lower lobe stage ia . a lobectomy was performed and the initial postoperative course was favourable . however , 5 days later , she started suffering from marked dyspnoea . during clinical examination developing respiratory insufficiency with extensive bilateral infiltrates visible on chest x - ray prompted intubation and mechanical ventilation . bronchoscopy revealed a large ( approximately 10 mm in diameter ) bronchopleural fistula in the middle of the right bronchial stump . since the patient had a high oxygen need , she was considered inoperable . due to the size and central location of the fistula unfortunately , the patient remained septic and repeated bronchoscopy was needed to remove extensive , sticky , endobronchial secretions . since no other therapeutic option was available , this device , originally developed for the endovascular closure of cardiac septal defects , consists of two expandable disks , made of nitinol wire mesh , connected by a central waist . the device can be compressed inside a catheter . by placing the catheter inside the defect and then releasing the device from the delivering sheath , the disks can anchor the device on either side of the defect . in our case , the distal disk was first extruded and pulled back against the distal side of the defect . the procedure was successful with almost complete closure of the fistula , but because of a small persistent air leak , bioglue surgical adhesive was applied after a couple of days to obtain a complete seal of the bronchopleural fistula ( fig . bronchoscopic reassessment showed granulation tissue developing progressively around the device which caused further closure of the fistula . the patient was eventually discharged from the hospital to a rehabilitation facility several weeks after the insertion of the device . there were no remaining symptoms and ct thorax showed a good position of the device and full expansion of the lung tissue without any remaining pleural cavity ( fig . bronchopleural fistulas can occur after lobectomy or pneumonectomy . although rare , this complication is associated with high morbidity and mortality rates . its management remains a difficult matter because no scientific evidence for optimal treatment is available . reported interventions are all based on small ( often retrospective ) series or individual cases . our patient suffered from a large bronchopleural fistula , following right lower lobectomy and was considered inoperable . in the absence of any other options , a novel endoscopic technique , using an amplatzer septal occluder device , the family of the patient was informed of the fact that an off - label use of the device was being performed . up until now , endoscopic techniques have been considered to be only useful for the treatment of small , peripheral , bronchopleural fistulas , most often as a bridge to surgery in high - risk surgical patients . in this case report we illustrate that the use of an amplatzer septal occluder device can expand the importance of endoscopic techniques in the treatment of bronchopleural fistulas . in this particular case , the endobronchial application of an amplatzer septal occluder device an experimental procedure which was the only possible option in this very weak , inoperable , ventilated patient led to the closure of a very large , central , bronchopleural fistula and furthermore provided a definite cure of the fistula . studies exploring the use of this technique as an alternative to surgery even in operable patients might be of interest . this case furthermore illustrates the potential use of devices designed for other purposes in other disciplines and highlights the advantages of close collaboration between different specialities , e.g. interventional pulmonologists and cardiologists as in our patient . none of the authors has any conflicts of interest regarding the content of this article .

bronchopleural fistulas can occur as a rare but severe complication after pulmonary resection . established guidelines for the proper treatment of patients with bronchopleural fistulas do not exist . apart from attempts to close the fistula , emphasis is placed on preventive measures , early treatment with antibiotics , drainage of the empyema and aggressive nutritional and rehabilitative support . for inoperable patients , endoscopic procedures are the only therapeutic option . unfortunately , large ( > 8 mm ) or central bronchopleural fistulas are usually not suitable for such endoscopic management . recently , some groups have published a few case reports about a novel technique for the endobronchial closure of bronchopleural fistulas , using an amplatzer device , originally designed for transcatheter closure of cardiac septal defects . we applied the same technique as a life - saving treatment in a ventilated patient who was considered inoperable due to a high oxygen need . the operation was successful . the patient could be weaned from ventilation and was eventually discharged from the hospital to a rehabilitation facility several weeks after the insertion of the device . until now , endoscopic techniques have only been useful for the treatment of small , peripheral , bronchopleural fistulas and even then only as a bridge to surgery in high - risk surgical patients . in this case report , we demonstrate that the use of an amplatzer device can expand the importance of endoscopic techniques in the treatment of bronchopleural fistulas . an amplatzer device , for endobronchial closure , can indeed be administered for large and central bronchopleural fistulas . moreover , it can be considered as a definite alternative to surgery in inoperable patients .

collan et al . classified liver abnormalities into 4 types , according to their sizes and connections to the liver : ( 1 ) ectopic liver , arising outside the liver and without connection to the liver ; ( 2 ) microscopic ectopic liver , which is found occasionally in the wall of the gallbladder ; ( 3 ) large accessory liver lobe attached to the liver by a stalk , ( 4 ) and small accessory liver lobe attached to the liver . the incidence of ectopic liver has been reported to be 0.23 - 0.7% based on autopsy and laparoscopic studies . here , we present a case of ectopic hepatocellular carcinoma which was diagnosed incidentally in a patient with a history of oropharyngeal cancer . a 65-year - old man with a 50-pack - year smoking history was diagnosed with squamous cell carcinoma of the left tonsil in august 2012 . the stage of the disease was t3 n2 m0 , and he underwent concurrent chemoradiotherapy with cisplatin from august 2012 to october 2012 . subsequently , he received a head and neck examination , contrast - enhanced chest computed tomography ( ct ) , and neck ct every 3 months for the purpose of surveillance . the mass was 3.2 cm in size and showed a multilobular enhancing pattern along the peritoneal lining ( fig . 1 ) . the patient did not have any symptoms . physical examination and blood chemistry he was negative for hepatitis b virus ( hbv ) surface antigen and anti - hepatitis c virus ( hcv ) antibody . -fetoprotein ( afp ) and protein induced by vitamin k absence or antagonist ii levels were not measured . the peritoneal mass was resected and found to be a 3.8 3.2 1.2 cm pale yellowish soft tumor . microscopically , the tumor showed large polygonal cells arranged in a trabecular or nodular pattern separated by sinusoid - like vessels ( fig . immunohistochemical stains showed that the tumor cells were diffusely positive for hepatocyte antigen ( fig . 3a ) , focally positive for afp and cytokeratin , and negative for vimentin , synaptophysin , chromogranin , s-100 , epithelial membrane antigen , and cd56 . several cytokeratin - positive bile ductules were noted at the peripheral portion of the tumor ( fig . seventeen months after surgical resection , the patient is doing well , and there have been no signs of recurrence after the operation . the liver is derived from the outgrowth of the foregut endoderm , which is called the hepatic diverticulum , in the fourth week of gestation . the cephalic portion of the hepatic diverticulum lies within the septum transversum , is called the liver primordium , and gives rises to the liver . in some cases , liver tissue migrates to various organs during embryogenesis , which may explain the occurrence of ectopic liver . ectopic liver can be detected at various sites in the body , including the gallbladder , spleen , retroperitoneum , pancreas , adrenal gland , portal vein , diaphragm , thorax , gastric serosa , testis , and umbilical vein . although hemangioma , adenoma , nodular hyperplasia , and other benign lesions may arise , malignancies are more frequent in cases of ectopic liver . some reports have noted that ectopic hepatocellular carcinoma can be observed in about 7 - 30% of cases of ectopic liver . common risk factors for hepatocellular carcinoma , such as hbv or hcv infection and cirrhosis , appear to be less relevant in cases of ectopic hepatocellular carcinoma . in a review of 22 cases of ectopic hepatocellular carcinoma , arakawa et al . reported positivity for hbv surface antigen or anti - hcv antibody in only 1 case each . further , only 7 ( 32% ) of the 22 cases of ectopic hepatocellular carcinoma had developed in patients with cirrhosis in the mother liver . in contrast , the incidences of hbv infection , hcv infection , and liver cirrhosis among patients with hepatocellular carcinoma in japan are 14.51 , 75 , and 80% , respectively . some authors have suggested that the relatively high propensity to malignant transformation in the ectopic liver may relate to its incomplete functional architecture , such as for vascular or biliary drainage , which could induce a metabolic handicap and thereby facilitate carcinogenesis . in addition to being elevated in hepatocellular carcinoma of the mother liver , it is also possible for afp to be elevated in ectopic hepatocellular carcinoma . a review of the literature reported that 63% of patients with ectopic hepatocellular carcinoma showed elevated afp levels > 1,000 ng / ml . furthermore , there have been additional case reports of ectopic hepatocellular carcinoma with elevated afp since the publication of that literature review . unfortunately , we did not measure afp , and a preoperative diagnosis of hepatocellular carcinoma was not considered . it is difficult to suspect ectopic hepatocellular carcinoma as the primary diagnosis for an unknown intra - abdominal mass . indeed , ectopic hepatocellular carcinoma is a very rare disease that does not have any specific signs or symptoms . in most cases , the diagnosis has been made based on postoperative histology , rather than preoperative imaging or biopsy . when a heterogeneously enhanced intra - abdominal mass is detected , ectopic hepatocellular carcinoma should be considered , and the measurement of afp would help to establish the diagnosis . in general , most clinicians who treat cases of ectopic hepatocellular carcinoma follow the same guidelines that are applied for hepatocellular carcinoma in the mother liver . we also treat our patients according to these guidelines , and we assess afp and provide ct scans of the liver every 6 months after complete resection . thirteen case reports have been published regarding patients with ectopic hepatocellular carcinoma who were alive and well at the most recent follow - up . furthermore , two case reports have described the administration of adjuvant chemotherapy after complete resection , even though the american association for the study of liver disease practice guideline and the national comprehensive cancer network clinical practice guideline do not recommend adjuvant chemotherapy routinely . in conclusion , we have reported a case of ectopic hepatocellular carcinoma arising from the peritoneum .

a subphrenic mass was noted on a surveillance computed tomography ( ct ) scan of a 65-year - old man who had achieved complete remission of oropharyngeal cancer after concurrent chemoradiotherapy . the mass was 3.2 cm in size and showed a multilobular enhancing pattern along the peritoneal lining . the patient was negative for hepatitis b surface antigen and anti - hepatitis c virus antibody . his carcinoembryonic antigen level was within the normal range . contrast - enhanced ct revealed no mass in the liver . he underwent surgery , and a pale yellowish soft tumor measuring 3.8 3.2 1.2 cm was resected . histologically , the tumor was confirmed to be a hepatocellular carcinoma . currently , he is doing well , and has been followed up without any signs of recurrence .

foreign body ( fb ) ingestions in the adult population are frequently incidental and the majority can successfully be removed endoscopically [ 1 , 2 ] . patients with prolonged impaction ( over 12 h ) and those who ingested sharp - pointed objects have a greater risk of complications . perforations of the duodenum are relatively rare ( 510% ) and present in a more innocuous manner . in cases of duodenal fb perforation into adjacent structures , surgical retrieval is necessary . a 38-year - old male presented to the hospital with a chief complaint of abdominal pain and hematuria 1 day after eating a barbecue pulled pork sandwich . he described the pain as deep in his abdomen , sharp and non - radiating with no episodes of nausea , emesis or diarrhea . he had no abnormalities on his laboratory values besides a urinalysis demonstrating 2 + blood and 1124 rbcs / hpf . physical examination revealed point tenderness to deep palpation to the right of his umbilicus , but his abdomen was soft with no signs of peritonitis . an upright abdominal x - ray was unremarkable ; however , a ct of his abdomen demonstrated a 2-cm linear density representing the fb in the posterior third portion of the duodenum perforating into the retroperitoneum abutting the right ureter causing hydronephrosis ( figs 1 and 2 , arrow ) . two separate attempts were made at endoscopic retrieval in the first 24 h without successful visualization or removal of the fb . due to the ureteral involvement and hydronephrosis , an operative intervention was recommended . figure 1:ct scan of linear opaque fb traversing the posterior wall of the duodenum abutting the ureter . figure 2:sagittal ct scan again demonstrating the fb through the posterior wall of the duodenum onto the anterior surface of the ureter . ct scan of linear opaque fb traversing the posterior wall of the duodenum abutting the ureter . sagittal ct scan again demonstrating the fb through the posterior wall of the duodenum onto the anterior surface of the ureter . urology performed a right retrograde pyelogram demonstrating a point of obstruction in the proximal ureter with no contrast extravasation . a guide wire and stent were passed through the obstruction into the hydronephrotic right kidney without difficulty . following successful stent insertion , fluoroscopy was used to confirm the location of the fb at the junction of the second and third portion of the duodenum . a full kocher maneuver was performed and the duodenum and ureter were separated . a small , linear , fb was identified projecting from the posterior third portion of the duodenum into the ureter ( fig . 3 ) and extracted without injury ( fig . he was able to tolerate a diet and be discharged on postoperative day 4 . roughly half of all patients with fb ingestion are unaware of swallowing the object at the time of consumption . reported that 54% of patients were unaware of having swallowed a toothpick and 23% of toothpicks were localized to the duodenum . duodenal perforations do not lead to peritonitis if perforation into the retroperitoneum occurs ; however , in this location , the toothpick may migrate into adjacent retroperitoneal structures ( i.e. ureter ) . there are few other case reports of fb ingestion leading to duodenal perforation and hydronephrosis . in one case , the toothpick perforated and fixed in the duodenal wall between the second and third duodenal portions causing inflammation and hydronephrosis . in another case , the patient presented with abdominal pain and hematuria , similar to our case due to a toothpick ingestion perforating the duodenal wall into the right kidney . endoscopic extraction was unsuccessful because the toothpick had migrated out of the duodenal lumen and exploratory laparotomy with surgical removal was required . the most common site of perforation in the gastrointestinal tract is the ileum , with rates from 18 to 66% [ 3 , 5 , 8 ] . duodenal injury occurs with fb ingestion 10% of the time with a complication rate three times higher than other gastrointestinal tract locations . ideally , endoscopic retrieval of the fb is the treatment of choice for objects located in the first and second portion of the duodenum . once objects migrate distally into the third portion of the duodenum , however , patients may experience complications such as perforation into a retroperitoneal organ and surgical intervention must ensue .

foreign body ( fb ) ingestion is a relatively common reason for visits to the emergency room . if the fb is symptomatic or damaging to the patient , either endoscopic or surgical intervention should ensue . we present a case of abdominal pain and hematuria beginning 24 h after an incidental fb ingestion . initial ct imaging defined a linear opacity perforating through the posterior duodenal wall abutting the ureter causing inflammation and hydronephrosis . after two unsuccessful endoscopic attempts at retrieval , we were able to identify the object with the aid of intraoperative fluoroscopy and surgically remove the fb . the patient recovered uneventfully and was discharged home . posterior duodenal perforation by an fb may not manifest with obvious localized or systemic symptoms unless the perforation involves surrounding structures such as the aorta , vena cava or ureter . in such cases , surgical intervention is required for fb removal .

the human genome keeps revealing surprises about the structure and function of its hereditary repertoire . when the draft structure of the human genome was unveiled just over ten years ago , many scientists spoke about the ' end of genetics ' . but in reality the opposite happened , and arguably human genetics has grown at a faster rate than ever before . among the many surprises has been the discovery of a pervasive role of non - coding rnas ( ncrnas ) , including mirnas , which was a major upset for those who thought that nothing new and paradigm changing could still be unveiled . extremely short rna molecules , treated for decades as junk in northern blots and other assays , turned out to be a formidably underestimated component of the natural history of multicellular organisms . the ncrna revolution , which caught all of us unprepared , is ongoing , with the discovery of new classes such as long ncrnas . but now , after so many intensive studies and perhaps hasty assumptions , could another genetics dogma - this time about the level of variation within mirnas in the genome - be on the verge of being wiped out ? a report by dopazo and colleagues in this issue of genome medicine suggests so . in the study , the investigators analyzed the genomic sequences , or more accurately the genomic variations , from over 1,100 individuals to examine the level of variation in mirnas . the majority of sequences were collected from the 1000 genomes project , but to these data they added 60 new exome sequences from healthy individuals from southern spain who participated in the medical genome project . a total of 527 variants from 1,152 individuals were found , with the average number of variants in mirnas per person being 28 . most geneticists , when interrogated on this subject , would reply that mirnas are among the most conserved genes across species . indeed , this is the property that aided their discovery . and some will probably continue by saying that this is true within the human species . the common sense notion being that no mutations would be found in mirna genes after more than a decade of studies of many diseases . the article by dopazo and colleagues strongly challenges this view , indicating that there are enough data in the databases ( with back - up novel exome sequences ) to convince us of the need to , at the very least , become acquainted with the notion of mirna mutations . most of the variation found in the study is not within the conserved seed region : only 44 of the 527 variants were in this recognition region . yet the small , but perhaps more interesting , percentage of mirna mutations the authors identified that do lie within this region might affect mirna functions , and seem to deviate from hardy - weinberg equilibrium . this would hint at the existence of a selective pressure working against these variant alleles . calin et al . in 2005 detected a mutation in the mir-15/16 precursor associated with chronic lymphocytic leukemia ( cll ) . this mutation negatively affected the processing of the precursor to mature mirna and was not found in normal individuals . an almost identical mutation was later found in mice , where the development of cll in the new zealand black murine model was shown to be associated with a point mutation in the primary mir-15a/16 - 1 region , which in turn correlated with a decrease in mature mir-16 and mir-15a levels . mutations in ncrnas can be tricky to detect , because the changes do not produce obvious functional changes in the way that mutations in protein - coding genes do . finding mutations within such ncrnas takes persistence ; for example , a mutation affecting a ncrna was reported recently in microcephalic osteodysplastic primordial dwarfism type i ( mopd i ) , a severe developmental disorder characterized by extreme intrauterine growth retardation and multiple organ abnormalities , after many years of careful genomic investigations . viewed within this context , the article by dopazo et al . shows that we need to challenge some of the assumptions made about mirnas , which would perhaps help us to understand better how mirnas function . are the star forms ( the complementary or passenger strand of mirnas ) more subject to variability ? are many of the ' novel mirnas ' really acting as classical mirnas ? and most of all , how do mirnas , with their multiple gene target modulation , really affect cellular functions ? it is likely that the discovery of mutations in mirnas will , in the coming years , allow geneticists to answer these , and other questions . cll : chronic lymphocytic leukemia ; mirna : microrna ; ncrna : non - coding rna . sv is supported by grant ig 8588 from the " associazione italiana per la ricerca sul cancro - airc " and by a " progetti di ricerca di interesse nazionale " prin 2008 grant from the ministero dell'istruzione dell'universit e della ricerca , italy .

short rna molecules were considered to be junk for decades , but in recent years they have been shown to have important functional roles . micrornas ( mirnas ) in particular have attracted much attention . they have been assumed to be highly conserved in humans and other species ; however , a recent study published in genome medicine reveals an unexpected level of variability in human mirnas , including variations within the seed region . this challenges the current view of mirnas , and may explain previous reports of pathogenic mutations in mirnas.see research article http://genomemedicine.com/content/4/8/62/abstract

examination demonstrated a partial left oculomotor palsy with ptosis and mydriasis . facial strength and sensation were normal . 1 ) , with erosion of the petrosquamous temporal bone and sphenoid wing . magnetic resonance imaging ( mri ) of the brain demonstrated an avidly enhancing mass in the left middle fossa , with mass effect on the left temporal lobe and effacement of the lateral ventricle ( fig . 2 ) . computed tomography scan demonstrating hyperdense left middle cranial fossa mass with osteolysis . he underwent left pterional craniotomy and infratemporal fossa approach for tumor resection with autologous fat graft . the tumor was pink - tan , firm , and lobulated , with numerous calcifications . 3 ) . postoperative magnetic resonance imaging demonstrating gross total resection of left middle fossa mass . pathology demonstrated a densely cellular neoplasm with predominately spindle cell morphology in a collagen - containing stroma , focal areas of hemangiopericytoma - like vascular proliferation , and focal mineralization ( fig . immunohistochemistry for s100 , epithelial membrane antigen ( ema ) , glial fibrillary acidic protein ( gfap ) , and synaptophysin was negative . based on the final diagnosis of osteosarcoma , the patient was referred for chemotherapy and radiation . hematoxylin and eosin stain demonstrating densely cellular neoplasm with predominately spindle cell morphology , vascular proliferation , and focal mineralization . high power view of hematoxylin and eosin stain demonstrating densely cellular neoplasm with high mitotic index , areas of necrosis , and regions of cartilage formation . intracranial osteosarcoma belongs to the class of nonmeningiomatous mesenchymal tumors , which encompass benign lesions , such as lipoma , and more malignant lesions , such as hemangiopericytoma , osteosarcoma , and chondrosarcoma . these tumors are relatively rare , with sarcomas comprising between 0.2 and 2.5% of intracranial tumors.1 in contrast to long bone osteosarcoma , intracranial osteosarcoma is more common in adults . presentation depends on tumor location and may include focal neurologic deficits , cranial neuropathy , seizures , or symptoms of increased intracranial pressure . the tumors are typically isointense on t1-weighted mri and hyperintense on t2-weighted imaging.2 mri may demonstrate areas of hemorrhage or necrosis . although osteosarcoma may appear well - circumscribed on imaging , there is typically some degree of parenchymal infiltration . differential diagnosis includes hemangiopericytoma , mesenchymal chondrosarcoma , synovial sarcoma , anaplastic meningioma , gliosarcoma , melanoma , and metastatic spindle cell carcinoma . immunostaining is helpful to eliminate meningioma on the basis of ema negativity , while gfap negativity rules out astrocytic tumors . evidence of direct osteoid formation by the tumor cells is key to the proper diagnosis.3 given the relative rarity of intracranial osteosarcoma , there are no established guidelines for treatment , and therapy is guided by experience with systemic osteosarcoma . both chemotherapy and radiation therapy are used as adjuvant therapy.4 5 regardless of treatment , osteosarcoma remains a highly aggressive malignancy with a poor prognosis . median survival of 12 to 18 months has been reported.1 5 morbidity and mortality may be the result of local recurrence or development of pulmonary or skeletal metastasis .

a 21-year - old male presented for evaluation of transient loss of consciousness and was found to have a hyperdense mass in the left middle fossa . he underwent craniotomy for tumor resection . intra- and extradural invasion was noted . gross total resection was achieved . pathology demonstrated a densely cellular neoplasm with predominately spindle cell morphology in a collagen - containing stroma , areas of vascular proliferation , focal mineralization , and regions of cartilage formation . high mitotic index and regions of necrosis were seen . based on the final diagnosis of osteosarcoma , the patient was referred for chemotherapy and radiation . intracranial osteosarcoma is a nonmeningiomatous mesenchymal tumor . most osteosarcomas are meningeal - based and supratentorial . presentation depends on tumor location and may include focal neurologic deficits , cranial neuropathy , seizures , or symptoms of increased intracranial pressure . given the relative rarity of intracranial osteosarcoma , there are no established guidelines for treatment , and therapy is guided by experience with systemic osteosarcoma . gross total resection is recommended whenever feasible . both chemotherapy and radiation therapy are used as adjuvant therapy . regardless of treatment , osteosarcoma remains a highly aggressive malignancy with a poor prognosis . morbidity and mortality may be the result of local recurrence or development of pulmonary or skeletal metastasis .

patients had bilateral symmetrical peripheral neuropathy along with severe emotional disturbances , anorexia , and impotence . they had mild diabetes , simultaneous onset of neuropathy , and without presence of other specific diabetic complications . the patients were all males and in the 6 decade of life in the original report . but , it was a female patient and she was relatively young . only , few female cases have been reported in literature . because of clinical presentation , this syndrome is often confused with neuropathic carcinomatosis or an occult cancer . a 42-year - old house wife ( p2 + 0 , last child birth 24 years ago ) was admitted with complaints of generalized body ache of 1 year duration . the patient had numbness and severe burning sensation over her feet , ascending rapidly to involve her legs and thighs . it was associated with loss of weight ( approximately 8 kg ) , loss of appetite , nausea , frequent vomiting , and altered bowel habits without history of fever or pain abdomen . she was detected to have diabetic 1 year ago and put on oral hypoglycemic agents ( ohas ) . she had undergone cholecystectomy 2 months back ( histopathological examination ( hpe ) suggestive of chronic cholecystitis without malignant change ) . she was amenorrhoic for last 2 months . on admission , she was biochemically euthyroid with normal creatinine phosphokinase ( cpk ) level , negative antinatriuretic factor ( anf ) , rheumatoid factor , anti - double stranded deoxyriobonucleic acid ( dsdna ) , and poor glycemic status ( hba1c 16.9% ) . weight : 36.5 kg , body mass index ( bmi ) : 15.7 kg / m , blood pressure ( bp ) : 100/70 mmhg , pulse : 70/min , regular , all peripheral pulses palpable , pallor : + , icterus : nil , edema : nil , goiter : gr i , acanthosis : absent , muscle function test ( mft ) : 6/6 b / l , ankle brachial pressure index ( abpi ) : left 0.9 , right 1.0 . profound symmetrical wasting was noted in all extremities with associated bilateral weakness more in proximal muscle groups of the lower extremities . hemoglobin ( hb ) : 9.4 ; total leukocyte count ( tlc ) : 12,400 ; differential leukocyte count ( dlc ) : neutrophils ( n)-78 , lymphocytes ( l)-18 , monocytes ( m)-2 , eosinophils ( e)-2 ; erythrocyte sedimentation rate ( esr ) : 86 ; adequate platelet . renal and hepatic profile was normal except hypoproteinemia ( albumin / globulin ( alb / glb):2.6/2.2 ) and hypokalemia . nerve conduction velocity ( ncv ) revealed symmetrical distal axonal and demyelinating type of sensorimotor polyneuropathy involving lower limbs more than upper limbs . computed tomography ( ct ) scan abdomen suggested hepatomegaly with mildly dilated common bile duct ( cbd ) ( post cholecystectomy ) along with suspected stricture ileum along with proximal narrowing . chest x - ray , ct scan brain , ultrasonography ( usg ) abdomen , upper gastrointestinal ( gi ) endoscopy , colonoscopy , ba meal follow through , ct enteroclysis were normal . weight : 36.5 kg , body mass index ( bmi ) : 15.7 kg / m , blood pressure ( bp ) : 100/70 mmhg , pulse : 70/min , regular , all peripheral pulses palpable , pallor : + , icterus : nil , edema : nil , goiter : gr i , acanthosis : absent , muscle function test ( mft ) : 6/6 b / l , ankle brachial pressure index ( abpi ) : left 0.9 , right 1.0 . profound symmetrical wasting was noted in all extremities with associated bilateral weakness more in proximal muscle groups of the lower extremities . hemoglobin ( hb ) : 9.4 ; total leukocyte count ( tlc ) : 12,400 ; differential leukocyte count ( dlc ) : neutrophils ( n)-78 , lymphocytes ( l)-18 , monocytes ( m)-2 , eosinophils ( e)-2 ; erythrocyte sedimentation rate ( esr ) : 86 ; adequate platelet . renal and hepatic profile was normal except hypoproteinemia ( albumin / globulin ( alb / glb):2.6/2.2 ) and hypokalemia . nerve conduction velocity ( ncv ) revealed symmetrical distal axonal and demyelinating type of sensorimotor polyneuropathy involving lower limbs more than upper limbs . computed tomography ( ct ) scan abdomen suggested hepatomegaly with mildly dilated common bile duct ( cbd ) ( post cholecystectomy ) along with suspected stricture ileum along with proximal narrowing . chest x - ray , ct scan brain , ultrasonography ( usg ) abdomen , upper gastrointestinal ( gi ) endoscopy , colonoscopy , ba meal follow through , ct enteroclysis were normal . this 42-year - old known diabetic female was admitted in our department with complaints of generalized body ache with disturbed sleep , anxiety , and irritability for 1 year , which was progressively increasing in severity along with history of significant loss of weight ( total 14 kg ) , nausea , frequent vomiting , and altered bowel habits with intermittent diarrhea and constipation . she was investigated initially to exclude hematological malignancy in the background of generalized bone pain and raised esr . gi malignancy was also excluded considering significant loss of weight , anorexia , nausea , frequent vomiting , and altered bowel habits . ct scan abdomen suggested suspected stricture ileum along with proximal narrowing which was not supported by colonoscopy , ba meal follow through , and ct enteroclysis . she was in very much depressed state and lost weight ( approximately 6 kg ) during institutional management . ncv revealed symmetrical distal axonal and demyelinating type of sensorimotor polyneuropathy involving lower limbs more than upper limbs . patient was discharged in relatively stable condition with a diagnosis of diabetic neuropathic cachexia . at follow - up ( 1 month ) our patient was found to be relatively symptom free with weight gain of 1 kg . our experience highlights that diabetic neuropathic cachexia may occur in young female and responds to psychopharmachological support . though it is a diagnosis of exclusion , low threshold of suspicion will help to reach the diagnosis and reassure patient and near relatives to get rid of the problem because of overall good prognosis .

a 42-year - old lady , a known diabetic presented with generalized body ache , severe burning sensation over her lower limbs , loss of weight ( approximately 8 kg ) , loss of appetite , nausea , frequent vomiting , and altered bowel habits without history of fever or pain abdomen . symmetrical wasting was noted in all limbs with bilateral proximal muscle weakness , particularly of lower limbs . ankle jerks were absent with symmetrically decreased reflexes . nerve conduction velocity ( ncv ) revealed symmetrical distal axonal and demyelinating type of sensorimotor polyneuropathy . hematological and gastrointestinal ( gi ) malignancy were excluded . patient responded to antidepressants .

it has been used primarily by military , construction workers , fire fighters to see through smoke , and to spot heat leaks or the enemy . there are few reports and small - scale studies done in various disciplines of ophthalmology . it has been used in the diagnosis of dry eye and to evaluate bleb functionality after glaucoma surgery . it has also been used to demonstrate low ocular temperature in vascular occlusions and high temperature in ocular inflammation . this is a report on the study of thermographic images of ocular inflammatory and non - inflammatory conditions . a non - contact thermographic camera ( flir p 620 ) was used to take thermal pictures of seven cases of ocular inflammation , two cases of non - inflammatory ocular pathology , and one healthy subject with mild refractive error only . ocular inflammatory cases included five cases of scleritis ( three anterior and two posterior ) , one case of postoperative anterior uveitis , and a case of bilateral meibomian gland dysfunction with keratitis ( mgd - keratitis ) . non - inflammatory conditions included a case of conjunctival benign reactive lymphoid hyperplasia ( brlh ) and a case of central serous chorio - retinopathy ( cscr ) . thermal and non - thermal photographs were taken using same magnification and same distance from the eye at atmospheric temperature of 27c , humidity 60% , and with stable airflow . physiological blinking was allowed , no ocular medications were applied , and thermal barriers like eyeglasses or contact lenses were removed before photography . all images were transferred to a computer , and using analyzing software , average ocular surface temperature ( ost ) and temperature at the area of interest were calculated . the eye of the patient with a fresh episode of scleritis , which was not on treatment , showed hot colors on thermography [ figure 1 ] indicating higher temperature in right eye compared to fellow healthy eye . eyes with scleritis on treatment showed slightly higher temperature compared to the fellow eye . a case of resolved necrotizing scleritis showed lower temperature at the site of the necrosis surrounded by normal tissue temperature [ figure 2 ] . eye with postoperative anterior uveitis also revealed increased temperature , but paradoxically , eyes with mgd - keratitis depicted lower temperature in clinically more affected right eye ( od ) [ figure 3 ] . eye with conjunctival brlh showed lower temperature at the site of the lesion [ figure 4 ] . eyes with only refractive error but no other pathology showed no temperature difference between both the eyes . od showed higher temperature compared to the fellow healthy eye a 65-year - old female presented with necrotizing scleritis . temperature at the site of the necrosis ( arrow ) was 31.8c a 35-year - old male presented with bilateral mgd - keratitis . paradoxically , od shows low temperature as compared to os a 34-year - old male presented with conjunctival brlh . ocular thermography is a relatively newer imaging modality . attempts to calculate normal eye temperature were made way back in 1950s using contact thermometry . in 1968 , r. mapstone introduced infrared thermometry using bolometer , a non - contact method of ocular thermometry . the normal corneal temperature measured by thermography ranges from 35.4 0.1c to 34.01 0.64c , and the mean inter - ocular difference of ost in normal individuals our first patient , a fresh case of scleritis who was not on any treatment , had higher ost difference ( t ) between the two eyes as compared to the eyes on treatment for more than a week [ table 1 ] . this may suggest that ocular temperature rapidly decreases with anti - inflammatory treatment and has a positive correlation with the signs and symptoms . in this study , the case of mgd - keratitis ( case 6 ) paradoxically showed low temperature in clinically more affected eye ( od ) . rolando et al . also noted similar finding in dry eye and suggested that the ocular surface would be 0.10c lesser in dry eyes due to the increased rate of evaporation . hence , applying the same logic , it is clear that in case 6 in the present study , due to decreased tear film lipids owing to mgd , the worse eye ( od ) was subjected to rapid evaporation of the tear film which resulted in a relatively colder eye on thermography . detailed patient information and temperature difference between affected and unaffected eye thermography can also be used to distinguish between a cold lesion of non - inflammatory condition from a hot lesion of ocular inflammation . conjunctival brlh is a poorly vascularized tumor , and hence , case 8 in the present study showed low temperature at the site of the lesion . in contrast case 1 who came with similar clinical presentation showed high temperature , suggestive of inflammation due to scleritis . our cases of posterior scleritis had minimal increase in temperature as compared to the non - affected eye , as they were on treatment . this may suggest that posterior segment inflammation can also cause increase in ost , which can be picked up on thermography when posterior segment examination is difficult as in the case of mature cataract or when it is normal as in the case of mild posterior scleritis . ocular thermal imaging is an underutilized diagnostic tool which can be used to distinguish inflammatory ocular conditions from non - inflammatory conditions . it can also be utilized in the evaluation of tear film in dry eye syndrome and other corneal pathologies .

background and objectives : the purpose of this study was to evaluate ocular inflammatory and non - inflammatory conditions using commercially available thermal camera.materials and methods : a non - contact thermographic camera ( flir p 620 ) was used to take thermal pictures of seven cases of ocular inflammation , two cases of non - inflammatory ocular pathology , and one healthy subject with mild refractive error only . ocular inflammatory cases included five cases of scleritis , one case of postoperative anterior uveitis , and a case of meibomian gland dysfunction with keratitis ( mgd - keratitis ) . non - inflammatory conditions included a case of conjunctival benign reactive lymphoid hyperplasia ( brlh ) and a case of central serous chorio - retinopathy . thermal and non - thermal photographs were taken , and using analyzing software , the ocular surface temperature was calculated.results:patient with fresh episode of scleritis revealed high temperature . eyes with mgd - keratitis depicted lower temperature in clinically more affected eye . conjunctival brlh showed a cold lesion on thermography at the site of involvement , in contrast to cases of scleritis with similar clinical presentation.conclusion:ocular thermal imaging is an underutilized diagnostic tool which can be used to distinguish inflammatory ocular conditions from non - inflammatory conditions . it can also be utilized in the evaluation of tear film in dry eye syndrome . its applications should be further explored in uveitis and other ocular disorders . dedicated ocular thermographic camera is today 's need of the hour .

a notice in diabetes forecast inviting participants to evaluate bgathome.com resulted in 210 individuals completing an online screening in 10 days . participants were the first 40 individuals who , by telephone interviews , met the following inclusion criteria : type 1 diabetes , routinely measuring blood glucose levels more than twice a day , and willingness to devote 12 h / week for 810 weeks to completing bgathome . of 108 responders telephoned , 38 were unreachable , 14 were ineligible , and 10 declined participation ( table 1 ) . after signing institutional review board approved informed consent , participants were mailed a handheld computer ( hhc ) and a lifescan one - touch meter with supplies for one month 's use . participants were instructed to 1 ) activate the hhc before performing routine self monitoring of blood glucose ( smbg ) ; 2 ) enter an estimate of their current blood glucose level ; 3 ) based on this estimate , indicate whether they should then eat fast - acting carbohydrates , engage in vigorous exercise , or drive ; and 4 ) perform smbg and record their actual blood glucose levels . after returning the hhc , participants completed online a demographic questionnaire , the diabetes knowledge scale , and the hypoglycemia fear survey ( 2 ) . the hhc and questionnaire data were collected again 12 weeks later , along with likert - scale items assessing bgathome 's benefits and usability . users were given 12 weeks to complete bgathome 's units , detailed elsewhere ( 1 ) . central to bgat is completing daily blood glucose diaries , in which participants 1 ) record relevant blood glucose information and symptoms , 2 ) estimate their current blood glucose , 3 ) receive feedback on their estimate accuracy by performing and recording smbg , 4 ) interpret the clinical significance of their accuracy with the error grid ( 2 ) , and 5 ) anticipate their blood glucose level 1 h later . to encourage use of blood glucose diaries , participants were only given access to the next unit 7 days following completion of the previous unit . with internet delivery to a heterogeneous sample , individuals would be expected to pursue bgat for various reasons . thus , the primary outcome variable would need to incorporate a variety of possible desired outcomes . consequently , our improved functioning score ( ifs ) is a composite score where assessment - dependent variables are converted to z scores . assessment 2 performance was converted to z scores based on assessment 1 's mean and sd . z scores for each outcome variable were totaled , where zero reflects average baseline functioning for all variables and + 1 reflects performance across all variables one sd above the sample 's baseline mean ( 3 ) . it incorporated the following variables from questionnaires : diabetes knowledge scale ( percent correct ) and hypoglycemic fear survey ( sum of worry subscale ) . it also incorporated the following variables from the hhc : percent smbg readings within target range ( 3.9 10.0 mmol / l ) , number of undetected blood glucose readings < 3.9 mmol / l , overall blood glucose estimation accuracy ( accuracy index ) ( 4 ) , when blood glucose levels are < 3.9 mmol / l , number of risky decisions to drive , not eat fast - acting carbohydrates , and exercise . two wait - list control group participants and one bgathome participant dropped out during the treatment period . two bgathome participants dropped out during assessment 1 . anova demonstrated that bgathome resulted in greater improvement in ifs : interaction f(1,33 ) = 4.20 ; p = 0.048 ( table 1 ) . on a scale of 15 in which 1 = not at all and 5 = very , treatment participants rated bgathome as beneficial , easy to use , and enjoyable ( 3.8 1.17 , 3.9 0.73 , and 3.8 1.04 , respectively ) . on average , participants completed bgathome in 11 weeks , logged onto bgathome.com 30.4 16.51 times , and spent 26.4 16.3 min on each unit . these measures of use indicate trends toward a relationship between more website use and increased benefits . more time spent on units was associated with greater ifs improvement ( r = 0.36 , p = 0.10 ) . more frequent log - ons were associated with greater improvement in knowledge ( r = 0.49 , p = 0.03 ) and lower blood glucose levels < 50 mg / dl ( r = 0.54 , p = 0.02 ) . age was not correlated with ifs improvement ; however , education tended to be associated with improved ifs ( r = 0.45 , p = 0.07 ) . this is the first time bgat was made available to individuals with various goals , needs , diabetes regimens , and resources . despite this heterogeneity , bgathome improved performance , summed across all eight dependent variables an average of 2.37 sds . engagement might be further enhanced by 1 ) incorporating a chat room where users share experiences and support ; 2 ) employing a group context , led by a diabetes educator ( 5 ) ; 3 ) undergoing an initial motivational interview ( 6 ) ; 4 ) fiscally investing in training ; and 5 ) having a pressing personal goal , such as achieving tight metabolic control because of pregnancy without increasing risk of severe hypoglycemia ( 7 ) or following a costly hypoglycemia - related driving mishap . while our final participant sample came from 35 different u.s . cities and 21 different states , allowing greater external validity , the sample size and its demographic composition ( white , middle - aged , educated individuals ) was a limitation of this study . a larger , more representative sample would also allow investigation into the role of socioeconomic status , race , and education . nevertheless , this study indicates the possible benefits of disseminating bgathome over the internet in a personalized and self - directed format , serving a large number of individuals in a cost - effective manner .

objective blood glucose awareness training ( bgat ) , a psycho - educational intervention , trains individuals with type 1 diabetes to 1 ) detect / interpret internal cues to better detect extreme blood glucose levels , e.g. , neurogenic and neuroglycopenic symptoms ; and 2 ) interpret external cues to detect current and anticipate future extreme blood glucose levels , e.g. , insulin timing / dose and recent self - monitoring of blood glucose results . although outcome studies using bgat are significant , limitations include the requirement of eight weekly meetings and limited professionals trained to deliver bgat.research design and methods due to the limitations mentioned above , bgat was converted for web - based delivery . the internet allows bgat delivery to be dynamic , engaging , convenient , and personalized . efficacy was evaluated using a 2 ( bgathome , n = 20 , vs. control , n = 20 ) 2 ( pre / post ) design.resultsbgathome was judged as useful and easy to use , was completed by 94% of the participants , and resulted in significant clinical improvements ( p < 0.05).conclusions the internet may be an efficient and effective means of delivering diabetes interventions like bgat .

adenocarcinomas usually originate from the gastrointestinal tract , prostate gland , or other glandular tissues of the body . metastasis from gastric adenocarcinoma typically occurs in the regional lymph nodes , liver , peritoneum , omentum , lungs , and mesentery . metastasis to the prostate gland is very rare and most commonly occurs owing to direct invasion from neighboring organs or infiltration , in cases of hematological malignancies . a 56-year - old man presented with dysphagia and diffuse abdominal pain lasting for 1 year . results of general and systemic examinations were normal , except for the presence of a perirectal growth on the anterior rectal wall , inseparable from the prostate gland . computed tomography ( ct ) of the abdomen and pelvis revealed mural wall thickening , measuring 3.33.3 cm , involving the gastroesophageal junction and extending to the lesser curvature of the stomach as well as a prostatic mass ( fig . upper gastrointestinal endoscopy revealed an ulceroinfiltrative growth at the gastroesophageal junction along the lesser curvature of the stomach ( fig . histopathological analysis showed gastric mucosa with ulceration and an infiltrative neoplasm composed of cells arranged in a glandular pattern , signet ring cells , and pools of mucin . the level of serum carcinoembryonic antigen was 2 ng / ml , and that of serum prostate - specific antigen ( psa ) was 1.94 ng / ml , within the normal range . to evaluate the prostatic mass , flexible sigmoidoscopy was performed , which revealed a sessile polyp in the rectum and enlargement of the prostate gland ; biopsy specimens were taken . the biopsy specimen from the sessile polyp showed colonic mucosa with the lamina propria showing lymphoplasmacytic cells and eosinophils . tru - cut biopsy of the specimen from the prostate gland showed infiltration of the small and large glands lined by tall columnar / cuboidal mucinous cells ( fig thus , we confirmed the diagnosis of prostatic metastasis from a carcinoma of the stomach . the patient was treated with palliative chemotherapy consisting of epirubicin , oxaliplatin , and capecitabine ; however , the patient was lost to follow - up after 3 courses of chemotherapy . patients with gastric carcinoma most commonly show metastasis to the regional lymph nodes , liver , peritoneum , omentum , lungs , and mesentery . metastasis of gastric adenocarcinoma to the prostate gland is extremely rare , and only a few cases have been documented in the literature.1,2 secondary prostatic neoplasms usually arise due to direct invasion from adjacent tumors or infiltration , in cases of hematological malignancies such as leukemia or lymphoma . true metastasis to the prostate gland from solid tumors is reported only in 0.2% of all surgical prostatic specimens and in 2.9% of all post - mortem analyses in men.3 most secondary tumors in the prostate gland arise via direct spread , from either the bladder or the rectum . metastasis from nonadjacent organs is often diagnosed during autopsies ; it primarily arises from cancers of the lung , skin ( melanoma ) , gastrointestinal tract , kidney , bladder , and testes . colorectal tumors including colorectal adenocarcinoma and gastrointestinal stromal tumor may also occasionally metastasize the prostate gland . the lung is the most common primary site in cases of metastasis to the prostate gland . bates and baithun reported that , in a series of 17 prostatic metastases , the lungs and pancreas were the sites of the primary lesions in 8 and 2 cases , respectively.3 the most common presentations of secondary neoplasms of the prostate gland are prostatism , pelvic pain , and hematuria ; patients with secondary neoplasms of the prostate gland are usually symptomatic , particularly those with widely disseminated disease . occasionally , these symptoms mimic those of a primary prostatic cancer . analysis of the clinical and radiological features , pathological characteristics , and immunohistochemical localization of psa is helpful in differentiating between primary and secondary tumors of the prostate gland . the prognosis and therapy of patients with metastatic adenocarcinoma are often linked to the site of origin ; these patients are usually investigated with clinical examination , radiological analysis , and evaluation of the levels of serum tumor markers . primary adenocarcinoma of the prostate gland can be differentiated from secondary tumors involving the prostate gland by using immunohistochemistry . among prostatic markers with the greatest specificity for the prostate gland , immunohistochemical staining for psa is diagnostically helpful in distinguishing between prostatic adenocarcinomas and other neoplasms that secondarily involve the prostate gland . it is also useful in establishing a prostatic origin in cases of metastatic carcinoma with an unknown primary site.4 psa , however , is not entirely specific for prostatic adenocarcinoma as its expression has also been detected in carcinomas of the ovary and the breast , including in male breast cancer , but it is still the most commonly used prostate cancer marker . p501s is used to distinguish a tumor of prostatic origin from those of the colon and bladder . it is still regarded to be among the best - validated immunohistochemical markers for tumors of prostatic origin . the prognosis of prostatic metastasis is very poor , as secondary tumors usually occur in patients with late - stage carcinoma .

metastasis of gastric adenocarcinoma to the prostate gland is extremely rare . herein , we report a case of gastric adenocarcinoma in a 56-year - old man with prostatic metastasis diagnosed through the analysis of biopsy specimens from representative lesions in the stomach and prostate gland . immunohistochemistry of the prostatic tissue showed positive staining for cytokeratin 7 and negative staining for prostate - specific antigen ( psa ) , whereas the serum psa level was normal , confirming the diagnosis of prostatic metastasis from carcinoma of the stomach .

a 65-year - old man with a medical history of stage iii nasopharyngeal cancer , end - stage renal disease treated with dialysis , hyperthyroidism , type 2 diabetes mellitus , hypertension , atrial fibrillation , and secondary hyperparathyroidism presented at the oral medicine clinic of the college of dentistry , university of florida for dental evaluation prior to the start of radiotherapy . intraoral radiographs were taken using a cs2000 intraoral x - ray system ( carestream dental llc , atlanta , ga , usa ) with # 2 soredex optime ( soredex , charlotte , nc , usa ) photo - stimulable phosphor ( psp ) sensors at 70 kvp , 7 ma , and a 0.142-second exposure time , using a standard bitewing technique . a panoramic image was obtained using an orthopantomograph op100 d digital panoramic x - ray unit ( instrumentarium dental , tuusula , finland ) with exposure factors of 70 kvp with 12 ma for 17.6 seconds . the panoramic and bitewing radiographs revealed multiple tortuous vascular calcifications in the soft tissue of the neck and cheek bilaterally , with a dense " rail track " pattern of linear calcifications within the facial artery . based upon the radiographic presentation and the patient 's known medical history , multiple radiopaque entities were also noted in the soft tissue of the neck , more on the left than on the right , consistent with a diagnosis of carotid atherosclerosis ( figs . 1 and 2 ) . positron emission tomography / computed tomography ( pet / ct ) scans were performed using a philips gemini gxl 16 pet / ct system with 5 mm thickness . the acquired data were reviewed by a medical radiologist , and " pipe - stem " calcifications were observed in the bilateral , lingual , and facial arteries ( fig . calcifications may occur in several locations in the cardiovascular system , including the intima and media of vessels . intimal arterial calcification is associated with atherosclerosis , and vascular plaques form within the intima of the involved vessel.3 however , in mnckeberg arteriosclerosis , the calcific deposits are located entirely within the medial layer of the arterial wall and both the internal and external elastic membranes are spared.712 in a recent study , the prevalence of mnckeberg arteriosclerosis in the population was found to be 13.3% for males and 6.9% for females , and it is a well - recognized age - related phenomenon.1112 medial artery calcification can lead to vascular stiffness , resulting in increased vascular resistance , reduced compliance of the artery , and an inability to properly vasodilate in the setting of increased stress.13 medial calcinosis contributes to significant adverse cardiovascular outcomes in patients with chronic kidney disease and diabetes , where higher levels of medial artery calcification are a risk factor for amputation.14 the affected artery may not demonstrate evidence of a pulse . the exact pathogenic mechanism of medial calcinosis is not well understood . however , degenerative processes leading to the apoptosis or necrosis of medial smooth muscle cells and osteogenic processes leading to formation of bone - like structures are two distinct pathologic mechanisms that have been suggested for mnckeberg arteriosclerosis.15 meema et al.8 have suggested the possibility that two clinically and histologically different types of medial calcifications may exist . the first type is a benign , slowly progressive , essentially asymptomatic form with thin medial calcifications and little or no narrowing of the arterial lumen . in contrast , the second type is defined as a malignant , rapidly progressive form , in which massive and extensive medial calcifications may displace the internal elastica toward the lumen , causing luminal narrowing.78 mnckeberg initially described medial calcinosis as primarily affecting the arteries of the lower limbs , and occasionally affecting the peripheral arteries of the upper extremities . however , the process rarely affects the intraabdominal arteries , with the exception of the renal and splenic arteries.7 some reports in the literature have described mnckeberg arteriosclerosis . in 1977 , lachman et al.7 described the involvement of coronary , peripheral , and visceral arteries with mnckeberg calcification . a case of mnckeberg arteriosclerosis involving the aorta , pelvic , and lower limb arteries was reported by lanzer in 1998.16 the nonvascular involvement of soft tissue ( pharynx and larynx ) with mnckeberg sclerosis was reported by couri et al.6 in this report , we described a diabetic patient with end - stage renal disease on dialysis , who had advanced and previously undiagnosed mnckeberg medial calcinosis of the facial and lingual arteries . knowledge of the radiographic appearance of this calcification is clinically useful in developing a differential diagnosis . the proper interpretation of radiographic images presupposes a thorough knowledge of the anatomy , distribution , number , size , and shape of the calcifications . the calcified vessel appears as a parallel pair of thin , radiopaque lines that may have a straight course or a tortuous path , showing a pattern of blood vessels that looks like railroad tracks.511 carotid artery calcifications and phleboliths are calcifications that can be seen in the same location on a panoramic radiograph . carotid artery calcifications radiographically appear as curvilinear irregular parallel radiopacities in the soft tissues of the neck at or below the third and fourth cervical vertebrae , and inferior and lateral to the hyoid bone.1718 phleboliths can be seen on a panoramic radiograph as round or oval in shape with a homogeneously radiopaque center , giving phleboliths a " target " appearance.19 mnckeberg sclerosis is listed among the primary diseases of vessels that can be visualized on panoramic radiographs . to the best of our knowledge , our report describes the first known case of medial calcification in the facial artery on the panoramic radiograph of a diabetic patient with end - stage renal disease . soft tissue calcifications in the maxillofacial area are relatively common and can occur as the result of physiologic or pathologic mineralization , and generally correspond to radiographic findings in routine examinations , such as panoramic radiographs . a comprehensive review and thorough interpretation of all conventional and routine dental radiographs , especially beyond the region of interest , is necessary , and dental practitioners should be aware of the various calcified structures seen on panoramic radiographs , especially those associated with systemic diseases . a proper knowledge of radiographic features , however subtle they may be , assists the clinician in following up and further managing the patient , including appropriate referrals .

mnckeberg sclerosis is a disease of unknown etiology , characterized by dystrophic calcification within the arterial tunica media of the lower extremities leading to reduced arterial compliance . medial calcinosis does not obstruct the lumina of the arteries , and therefore does not lead to symptoms or signs of limb or organ ischemia . mnckeberg sclerosis most commonly occurs in aged and diabetic individuals and in patients on dialysis . mnckeberg arteriosclerosis is frequently observed in the visceral arteries , and it can occur in the head and neck region as well . this report describes a remarkable case of mnckeberg arteriosclerosis in the head and neck region as detected on dental imaging studies . to the best of our knowledge , this is the first case that has been reported in which this condition presented in the facial vasculature . the aim of this report was to define the radiographic characteristics of mnckeberg arteriosclerosis in an effort to assist health care providers in diagnosing and managing this condition .

treacher collins syndrome ( tcs , omim number 154500)[13 ] is an autosomal dominant disorder of craniofacial morphogenesis with high penetrance and variable expressivity . the essential features of this syndrome were described by treacher collins , a british ophthalmologist , in the year 1900 , but the first extensive description of the condition was given by franceschetti and klein in 1949 in which they coined the term mandibulofacial dysostosis . it is found on chromosome 5q31.3 - 32 and encodes a serine / alanine rich nucleolar phosphoprotein responsible for the craniofacial development . these include down slanting eyes with notched lower lids , sunken cheekbones and jawbones , pointed nasal prominence , broad mouth and high arched palate . they also show malformation of the auricular pinnae and conducting hearing loss and preauricular hair extension . a 20-year - old boy reported to the institute with a chief complaint of multiple carious and malaligned teeth . when the patient was examined , he revealed downward slanting of eyes , depressed zygomatic arches , sunken cheek bones , deformed external ears , coloboma of lower eyelids with deficient eyelashes medial to it , tongue - shaped extension of hairs on cheek , retruded chin giving bird - like appearance [ figures 1 and 2 ] . significant hearing loss was not observed nor did he show any signs of mental retardation . intraoral examination revealed crowding of upper and lower anterior teeth , caries with 11 , 14 , 15 , 21 , 25 , 26 , 27 , 36 , 46 and 47 . the patient also had high arched palate , anterior open bite and bifid uvula [ figures 3 and 4 ] . family history revealed that patient 's father also suffered from same condition but in milder form . antimongoloid features , coloboma of eyes , sunken cheekbones fish facies ( retruded chin ) , preauricular hair displacement , malformation of external ear bifid uvula , crowding of teeth radiographic examination revealed underdeveloped condylar and coronoid processes , hypoplastic zygomatic arches , bilateral prominent antegonial notch on mandibular angle and short rami with resultant crowding of teeth [ figures 5 and 6 ] . a noncontrast high - resolution ( 0.6 ) ct scan report of skull and face clearly demonstrated maxillofacial deformity with more prominent mandibular hypoplasia . however , the cranial vault and skull base appeared normal . it also revealed bilateral hypoplasia of external pinnae , marked hypoplasia / aplasia of bony external auditory canals , hypoplasia of middle ear cavity on either side with absence of ear ossicles [ figure 7 ] . bilateral antegonial notches , short rami underdeveloped condylar and coronoid processes , hypoplastic zygomatic arches 3d ct ( 3 dimensional computed tomography ) scan showing mandibular hypoplasia involving condylar andcoronoid processes based on clinical and radiographical findings he was diagnosed with tcs . tcs is a severe congenital disorder of craniofacial development of the head and neck region . it is characterized by numerous bilateral symmetrical developmental anomalies derived from first and second branchial arches . this was describe for the first time by thompson ( 1846 ) ; however , the syndrome is named after edward treacher collins , the english surgeon and ophthalmologist who described two cases in 1900 . franceschetti and klein in 1949 published an extensive review of the condition in which they expanded the phenotype , employing the designation mandibulo - facial dysostosis . it is caused by a mutation of the tcof1gene which exhibit linkage to the human chromosome 5q32 locus . individuals who have the tcof1 mutation have a 50% chance of passing it on to their children . it is common for mildly affected tcs patients to be diagnosed retrospectively after the birth of a more severely affected child . intriguingly , however , 60% of the cases do not appear to have a previous family history and are thought to arise as the result of a de novo mutation . tcof1 is found to encode a low complexity , serine / alanine - rich , nucleolar phosphoprotein - termed treacle . it helps in neuroepithelial survival and neural crest cell proliferation that is central to normal craniofacial development . mutations in tcof1gene leads to high degree of neuroepithelial apoptosis and consequent loss of neural crest cells . clinical features include hypoplasia of the facial bones , particularly the mandible ( 78% of cases ) and zygomatic complex ( 81% ) , considered as an extremely common feature of tcs . in severe cases , the zygomatic arches may be completely absent ( poswillo , 1975 ) . hypoplasia of the facial bones may manifest dental malocclusion , with resultant anterior open bite . the teeth may be widely spaced , malpositioned or reduced in number . in a large proportion of cases , however , this patient has unique presentation of bifid uvula which is as yet unreported in referred literature . ophthalmic abnormalities include downward slanting of the palpebral fissures ( 89% ) with colobomata or notching of the lower eyelids ( 69% ) and a paucity of lid lashes medial to the defect ( 69% ) which was seen in current case . other clinical features of tcs include alterations in the shape , size and position of the external ears , which are frequently associated with atresia of the external auditory canals and anomalies of the middle ear ossicles . radiographic analysis of the middle ears of tcs patients has revealed irregular or absent auditory ossicles with fusion between rudiments of the malleus and incus , partial absence of the stapes and oval window , or even complete absence of the middle ear and epitympanic space . consequently , bilateral conductive hearing loss is common in tcs patients , whereas mixed or sensorineural hearing loss is rare . a number of conditions exhibit phenotypic overlap with tcs , including nager and miller syndrome . nager syndrome has similar facial features to tcs , but preaxial limb abnormalities are a consistent feature of nager syndrome , unlike tcs . thumbs may be hypoplastic , aplastic , or duplicated and the radius and ulna may be fused . miller syndrome also has features in common with tcs , with the additional diagnostic feature of ectropion or out - turning of the lower lids . the cleft lip , with or without cleft palate , is more common than in tcs . the limb anomalies are postaxial , most commonly with absence or incomplete development of the fifth digital ray of all four limbs . this present case has shown almost all the features describe above for tcs except the complete development of cleft palate and hearing loss . according to the franceschetti 's classification of this syndrome present case also has shown the positive family history suggesting familial mutation transfer in tcof1gene which is seen in 40% of cases . this gene can be detected by prenatal testing in embryos . thus prenatal diagnosis and genetic counseling helps parents to make intelligent decisions regarding the pregnancy which will reduce the incidence of tcs .

treacher collins syndrome ( tcs ) is a rare autosomal dominant disorder of craniofacial development . it is a congenital malformation of first and second branchial arch which may affect the size and shape of the ears , eyelids , cheek bones , and jaws . the extent of facial deformity varies from one affected individual to another . a case of 20-year - old boy having tcs is briefly described in this article .

clinically and/or radiologically these lesions are often misinterpreted as meningioma , which may delay surgery and consequently have a deleterious impact on patient care . en plaque lesions refer to a flattened sheath of tumor , taking the shape of an underlying bone . we report a case of dural metastasis which on pre - operative imaging had the appearance of an en plaque meningioma . the primary lesion in lung was found on subsequent fluorodeoxyglucose positron emission tomography ( fdg - pet ) and chest ct scan . a 50-year - old female presented to us with headache , vision loss in the right eye and progressive left hemiparesis ( grade 4/5 ) for 1 month . mri revealed extra - axial dural - based enhancing mass all along the right hemispheric convexity dura with mass effect , simulating an en plaque meningioma [ figure 1 ] . the lesion was hypointense on t1wi , hyperintense on t2wi , and intensely enhancing on contrast administration . the atypical features were the disproportionate mass effect caused by the lesion , the highly irregular and nodular surface of the lesion and the presence of a heterogeneous interface between the lesion and the normal brain ( suggestive of micro - bleeds ) . also , on plain ct scan the bone overlying the lesion was found to be normal , without evidence of hyperostosis . ( a ) axial t1 image showing the dural - based lesion all along right convexity with mass effect . ( b ) ( c and d ) t1 post - gadolinium axial and sagittal images showing contrast enhancement of the dural - based en plaque lesion in view of rapid progression of symptoms , the patient underwent right fronto - parietal craniotomy and gross total excision of the lesion . the tumor was extra - axial though there were areas with poor arachnoid plane , suggesting invasion into brain parenchyma . whole body pet ct showed mass in the right lung apex with active lung nodules , mediastinal lymphadenopathy and metastasis in adjacent vertebral bodies [ figure 2 ] . the patient was started on adjuvant chemotherapy with irinotecan 100 mg per day but went on to develop recurrence in 8 weeks time and expired at 4 months after surgery . ( a ) pet scan showing fdg uptake in the right lung ( arrow ) . ( c ) photomicrograph showing a tumor attached to the inner surface of dura ( he , x2 ) . ( d ) tumor cells are arranged in a form of glands and trabeculae with mucin production ( he , x20 ) uncommon primary dural lesions include solitary fibrous tumors ( sft ) , hemangiopericytomas , sarcomas , chondromas , etc . occasionally , non - neoplastic lesions like tuberculomas , neurosarcoidosis , plasma cell granulomas , rosai dorfman disease , xanthomas and rheumatoid nodule may present as a dural - based lesion . primary brain tumors such as gliosarcoma may mimic a meningioma , particularly if associated with desmoplastic response from involved meninges . of the dural - based metastatic lesions , carcinomas of breast , lung and prostate typically , these tumors produce mri images with increased signals on t2-weighted images and often with an enhancing dural tail mimicking a meningioma . mr spectroscopy of metastases has been found to have different lipid signals and lactate peaks to meningiomas . however , radiological presentation of dural - based metastasis as en plaque variety is extremely rare . our patient presented with holocranial headache , right eye vision loss and progressive left hemiparesis . on retrospective evaluation after considering radiological and operative findings , the right eye vision loss was explained due to the involvement of the right optic nerve at the optic canal due to en plaque nature of the metastases . a rapid progression of symptoms should have raised our suspicion that we were dealing with a malignant entity . dural involvement in metastatic lesions may be due to direct extension of metastatic skull metastases or by hematogenous spread through venous or arterial dissemination from the primary site . histopathologic evaluation combined with a judicious immunohistochemical panel can reliably differentiate metastatic carcinomas from anaplastic meningioma , a known close differential diagnostic consideration . however , lobular growth may mimic whorls or a syncytial growth pattern , raising the possibility of a malignant meningioma . because metastatic carcinomas usually exhibit more intense cytokeratin and no s-100 protein expression , immunohistochemical analysis generally excludes meningioma . advances in chemotherapy may be altering the current incidence , but in the last decade , solitary metastasis to the dura in breast cancer occurred in approximately 8% of autopsied patients with this . by consensus the median survival in untreated patients is 4 to 6 weeks , which may increase to 4 to 6 months with aggressive treatment in some cases . the importance of awareness among oncologists , neurosurgeons , and neuropathologists that carcinomas of diverse primary sites can present with dural - based masses and radiologically mimicking meningiomas can not be overstated . metastasis should be kept in mind , even for a relatively benign looking en plaque lesion especially with rapid clinical progression .

metastatic adenocarcinoma mimicking meningioma is rare ; and any metastatic lesion masquerading as an en plaque meningioma is extremely rare . we report the case of a 50-year - old female , who presented with headache and left hemiparesis for 1 month and on imaging showed a dural - based enhancing mass along the right hemisphere . the patient was operated with a working diagnosis of en plaque meningioma . histopathology revealed metastatic adenocarcinoma . this report highlights an unusual radiological presentation of a metastatic lesion as dural based en plaque variety . metastasis should be borne in mind for any en plaque lesion with rapid clinical progression .

autoimmune pancreatitis ( aip ) is a rare disease that closely mimics pancreatic cancer ( pc ) in its presentation . it is very important for clinicians to distinguish one from the other because their treatment and prognosis are vastly different . typical radiological imaging findings , in particular observation of diffusely or segmentally narrowed main pancreatic duct ( mpd ) with an irregular wall by endoscopic retrograde cholangiopancreatography ( ercp ) , are essential for making the diagnosis of aip . on the other hand , we report a rare case of a patient with focal mass - forming aip strongly suspected of being pc because of mpd obstruction on ercp . . we will continue to make an effort to distinguish between the two disorders to prevent unnecessary surgery . a previously healthy 79-year - old man with epigastric pain was admitted to another hospital . after examination , he was diagnosed as having acute pancreatitis due to a tumor of the pancreatic tail . after treatment for pancreatitis , he was referred to our hospital for further examination and treatment of the tumor . the patient 's blood chemistry data were within normal limits except for slightly elevated serum pancreatic amylase ( 264 serum gamma globulin and total igg were normal , but igg4 was elevated ( 256 mg / dl ) . dynamic ct imaging revealed an irregular mass measuring 40 23 mm in the tail of the pancreas . the tumor was not enhanced on the arterial phase and slightly enhanced on the portal phase ( fig . mri imaging showed that the intensity decreased in the t1-weighted images of the pancreas and increased in the t2-weighted images . endoscopic ultrasonography ( eus ) revealed a hypoechoic lesion detected in the tail of the pancreas . eus - guided fine needle aspiration ( eus - fna ) , however , did not reveal any cancer cells . 18-fluorodeoxyglucose positron emission tomography ( fdg - pet ) showed hot spots of fdg uptake at the site of the pancreatic mass . ercp revealed an obstruction of the mpd at the site of the tumor ( fig . 1dynamic abdominal ct scans in arterial phase showed a low - density mass ( arrow ) measuring 40 23 mm in the tail of the pancreas ( a ) . ercp showed an obstruction of the mpd ( arrow ) at the site of the pancreatic mass ( b ) dynamic abdominal ct scans in arterial phase showed a low - density mass ( arrow ) measuring 40 23 mm in the tail of the pancreas ( a ) . ercp showed an obstruction of the mpd ( arrow ) at the site of the pancreatic mass ( b ) gross inspection of the resected specimen revealed a diffusely enlarged and firm pancreas . histologically , it was remarkable for an intense mixed inflammatory cell infiltrate predominantly composed of lymphocytes and plasma cells , and centered on the pancreatic ducts . although diagnosis of aip has improved thanks to a growing awareness of the condition and proposed diagnostic criteria,1 there remains no practical strategy to differentiate pc from aip . one must distinguish between the two disorders to prevent unnecessary surgery or delayed initiation of corticosteroid therapy . however , about 35% of patients undergoing pancreatic resection for presumed pc in fact has aip.2 kamisawa et al.3 reported that 7 of 37 ( 18.9% ) aip patients had surgery because they were misdiagnosed as having pc or bile duct cancer . in particular , it is very difficult to differentiate between fmf aip and pc . chang et al.4 reported that 8 of 26 ( 31.8% ) aip patients were fmf aip who were frequently surgically treated because differentiating fmf aip from pc was so difficult . kamisawa et al.3 also reported that 6 of 17 ( 35.3% ) fmf aip patients were surgically treated ( resection ; 3 , bypass operation ; 3 ) because pc was suspected . to obtain images of the pancreatic duct , it is necessary to use ercp , and additionally direct images taken during the operation or of specimens . kamisawa et al.3 reported that the three ercp features required for aip diagnosis were ( 1 ) a > 3-cm - long narrowed main pancreatic duct ; ( 2 ) skip lesion of the mpd ; and ( 3 ) maximal upstream mpd diameter of < 5 mm . on the other hand , features highly suggestive of pc were a pancreatic low density mass , mpd obstruction , distal pancreatic atrophy , and metastases . there have been four reports of retrospective evaluation of ercp imaging in aip patient.3,57 the frequency of mpd obstruction on ercp in aip patients was 05.9% , whereas in pc patients , it was 3560% , but only three patients with mpd obstruction have been reported . although the measurement of serum igg4 level is useful for differentiating between the two diseases , 10% of pc patients also has elevated igg4.8 moreover , there are a few reports of aip patients with concomitant pc.9,10 eus - fna is frequently used to rule out pc . however , its accuracy for pc is not perfect ( about 7090% ) because some cases of pc are accompanied by chronic inflammation and fibrosis around the mass , so a negative biopsy does not rule out cancer . diagnosis of aip by eus - fna is difficult because the specimen is too small . taken together , we can not exclude the presence of pc in many cases . further improvement of diagnostic strategies , such as core biopsy techniques , or development of new immunohistological diagnostic criteria from results of cytologic and tissue specimen analyses are needed to avoid unnecessary surgery . in conclusion , we report an extremely rare case of fmf aip mimicking pc with mpd obstruction .

introduction and backgroundautoimmune pancreatitis ( aip ) is a rare disease that closely mimics pancreatic cancer ( pc ) in its presentation . it is very important for clinicians to distinguish one from the other because their treatment and prognosis are vastly different . typical radiological imaging findings , in particular observation of diffusely or segmentally narrowed main pancreatic duct ( mpd ) with an irregular wall by endoscopic retrograde cholangiopancreatography ( ercp ) , are essential for making the diagnosis of aip . on the other hand , mpd obstruction is one of the most frequent features on ercp.case reportwe report a rare case of a patient with focal mass - forming aip strongly suspected of being pc because of mpd obstruction on ercp.conclusionit was difficult to distinguish pc from aip with current diagnostic modalities . we will continue to make an effort to distinguish between the two disorders to prevent unnecessary surgery .

we examined 55 patients with fulminant type 1 diabetes ( 49% female , median age at onset 35.0 years ) , 91 patients with classic type 1a diabetes ( 57% female , median age at onset 17.0 years ) , and 369 healthy control subjects . the criteria for the recruitment of type 1a diabetic patients were presence of diabetic ketosis at onset , duration of hyperglycemic symptoms <3 months prior to initiation of insulin therapy , and positive for at least one of the anti - islet autoantibodies . this study was approved by the appropriate ethics committees , and informed consent was obtained from all subjects . genotyping of two single nucleotide polymorphisms in the ctla4 gene , + 49g > a ( rs231775 ) and ct60 ( rs3087243 ) , was performed as reported previously ( 5 ) . serum concentration of sctla4 was measured by enzyme - linked immunosorbent assay using human soluble ctla4 ( sctla4 ) kit ( medsystems diagnostics , vienna , austria ) , according to the manufacturer 's protocol . sera from type 1 diabetic patients were obtained at disease onset and stored at 20c until use . the significance of differences in the distribution of genotypes between case and control subjects was determined by test or fisher 's exact probability test . comparisons of the sctla4 levels were made by anova with phenotypic group alone and anova with phenotypic group and ctla4 genotype . the + 49g > a variant was associated with classic type 1a diabetes but not with fulminant type 1 diabetes ( table 1 ) . in contrast , the contribution of ct60 to disease is distinct from that of + 49g > a . the frequency of the ct60aa genotype in fulminant type 1 diabetic patients was significantly higher than in control subjects ( p = 0.021 ) and type 1a diabetic patients ( p = 0.031 ) . because of the strong association of hla - dr4 in both patient groups ( 1 ) , the effect of ctla4 on type 1 diabetes susceptibility relative to hla - dr4 was also examined . among dr4-positive individuals , the frequency of the ct60aa genotype was significantly increased in patients with fulminant type 1 diabetes ( p = 0.005 ) . however , stratification of patients by the presence or absence of hla - dr4 did not affect the association between the + 49gg genotype and type 1a diabetes ( table 1 ) . it has been reported that the ct60 g allele might be associated with lower production of sctla4 mrna ( 3 ) . the mean sctla4 levels in fulminant type 1 diabetic patients ( 0.56 0.24 ng / ml [ mean sd ] , n = 36 ) was significantly lower than those in type 1a diabetic patients ( 0.94 0.87 ng / ml , n = 45 ) and control subjects ( 0.89 0.76 ng / ml , n = 23 ) ( p = 0.043 ) . a mixed - model anova using phenotypic group ( fulminant type 1 diabetes , type 1a diabetes , and control ) and ct60 genotype ( gg and ga+aa ) as factorial fixed effects revealed no differences in sctla4 levels between ct60 genotypes ( p = 0.76 ) or phenotype / genotype interactions ( p = 0.40 ) . ctla4 , which delivers inhibitory signals to t - cell activation , is expressed on the surface of activated t - cells and regulatory t - cells , and the lack of ctla4 results in uncontrolled t - cell mediated lymphoproliferative disease ( 6 ) . furthermore , ctla4 also has a significant biological role in attenuating t - cell responses in the context of an inflammatory environment , such as infection with a pathogen ( 7 ) . we showed that ctla4 is associated with an increased risk of fulminant type 1 diabetes and that its contribution is distinct from classic type 1a diabetes . as reported previously ( 5,8 ) , a significant association between classic type 1a diabetes and + 49gg and ct60gg genotype was also found in the present study . moreover , it is implicated that hla - dr4 influences the association of fulminant type 1 diabetes with the ct60aa genotype . in this study , we also revealed that serum sctla4 level in fulminant type 1 diabetic patients were significantly lower than those in type 1a diabetic patients and control subjects . although it remains unknown how sctla4 regulates t - cell activation , recombinant sctla4 inhibits t - cell proliferation in vitro . furthermore , sctla4 is constitutively expressed in nonstimulated t - cells , and its expression is downregulated after t - cell activation ( 9 ) . therefore , the decreased levels of sctla4 might indicate a lower potential of t - cell inhibition in fulminant type 1 diabetes , which might be caused by functional defects leading to reduced production of sctla4 or ongoing activation of the immune system eventually leading to decreased levels of sctla4 . in conclusion furthermore , the different contributions of ctla4 to susceptibility to fulminant and classic type 1a diabetes indicate that the underlying immune process primed -cell injury might be distinct between these subtypes of type 1 diabetes .

objective to examine the contribution of the ctla4 gene in the susceptibility to fulminant type 1 diabetes and compare it with classic type 1a diabetes.research design and methods we genotyped the + 49g > a and ct60g > a variants of the ctla4 gene in fulminant type 1 diabetic patients ( n = 55 ) , classic type 1a diabetic patients ( n = 91 ) , and healthy control subjects ( n = 369 ) . we also assessed serum levels of the soluble form of ctla4 ( sctla4).results the + 49gg and ct60gg genotypes were associated with type 1a diabetes ( p < 0.001 ) . in contrast , the ct60aa genotype , but not the + 49g > a variation , was associated with fulminant type 1 diabetes ( p < 0.05 ) , especially in patients carrying hla - dr4 ( p < 0.01 ) . serum levels of sctla4 were significantly decreased in patients with fulminant type 1 diabetes ( p < 0.05).conclusions these results suggest that ctla4 ct60 affects the genetic susceptibility to fulminant type 1 diabetes . furthermore , the contribution of ctla4 to disease susceptibility is distinct between fulminant type 1 diabetes and classic type 1a diabetes .

we present a case of an 80-year - old gentleman presenting to the emergency department with a swelling on the anterior abdominal wall in the right upper quadrant extending over the right costal margin . the swelling had developed spontaneously , was painless and increasing in size over two months . he had specifically not complained of any symptoms at all previously in this area and had no other significant medical or surgical history apart from hypertension controlled by beta blockers and thiazide diuretics . prior to admission the area had acutely become inflamed with a purulent discharge and an abscess was diagnosed . the patient showed no signs of systemic sepsis with a mildly elevated serum c - reactive protein of 55 mg / ml being the only biochemical abnormality . incision and drainage was performed and antibiotics were given . at operation a hard , smooth surfaced structure was encountered deep into the cavity , thought to be either a costal margin edge or possibly an intraperitoneal structure . ultrasonography of the area was performed postoperatively , but this was unable to define the anatomy surrounding the abscess cavity . in the postoperative period the wound continued to discharge pus , which on gram stain demonstrated leucocytes and gram - negative bacilli , culturing lactose - fermenting coliforms . from this , the possibility of a colonic component to the pathological process was raised . subsequent computerised tomography scanning of the area revealed an intraabdominal collection in front of segment iv of the liver which was extending inferiorly and pointing on to the abdominal wall where three low density structures were identified , presumed to be loculations of pus ( fig . a kocher incision was made and an inflammatory mass containing the gallbladder , an edge of the duodenum and omentum was found to be adherent to the under surface of the liver and to the anterior abdominal wall . careful dissection revealed a chronic fistula from the fundus of the gallbladder to the abdominal wall measuring around 5 mm in diameter . the gallstones were smooth , firm , brown coloured structures consistent with cholesterol - based gallstones usually encountered during elective cholecystectomy . histological examination of the removed gallbladder and adherent abdominal wall demonstrated evidence of gallstone migration both microscopically and macroscopically secondary to active chronic cholecystitis . the abdominal wall section showed inflamed fibrofatty connective tissue containing gallbladder lumen and wall demonstrating fibrosis , chronic inflammation , rotitansky aschoff sinuses and muscular hypertrophy , confirming gallstone fistulation as the underlying aetiology . the patient made an uneventful recovery and was discharged home on day 7 following the procedure with the abscess cavity wound dressed regularly , with a calcium alginate fibre dressing , and allowed to heal by secondary intention . he suffered no recurrence in the following months and was discharged from the surgical team after 12 months having made a full recovery . abdominal wall and intraperitoneal abscess formation after iatrogenic spillage of gallstones following laparoscopic cholecystectomy has been reported [ 1 , 2 , 3 ] , although this is rare . cholecystocutaneous fistulation is noted in the literature but is still extremely uncommon , with only around 22 cases described over the last two decades or so . the aetiology described usually results from spilled gallstones following laparoscopic cholecystectomy , as mentioned , or is associated with previous episodes of recurrent acute cholecystitis , although rarely other aetiologies such as malignancy have to be considered [ 6 , 7 ] . this is presumably due to the fact that one may suffer symptoms early such as those experienced with cholecystitis and , therefore , have definitive imaging and treatment before this particular eventuality is reached . this theory is also supported by the evidence that prior to 1950 , courvoisier had described 169 cases , with henry and orr adding another 36 cases , of external biliary fistulae , but subsequently relatively few cases described . the great majority of biliary fistulas occur with connection to the duodenum , colon , stomach and choledochal duct . spontaneous cholecystocutaneous fistula presenting as a subcutaneous abscess is an exceedingly rare presentation of this pathological process , having only been reported a few times [ 8 , 9 ] in the last twenty years with the patients usually having a known history of gallbladder disease . as is the case represented here , the most likely pathological process is recurrent gallbladder inflammation secondary to gallbladder calculi and chronic cholecystitis , causing adherence to the abdominal wall with eventual fistulation ( confirmed histologically ) , but what is surprising in this case is the lack of symptoms experienced by he patient prior to abscess formation . this case report highlights the need for vigilance by the clinician when investigating unusual and suspicious clinical presentations in this particular area and supports the liberal use of computerised tomography , or other modalities such as magnetic resonance cholangiopancreatography , early to gain an accurate diagnosis so that the appropriate treatment can be instituted .

abdominal wall abscess secondary to spontaneous cholecystocutaneous gallstone fistulation is an uncommon presentation of a rare pathological process . having been described relatively frequently in the 19th century , it is now much less common in the late 20th and early 21st century , probably due to earlier recognition of symptoms , better imaging and surgical treatment of biliary tract disease . here we describe a report of a case with an unusual clinical presentation of the already rare pathological disease process of spontaneous cholecystocutaneous fistula .

a male patient , 65 years of age reported to adhiparasakthi dental college and hospital outpatient department with a chief complaint of painful swelling in the lower lip for 1 month . on intraoral examination , a well - defined sessile swelling of size 1 cm 1.5 cm was noticed in the lower lip . secondary surface changes such as pseudomembrane was noticed over the swelling [ figure 1a ] , and it was tender on palpation and firm in consistency . preoperative ( a ) and postoperative ( b ) photographs of the lesional area based on the clinical findings , provisional diagnosis of traumatic fibroma was made . surgical excision of the lesion was done [ figure 1b ] under 2% lignocaine local anesthetic solution and submitted for histopathologic examination in 10% formalin fixative solution . on histopathologic examination , a layer of hyperplastic stratified squamous epithelium with bulbous rete ridges was noticed with an underlying inflamed connective tissue [ figure 2 ] . fibrinopurulent membrane replaced the epithelium at one end of the section [ figure 3 ] . the underlying connective tissue showed a wide distribution of chronic inflammatory cell infiltrate amidst which foci of acute inflammatory cells mainly neutrophils were evident . areas of mucin spillage were noticed which were surrounded by a connective tissue wall made of granulation tissue . large cells with a foamy cytoplasm resembling macrophages were seen in the areas of mucin spillage along with chronic inflammatory cells . large areas of adipose tissue were seen in the connective tissue [ figures 3 and 4 ] . based on the histopathological report , it was diagnosed as mucous extravasation phenomenon with the adjoining reactive lesion . hyperplastic epithelium , loss of acinar architecture , largely replaced by adipose tissue , and areas of mucin pooling walled off by granulation tissue wall , ( h and e stained section , 10 ) replacement of epithelium by fibrinopurulent membrane with dense inflammation underneath , ( h and e stained section , 10 ) foci of acute inflammatory cells amidst a sea of chronic inflammation , ( h and e stained section , 40 ) resolution of mucocele can happen by means of enzymatic digestion of acini by neutrophils and macrophages . in this case , mucocele was not the provisional diagnosis because it did not present as a soft fluctuant mass with a bluish tinge . instead , the lesion appears as a firm mass with yellowish surface changes which was more in favor of traumatic fibroma . as such mucocele is a benign lesion . however , in this case , repeated insult to the lesion has prevented it from autoresolution , and the persistent chronic irritation has prompted the reactionary features to set in . the repeated trauma can be attributed to the reduction in the occlusal height due to generalized attrition of teeth which makes the swelling in lower lip more vulnerable to irritation . acute episodes of trauma could be the reason for the foci of acute inflammatory cells seen amidst the sea of chronic inflammatory cells in the long - standing lesion . the replacement of epithelium by fibrinopurulent membrane with subjacent dense chronic inflammatory cells again shows the effect of body 's response to chronic irritation . loss of mucous salivary gland architecture could be attributed to the long - standing chronic inflammation . one interesting aspect of the lesion is the increased adipose tissue found in the hematoxylin and eosin section . however , the fact that with advancing age , the normal salivary glands are replaced by adipose tissue made us to consider this as a normal response to aging and thus lipoma was ruled out . the habit of chronic smoking could also be the reason for the change observed . this shows the importance of detailed case history taking along with the necessary correlation with the histopathological picture before the final diagnosis . one thing of concern is the hyperplastic epithelium with bulbous rete ridges pushing into the underlying connective tissue . it could be the reason for the parallel arrangement of fibers observed subepithelially giving the effect of a fibrous capsule . the basilar hyperplasia and acanthosis clearly show that the epithelium is making an attempt to overcome the effects deleterious associated with persistent irritation and protect the underlying structures . the basement membrane was intact indicating that there was no risk of malignancy in this patient as the lesion has been excised in toto . however , the risk of uncontrolled proliferation of epithelial cells leading to invasive carcinoma could have happened due to persistent irritation had it not been intervened at this stage . the quality of life of patient could be severely compromised if that be the case . this implies the importance of early surgical intervention and to histopathologically examine any excised tissue which will guide us to plan the postoperative follow - up schedule in the best interest of the patient . this case report is an effort to emphasize the importance of good clinicopathologic correlation and also to understand the basic concept as how the body 's response to chronic irritation can transform a trivial benign lesion to reactive lesion and possibly to malignancy . the vigilant role of a dentist in the handling of a lesion is reinforced through our case report .

oral mucocele is the most common benign lesion of minor salivary gland caused due to any form of mechanical trauma to the excretory duct of the gland . it is of two types mucous extravasation phenomenon and mucous retention type . extravasation type is more common . here , we report a case of a 65-year - old male patient with a complaint of painful swelling in the lower lip for 1 month with a history of trauma in the lower lip . it was clinically diagnosed as traumatic fibroma , and an excisional biopsy was done . however , the histopathology was basically that of mucous extravasation phenomenon coexisting with the features of a reactive lesion implicating the potential role of chronic irritation in the impairment of normal healing process . considering the fact that long - standing chronic inflammation in a benign lesion can even lead to malignant transformation of that lesion ; here , we highlight the role of a good clinicopathologic correlation and the significance of prompt intervention and treatment . furthermore , emphasizing the potential need for postoperative follow - up by the dentist , how much ever trivial the lesion may appear to be in the best interest of the patient .

esophageal - pleural fistula ( epf ) is a rare entity which is formed secondary to esophageal instrumentation , surgery , malignancy , or as a rarer complication of postpneumonectomy.[16 ] spontaneous development of fistula between esophagus and pleura is very rare . contrast - enhanced computed tomography ( ct ) is the very useful imaging technique to evaluate esophagopleural fistula . a 40-year - old female presented with severe retrosternal chest pain with vomiting for four days . it was noncolicky , radiating to back , and there was no aggravating or relieving factors . there was no history of the upper gastrointestinal endoscopy or esophageal instrumentation . on chest examination , chest ct [ figure 1 ] showed linear fistulous tract from right lateral aspect of midthoracic esophagus to the right pleural cavity suggestive of epf . there was right hydropneumothorax with chest tube in situ and collapse - consolidation of the adjoining right lower lobe . in addition , there was volume loss on right hemithorax as compared to left side with associated pleural thickening . esophagography [ figure 2 ] with nonionic contrast was done to demonstrate the exact site and size of fistula , showed approximately 2 cm right epf at the level of junction of mid and lower thoracic esophagus . upper gi endoscopy was also done to rule out any associated esophageal inflammation or malignancy . hence , with the final diagnosis as spontaneous epf , she was managed conservatively keeping the chest tube drainage and performing feeding jejunostomy . definite surgery could not be performed as the patient lost to follow - up even after six months . axial sections of chest ct showing right hydropneumothorax with collapse - consolidation of the adjoining right lower lobe and chest tube in situ . linear fistulous tract ( arrow ) is seen from mid - thoracic esophagus to the right pleural cavity , which is suggestive of esophageal - pleural fistula esophagogram done with nonionic contrast showing approximately 2 cm right epf ( arrow ) at level of junction of mid and lower thoracic esophagus . esophageal injury may be caused by iatrogenic trauma ( esophageal instrumentation or external trauma ) , diseases of the esophagus such as corrosive esophagitis , esophageal ulcer and neoplasm , and rarely spontaneously.[16 ] spontaneous rupture of esophagus or boerhaave 's syndrome usually results from sudden rise of intraesophageal pressure ( due to contraction of cricopharyngeus muscle and closing of pyloric sphincter ) associated with forceful vomiting or retching . mallory - weiss tears are mucosal tears caused by forceful or long - term vomiting , retching , or coughing . epf is an uncommon complication of iatrogenic trauma ( endoscopic instrumentation ) or postpneumonectomy.[16 ] the postpneumonectomy epf are caused by surgical injury , local cancer recurrence , and chronic inflammation or infection . the possibility of direct epf and extent of mediastinitis are determined by anatomic relationship of esophagus to the pleura , the amount of mediastinal fat and intervening connective tissue . esophagus injury should be considered when patient presents with retrosternal chest pain , fever , dysphasia , and dyspnea , especially when the patient gives antecedent history of esophageal instrumentation or surgery . the radiological signs of the epf depend upon site , duration , and severity of perforation ; and more importantly , the integrity of pleura . if the pleura remain intact , mediastinitis sets in with subsequent occurrence of rupture of mediastinal pleura , pneumothorax , or hydrothorax . if the pleura is not intact , epf can occur with resultant decompression of esophageal contents into the pleura and thus mediastinum may not be involved . the diagnosis of esophageal rupture / epf is made clinically ; however , for confirmation , the imaging is required . the imaging modalities include chest radiograph , ultrasound , barium swallow , contrast - enhanced ct , and mri with each modality having its advantages , and chest ct is very useful modality . the findings on the chest radiograph are nonspecific and include pleural effusion , pneumothorax , or hydropneumothorax . pneumomediastinum or subcutaneous emphysema is seen late in the course of the disease when pleural pressure exceeds mediastinal pressure . ct shows the exact extent of mediastinum involvement , confirms the x - ray findings , and can differentiate pleural from pulmonary disease . other nonspecific ct findings include focal thickening or ballooning and thinning of the esophageal wall at the site of perforation . rarely small rent along the esophageal wall can be seen communicating with diseased pleural cavity . contrast medium , if given orally , seen in the pleural space is pathognomonic sign of the entity . ct also helps in the management of the epf as its prognosis depends upon the extent of the mediastinal involvement . epf without mediastinal involvement usually carries good prognosis and may respond to percutaneous pleural drainage . esophagographic studies may confirm the presence of the epf and demonstrate its site , although rarely indicated in this ct era . management of the epf depends on site , size , duration , and severity of perforation . another important prognostic factor is extent of mediastinal involvement , which is better evaluated by chest ct . conservative therapy includes drainage of the empyema , local irrigation , tube feeding , gastrostomy , or jejunostomy . it is followed by definite surgery which includes repair or direct reconstruction of the esophagus . the differential causes of this nonspecific presentation include aspiration pneumonia , spontaneous pneumothorax , pulmonary emboli , myocardial infarction , aortic dissection , or intra - abdominal abnormalities such as cholecystitis , and gastritis . chest ct showed epf with collapse - consolidation of right lower lobe and pleural thickening which may be attributed to infection of the airspace or pleural cavity . it should be done in patients with pleural effusion presenting with nonspecific clinical symptoms before any intervention or drainage .

spontaneous esophageal - pleural fistula ( epf ) is a rare entity . we describe a case in a middle - aged female who presented with severe retrosternal chest pain and shortness of breadth . chest computed tomography showed right epf and hydropneumothorax . she was managed conservatively keeping the chest tube drainage and performing feeding jejunostomy . a brief review of the imaging finding and management of epf is discussed .

we analyzed the patients with prion diseases who had been registered by the cjd surveillance committee in japan from april 1999 through march 2005 . we prospectively investigated each patient with a surveillance protocol that assembled information about life history , previous medical history , clinical history , laboratory data , and results of molecular genetic and pathologic analyses . written consent , approved by the institutional ethics committee , was obtained from all the patients families ; members of the surveillance committee examined the patients and collected the data . we classified the patients into 4 categories : scjd , infectious prion diseases , inherited prion diseases , and unclassified prion diseases . infectious prion diseases included cjd associated with cadaveric dura mater graft ( dcjd ) or other iatrogenic opportunities for prion infection , in which the criteria for scjd were applied for the diagnosis , and vcjd , in which the diagnosis was based on who criteria ( 2001 ) ( 11 ) . regarding the accuracy of the diagnosis of inherited prion diseases , cases verified by pathology report were defined as definite , and cases with mutations in the prion protein gene and neuropsychiatric manifestations compatible with prion diseases were defined as probable . among patients with a history of ophthalmic surgery , we directed special attention to the patients who had a history of eye surgery within 1 month before the obvious onset of prion disease or after the onset . because the onset of prion diseases often overlaps with various kinds of prodromal symptoms , determining the precise time point of onset is difficult ; therefore , we included the period of 1 month before the obvious onset . to gather information about the ophthalmic surgery , we mailed questionnaires to the ophthalmologists who operated on these patients , requesting the following information : diagnosis of ophthalmologic diseases , surgical procedures performed , changes in the symptoms after the surgery , whether the instruments were reused , and methods of cleaning reused instruments . to ascertain the clinical features of prion diseases , we analyzed the patient s age at onset and duration of disease course , which was calculated as the interval between the onset and the appearance of the akinetic mutism state or death in the patients who died without akinetic mutism . among early clinical manifestations of prion diseases , dementia and visual disturbance are major determinants that would influence the indication for ophthalmic surgery , so we grouped the patients according to whether they had sought treatment for dementia or visual impairment within 2 months after onset of symptoms . the sex distribution of the patients who had ophthalmic surgery around the time of onset of clinical symptoms and those who did not was compared by fisher exact tests , and differences in age at onset and disease duration were compared by mann - whitney u tests . we used tests to compare the distribution of the patients with or without dementia or visual impairment within 2 months of onset . we found 597 patients with definite or probable diagnosis of prion diseases : 468 ( 78.4% ) with scjd ; 78 ( 13.1% ) with inherited prion diseases ; 48 ( 8.0% ) with infectious prion diseases , including 47 cases of dcjd ; and 1 patient with vcjd and 3 patients with unclassified cjd . thirty - seven patients ( 6.2% ) had a history of ophthalmic surgery at some time in their lives . among them , 11 patients ( 1.8% ) underwent ophthalmic surgery within 1 month before the obvious onset of prion disease or after the onset . except for 1 patient with gerstmann - strussler - scheinker disease , there have been no reports of the development of prion diseases in patients who underwent ophthalmic surgery after the ophthalmic surgery of patients with prion diseases . ten patients with scjd underwent ophthalmic surgery within 14 months of symptom onset , and 8 of them had onset within 4 months of surgery ( table 1 ) . at clinical onset , 4 patients exhibited visual symptoms , 5 had dementia , and 1 patient had a gait disturbance . all patients underwent surgery for cataracts , except for 1 patient who underwent surgery for a detached retina . according to the reports on the surgical outcome by the ophthalmologists of 7 patients , visual disturbance was unchanged in 2 patients , deteriorated in 1 , and improved to some extent in 4 after surgery . all ophthalmologists reused some surgical instruments and cleaned instruments by either autoclaving or the ethylene oxide gas method , which have been reported to incompletely sterilize prp ( 9,12 ) . * scjd , sporadic creutzfeldt - jakob disease ; visual , visual impairment ; na , not available ; eog , ethylene oxide gas . disease duration , the duration from onset to akinetic mutism state or death if the patients never displayed akinetic mutism . clinical features were compared between scjd patients who did and did not have ophthalmic surgery ( table 2 ) . the patients who had ophthalmic surgery had a significantly longer disease duration than those without ( p = 0.0004 ) . among those who had early clinical symptoms within 2 months after onset , the subgroup with visual symptoms without dementia who had ophthalmic surgery was significantly more likely to have had early symptoms than those who did not have surgery ( p = 0.0004 ) . * scjd , sporadic creutzfeldt - jakob disease ; sd , standard deviation ; + , with ; , without . disease duration , the duration from onset to akinetic mutism or death if patients never displayed akinetic mutism . our study showed that , in 1.8% of the patients with prion diseases , eye tissues were operated on within 1 month before the obvious onset of prion disease or after the onset . in addition , the scjd patients who underwent surgery had a significantly longer duration of the disease course as well as significant overrepresentation of visual symptoms without dementia in the early phase , compared with patients who did not have ophthalmic surgery . the prevalence of ophthalmic surgery around the time of clinical onset of prion diseases in our study is similar to that ( 2.0% ) in a report from the united kingdom ( 13 ) . in the uk study ( 13 ) , patients with heidenhain variant cases constituted 40% of scjd patients who had ophthalmic surgery . early visual impairment ( due to prion diseases ) would prompt ophthalmologists to perform surgery . currently , cataract surgery is recommended to improve physical or cognitive function in elderly patients ( 14,15 ) . it should be noted that , after performing eye surgery on patients with prion disease , all ophthalmologists reused surgical instruments that were sterilized with procedures that are incomplete for the sterilization of prp , although the who infection control guidelines for prion diseases ( 9 ) strongly recommend single - use surgical instruments for procedures involving highly infective tissues . the fact that no secondary iatrogenic cases that could be attributed to surgical procedures were found during our investigation does not diminish the need for ophthalmologists to be aware of cjd as a cause of visual symptoms ( including symptoms mimicking those of cataracts ) and highlight the importance of using disposable instruments whenever possible to avoid cross - contamination .

eleven ( 1.8% ) of 597 patients underwent ophthalmic surgery within 1 month before the onset of prion disease or after the onset . all ophthalmologists reused surgical instruments that had been incompletely sterilized to eliminate infectious prion protein . ophthalmologists should be aware of prion diseases as a possible cause of visual symptoms and use disposable instruments whenever possible .

pyogenic granuloma ( pg ) is benign , nonneoplastic , and the most commonly found lesion in the oral cavity . pgs can be found in all age groups ; however , females are 2 times more frequently affected than males . traumatic injuries , local irritations , hormonal factors , certain kinds of drugs , and even some vascular morphogenesis factors have been associated with pg . an excisional biopsy should be performed with a safe distance of 1 mm apart from the borders of the lesion , due to the possibility of recurrence and possible irritants and local factors must be removed . however , total removal of a lesion with healthy adjacent tissue by an excisional biopsy can lead to mucogingival problems such as gingival recession and disharmonic gingival margins . however , it may result in hypersensitivity and esthetical problems . in this case report , the treatment of a gingival recession defect resulting from an excisional biopsy by a free gingival grafting procedure performed in the same appointment and its 12-year follow - up were presented . a 44-year - old female patient applied to the periodontology clinic at ondokuz mays university with a complaint of a gingival enlargement on the labial surface of lower incisors [ figure 1a ] . the patient had noticed the enlargement for nearly 4 months previous to the appointment and was concerned about its gradually increasing size . the clinical examination revealed a firm , pedunculated , red gingival enlargement located on the vestibular surfaces of tooth # 41 and # 42 . the lesion 's diameter was approximately 1 cm , and bleeding was easily caused by the mechanical stimulus . she was nonsmoker who had a good general and dental health but with poor plaque control . there were no evident signs of pathology as evaluated by a periapical radiograph and orthopantomography . treatment options were explained to the patient , and an excisional biopsy and free gingival graft surgery were scheduled . before surgery , ( a ) preoperative clinical view of lesion , ( b ) clinical view after excisional biopsy ( c ) free gingival graft placed on biopsy site following local anesthesia , the lesion was removed with a safe distance of approximately 1 mm apart from the healthy tissue to reduce the recurrence . following surgical excision , a mucogingival defect with exposed root surfaces root planing was performed , and a free gingival graft obtained from the palatal premolar region was stabilized with 4/0 silk sutures [ figure 1c ] . no periodontal dressing was used . the tissue specimen was fixed in formalin and sent for pathological investigation and it diagnosed as pg . the healing was uneventful , and the sutures were removed at 10 days . in the early postoperative phase , no recurrence was observed , but partial root coverage was achieved . at the 6-month follow - up , root coverage resulting from creeping attachment was observed , and this situation was maintained throughout the 12-year follow - up period [ figure 2a d ] . the pocket depths measured as 12 mm from the mid - labial surfaces of teeth # 41 and # 42 at 1-month were maintained until the 12-year follow - up control . postoperative clinical views at ( a ) 1 month ( b ) 6 months , ( c ) 2 years , ( d ) 12 years a recurrence rate of between 16% and 21% has been reported for pgs with conventional excision techniques . one of the most important predisposing factors for recurrence is an inadequate excision . in our case , no recurrence occurred through 12 years of follow - up . in the light of the literature , an excision technique in which the lesion was removed with a safe distance of approximately 1 mm apart from the healthy tissue , and the removal of the remnants at the base of the original lesion , might have played key roles in this outcome . surgical excision of the lesion often results in a large amount of bone exposure because of the difficulty in obtaining primary closure of the wound area , a situation that may subsequently lead to bone resorption . postoperative soft tissue recession may cause exposure of the root surface with esthetic problems , possible root sensitivity , and caries . for this reason , the present case required a treatment that included a periodontal plastic surgery procedure . several different soft tissue surgical techniques may be suggested to treat similar lesions , including coronally positioned flaps and pedicle flaps with or without connective tissue grafts , as well as free keratinized tissue grafts . in the present case , especially in view of the relatively narrow band of keratinized tissue apical to the biopsy defect , we avoided performing coronally positioned flap . a laterally positioned flap was also could not be performed due to the extension of the biopsy area to the gingiva of the neighboring teeth , and the difficulty in covering two exposed root surfaces ; in addition , a laterally positioned flap could have involved the risk of gingival recession if the facial bone at the donor site has a fenestration or dehiscence . although the free keratinized tissue graft has the advantage of restoring an adequate amount of keratinized tissue width and thickness , coupled with root coverage , the white color of the gingiva may be disadvantageous . were the first to describe the term creeping attachment as the postoperative migration of the gingival margin in a coronal direction over a previously denuded root . based on the research of borghetti and gardella , creeping attachment is any root coverage that occurs postoperatively after 1 month , with the mandibular anterior teeth best showing this condition with their narrow recessions . the average coverage is approximately 1 mm , which can be detected 112 months after graft surgery . the creeping attachment improved the results in our case , and the exposed root surfaces seen at 1-month were covered with creeping attachment at 6 months , which was maintained after 12 years . in this particular case , a recession defect and a shallow vestibule sulcus although defects following excisional surgery can be treated at a later stage , this may result in postoperative discomfort and emotional stress for the patient . the two dental procedures performed ( biopsy and free gingival graft ) in a single appointment are timesaving both for the patient and the clinician . our technique of eliminating the pg seemed to offer good esthetic results , with a stable and healthy gingival situation without recurrence at the 12-year follow - up .

the total removal of a lesion via excisional biopsy causes gingival recession , resulting in dentin hypersensitivity and esthetical problems . in this case report , a gingival recession defect resulting from an excisional biopsy was treated with a free gingival grafting procedure performed during the same appointment , and its 12-year follow - up was presented . a 44-year - old female patient was presented to our clinic with a firm , pedunculated , red gingival enlargement located on the labial surface of lower incisors . the exposed root surface , after the excisional biopsy , was covered with a free gingival graft . the lesion was pathologically diagnosed as pyogenic granuloma , and in the early postoperative phase , no recurrence was observed , but partial root coverage was determined . at 6-month follow - up , root coverage resulting from creeping attachment was observed , and this situation was maintained throughout the 12-year follow - up period . repetitious postoperative discomfort and emotional stress for the patient may be avoided with a timesaving single appointment performing excisional biopsy and free gingival graft . free gingival grafting procedure was used for this purpose not only to cover exposed root surfaces but also to eliminate dentin hypersensitivity and make oral hygiene procedures more effective .

the use of 18-fluorodeoxyglucose positron emission tomography - computed tomography ( 18-fdg pet / ct ) in the field of oncology is well - established ; however , 18f - fdg is not tumor specific . aside from physiological uptake fdg knowledge of these fdg - avid non - malignant lesions is essential for accurate pet interpretation . also this non - specificity of fdg uptake is being successfully used in evaluating infective , inflammatory diseases and pyrexia of unknown origin . we describe use of fdg pet / ct for evaluation xanthogranulomatous pyelonephritis ( xgnp ) , a benign , chronic suppurative renal inflammation . a 60-year - old indian man underwent 18-fdg pet / ct for evaluation of pyrexia and weight loss of 9 kg in 2 months . his blood examination revealed anemia , leukocytosis , and high erythrocyte sedimentation rate . patient 's serum creatinine was found to be raised ( 1.9 mg% , normal up to 1.4 mg% ) . he was found to have multiple calculi in left kidney on ultrasonography ( usg ) . usg also demonstrated multiple hypo echoic masses ; thinned out parenchyma ; and a dilated collecting system suggesting a chronically obstructed kidney . suspicion of pyelonephritis was raised in the usg report and rest of the abdominal organs were unremarkable . antibiotic therapy was instituted for treatment of the urinary tract infection . even after extensive antibiotic therapy , the fever remained unresolved and weight loss continued . before invasive diagnosis , patient was referred for a whole body 18-fdg pet / ct to confirm kidney as the sole site of active disease and rule out any extra - renal pathology as the cause of symptoms . for pet / ct , 10 mci of fdg was injected intravenously to patient after 6 h of fasting . whole body ct scan was obtained as part of pet / ct protocol on a multi - slice ct with 3.5 mm slice thickness without contrast injection . standardized uptake value ( suv ) of fdg was normalized to body weight obtained over lesions . the maximum intensity projection ( mip ) image [ figures 1a and b , arrows ] revealed intense fdg uptake in cortex of grossly enlarged hydronephrotic left kidney . multiple calculi were seen in pelviureteric junction and in pelvi - calyceal system [ figure 1c red arrows ] . extensive perinephric fat stranding and mild fdg uptake in perinephric , retroperitoneal lymph nodes was seen with suv max of 4.1 [ figure 1c and d white arrows ] . in addition to these findings diffuse fdg uptake was seen in bone marrow [ black arrow in figure 1a ] . post - nephrectomy , patient 's fever resolved and he reported gradual weight gain over last 4 months . the maximum intensity projection image [ figure 1a and b , arrows ] revealed intense fluorodeoxyglucose ( fdg ) uptake in cortex of grossly enlarged , hydronephrotic left kidney . standardized uptake value ( suv ) max of uptake was 14.1 . multiple calculi were seen in pelviureteric junction and in pelvi - calyceal system [ figure 1c red arrows ] . extensive perinephric fat stranding and mild fdg uptake in perinephric , retroperitoneal lymph nodes was seen with suv max of 4.1 [ figure 1c and d white arrows ] . the findings were reported as possible xanthogranulomatous pyelonephritis . this diagnosis was confirmed post - nephrectomy xgnp is an uncommon condition characterized by chronic suppurative renal inflammation that leads to progressive parenchymal destruction . the disease process is characterized by the destruction and replacement of renal parenchyma by lipid - laden macrophages ( xanthoma cells ) . the patients present with recurrent urinary tract infections , flank pain , hematuria , and occasionally sepsis and weight loss . the main predisposing factor appears to be obstruction , and an obstructing renal calculus is present in up to 75% of cases . it is postulated that renal obstruction promotes recurrent renal tract infections that lead to an abnormal immune response causing parenchymal destruction . whereas the first - line treatment of choice for both , localized and diffuse xgnps , is conservative ( appropriate antibiotic treatment ) , the approach to surgical treatment is different : nephron - sparing surgery is warranted in focal xgnp , but diffuse xgnp will require nephrectomy with resection of all other involved tissue in most cases . early diagnosis and prompt treatment play a crucial role in minimizing the morbidity and mortality rates of xgnp . multiple areas of low attenuation are visualized within the kidney , representing dilated renal calyces and pus - filled cavities replacing destroyed renal parenchyma . ct is also very useful in determining the extent of the inflammatory process in perirenal tissues including the iliopsoas muscle , abdominal wall , diaphragm , skin , and bowel . though xgnp can be suspected on ct findings alone , with rising use of pet / ct in evaluation of pyrexic states , added knowledge of pet findings can be advantageous to add certainty to the diagnosis . scarce data is available on fdg pet / ct findings in xgnp , in form of case reports . ct findings characteristic of xgnp along with diffuse fdg uptake in kidney and mild fdg uptake in retroperitoneal lymph nodes is seen in the diffuse xgnp . mild fdg uptake in retroperitoneal lymph nodes and extensive perinephric fat stranding are suggestive of benign inflammatory pathology in such cases . however it should be kept in mind that focal xgnp can mimic pathologies such as pyelonephritis , tuberculosis , renal abscess , renal cell carcinoma , and renal metastasis on pet / ct . thorough clinical history , and correlation with anatomical imaging are essential components to arrive at diagnosis of xgnp . to conclude , we present fdg pet / ct findings in xgnp . because , use of fdg pet / ct for evaluation of infective and inflammatory conditions is on the rise and such condition can co - exist in patients with known malignancy , this possible diagnosis should be kept in mind , when similar uptake pattern and ct findings are encountered in practice .

xanthogranulomatous pyelonephritis ( xgnp ) is an uncommon condition characterized by chronic suppurative renal inflammation that leads to progressive parenchymal destruction . this condition can clinically present as recurrent urinary tract infections , flank pain , hematuria , and occasionally sepsis , and weight loss . this condition is usually associated with obstructing renal calculus . we present 18-fluorodeoxyglucose positron emission tomography - computed tomography ( 18-fdg pet / ct ) findings in an elderly male suffering from pyrexia and weight loss and suspected urinary tract infection . pet / ct findings in this case lead to diagnosis of xgnp . this diagnosis should be kept in mind while evaluating similar symptoms and pet / ct scan findings .

tracheal extubation of patients is still a major challenge , with the possibility of post - extubation stridor and then re - intubation if the patient is unable to sustain the increase in respiratory work . stridor is responsible for 1538% of extubation failures [ 1 - 3 ] and for close to 38% of early extubation failures . recognition of stridor is important because these patients can benefit from close monitoring and from specific therapies including non - invasive respiratory assistance , aerosolized adrenaline ( epinephrine ) , and steroids ( even though the efficacy of steroids remains under debate ) . ideally , patients at risk of developing laryngeal edema should be identified as early as possible , and the cuff - leak test has been proposed for this purpose . the principle of this test is quite simple and is based on the fact that the air leak around a tracheal tube with a cuff deflated will be inversely related to the degree of laryngeal obstruction generated by laryngeal edema . the cuff - leak test was developed initially in children with croup ; extubation was likely to be successful if an air leak could be heard when the baby coughed during positive pressure ventilation . the test was further refined to allow quantitative measurements , using the difference between the expired tidal volume with the cuff inflated and with the cuff deflated : the higher the leak , the lower the likelihood that post - extubation stridor will occur . the discrimination power of the test is highly variable ( table 1 ) , depending on the population investigated , the incidence of post - extubation stridor ( ranging from to 4% to 38% ) , the method of determination of cuff leak ( absolute value versus value relative to tidal volume measured with an inflated cuff , number of measurements of tidal volumes averaged , and so on ) . but perhaps more importantly , the cut - off value should be adapted to the situation ; the cut - off that is usually given in most studies assumes an equivalent impact of false positive and false negative values . however , in clinical practice , both may not have equivalent weight . in some cases , a policy of minimizing the risk of false negatives and thus accepting a lower specificity may be preferred . this policy minimizes the risk of extubation failure and may be preferred in patients in whom tracheal intubation is difficult . on the other hand , a policy minimizing the risk of false positives , and thus less sensitive , may be preferred if one wishes to minimize the risk of unnecessary prolonged intubation . in any case , a low cuff - leak should never be used to preclude extubation because the specificity of the test is still low , even when the policy favoring minimizing false negatives is chosen so that the test can be used mainly to characterize patients at risk of developing post - extubation stridor . in this issue , prinianakis and colleagues shed some new light on the factors that might affect the leak , and hence the evaluation of the cuff - leak test . first , they separate the inspiratory and expiratory components of the leak . usually , the leak is calculated by measuring five or more tidal volumes after deflation of the cuff . of course , the inspired tidal volume effectively reaching the alveoli will also decrease so that the tidal volume measured with the cuff deflated is influenced by both inspiratory and expiratory leaks . prinianakis and colleagues developed a method that limits the influence of the inspiratory leak . after an inspiratory pause , the cuff is deflated and the subsequent expired tidal volume is measured . the two methods were compared in 15 patients and the expiratory leak was consistently lower than the total leak . as the ratio between expiratory and total leak was constant ( around one - third to one - quarter of the total leak ) whatever the value of the leak , this suggests that factors affecting expiratory leak would similarly affect inspiratory leak , and thus total leak . in the second part of the study , the authors used a lung model to evaluate the factors influencing the expiratory leak ( and thus probably similarly the inspiratory leak ) . they obstructed the lumen between the endotracheal tube and the common tube of the model to obtain calibrated low and high leaks . factors affecting the leak were similar , independently of the size of the endotracheal tube . the total leak was not affected by the breathing pattern , with similar values in normal , restrictive , and obstructive patterns . as expected , factors influencing predominantly the inspiratory phase influenced total leak but not expiratory leak . airway resistance , which mostly affects expiration , affected total and expiratory leak similarly , with higher leaks at high resistances . first , should the measurement of the expiratory leak alone replace the measurement of the total leak ? the design of this study was not to compare the performance of both tests but rather to evaluate the factors influencing the leak . as these patients were deeply sedated and even muscle relaxants were used , extubation occurred quite a long time after the test so that the ability of the test to predict extubation failure could not be assessed . it is even likely that the inspiratory component might have a major role in the validity of the test . inspiration occurs at high pressure , which favors leakage of gas around the tracheal tube . as the problem of the test is mainly its lack of specificity , suggesting that in some patients an absence of leak can be observed even when there is no airway obstruction , it is likely that using the expiratory leak would be even less discriminatory because this occurs at low pressure . second , the findings reported by prinianakis and colleagues might explain , at least in part , some of the differences between studies . series in post - operative patients showed that the capacity of the cuff - leak test to predict extubation failure was much lower than in populations of critically ill patients ventilated for at least 48 hours . this might be explained by the influence of decreased compliance or increased airflow resistance , which are more likely to occur in these patients . because these factors increase the amount of leak , an absent leak is even more suggestive of airway obstruction . in conclusion , the cuff - leak test can be used to identify patients at high risk of developing post - extubation stridor , who often require re - intubation . the use of this test in non - selected populations is of limited value but it might be more efficient in selected patients , even though an absent leak should never postpone extubation given the non - negligible rate of false positive tests . the data presented by prinianakis and colleagues help us understand the major roles of compliance , airway resistance , and flow in the interpretation of the cuff - leak test . characteristics of studies that used the cuff - leak test to predict extubation failure n.a . , not applicable .

stridor is one of the most frequent causes of early extubation failure . the cuff - leak test may help to identify patients at risk to develop post - extubation laryngeal edema . however the discrimination power of the cuff - leak test is highly variable and can be use , at best , to detect patients at risk to develop edema but should not be used to postpone extubation as tracheal extubation can still be successful in many patients with a positive test . in this editorial , the author discuss the factors influencing the leak and hence its predictive value .

to report a case of primary acquired melanosis ( pam ) successfully treated with combined topical mitomycin c and interferon-2b . a 75-year - old man presented with an extensive unilateral pigmented lesion involving 360 of the bulbar conjunctiva , extending to the fornices and palpebral conjunctiva . although the lesion markedly responded to the treatment , residual lesions justified further therapy . to avoid ocular surface toxicity induced by excessive mitomycin c exposure , interferon-2b eye drops were started and continued for 6 months . the pigmented lesion almost completely disappeared and no significant complication or recurrence was observed for 24 months . combination therapy using mitomycin c and interferon-2b for pam with atypia may be recommended as an effective treatment modality , avoiding the ocular surface toxicity due to excessive mitomycin c exposure . primary acquired melanosis ( pam ) of the conjunctiva appears as a flat , brown pigmented lesion in the conjunctival epithelium . pam with atypia is the most important precursor for conjunctival melanoma and its rate of of progression to malignant melanoma has been reported up to 46% . the most commonly used treatments for pam with atypia include surgical excision with cryotherapy and topical mitomycin c ( mmc ) or interferon-2b ( ifn-2b ) . mmc and ifn-2b are not only effective for treating conjunctival melanocytic tumors , but also have been used successfully for treatment of conjunctival squamous neoplasia , including conjunctival / corneal intraepithelial neoplasia ( cin ) and squamous cell carcinoma ( scc ) . ifn-2b was used for patients with cin or scc of conjunctiva in whom intolerance or recurrence of the tumor developed under treatment with mmc . however , to the best of our knowledge , combined use of mmc and ifn-2b for treatment of conjunctival melanocytic tumors has not been reported yet . a 75-year - old man presented with extensive brown pigmented lesions of the conjunctiva in his left eye . on examination , visual acuity was 6/10 with pseudophakia in the right eye and 2/10 with 3 + nuclear sclerosis in the left eye . the lesion involved the whole bulbar and palpebral conjunctiva extending into the upper and lower fornices . near the superotemporal limbus , a nodular portion of the lesion seemed to be encroaching onto the cornea . map biopsy confirmed pam with atypia especially in the nodular part of the lesion [ figure 1a c ] . treatment was initiated with two courses of topical chemotherapy each lasting for 2 months composed of three cycles mmc 0.04% eye drops every 6 h. the first cycle lasted for 3 weeks followed by a 1-week gap to allow the ocular surface to recover , then the second cycle was initiated for 2 weeks followed by another 1-week treatment gap of mmc and finally , mmc was administered again for 1-week . after 1-month of rest , the second 2-month episode of treatment with mmc was implemented as described above . the lesion decreased in size and pigmentation in the middle of the second course of therapy [ figure 1d f ] . therapeutic trend of our patient with extensive primary acquired melanosis ( pam ) in the left eye treated with topical mitomycin c ( mmc ) and interferon ( inf ) -2b ; ( a - c ) , appearance of the eye at presentation : histologically confirmed pam with atypia involved the bulbar and palpebral conjunctiva as well as the superotemporal cornea ; ( d - f ) , relative response to therapy in the middle of the treatment , note decreased size and pigmentation of the lesion ; ( g - i ) , final result of treatment after receiving mmc followed by 6 months of therapy with inf-2b ; ( j - l ) , appearance of the ocular surface after another year of follow - up , note more significant dilution of residual pigmentation without treatment . the second course of mmc therapy was interrupted in its final week because of severe discomfort of the patient . thus , ifn-2b ( 1 million units / ml ) eye drops were initiated every 6 h for 6 months , during which no significant topical or systemic side effect occurred [ figure 1d f ] . after 4 months , due to residual pigmentation close to the limbus and on the superior tarsal conjunctiva , the patient received inf-2b eye drops for more 3 months . no significant complications and no recurrence of the tumor the cataract was operated at this point of time and visual acuity was 7/10 at final follow - up . al this approach offers potential benefits over surgery including less dependence on surgical margins , direct delivery of a high concentration of the chemotherapeutic agent to the tumoral tissue , treatment of tumor extension on the cornea and comfortable repetition . the agent incurs low toxicity to the ocular surface and as compared to mmc , imposes a lower risk of complications such as corneal erosions , pain , conjunctivitis , punctal occlusion and scleral melting . over a mean follow - up period of 15 - 24.8 months and 15 months , complete clinical regression was reported in all treated patients except one subject with no systemic side effects . some investigators have used ifn-2b for patients with conjunctival cin or scc after intolerance to therapy or tumor recurrence following the application of mmc . since mmc toxicity is dose - dependent , replacing it with an effective topical medication with fewer side effects such as ifn-2b seems rational . to our knowledge , the patient presented herein , is the first reported case of pam successfully treated with topical mmc followed by long term topical ifn-2b . further studies with larger sample size using such combined therapy as primary treatment for pam and conjunctival melanoma are recommended .

purpose : to report a case of primary acquired melanosis ( pam ) successfully treated with combined topical mitomycin c and interferon-2b.case report : a 75-year - old man presented with an extensive unilateral pigmented lesion involving 360 of the bulbar conjunctiva , extending to the fornices and palpebral conjunctiva . map biopsy confirmed pam with local atypia . treatment was initiated with mitomycin c 0.04% eye drops for two courses . although the lesion markedly responded to the treatment , residual lesions justified further therapy . to avoid ocular surface toxicity induced by excessive mitomycin c exposure , interferon-2b eye drops were started and continued for 6 months . the pigmented lesion almost completely disappeared and no significant complication or recurrence was observed for 24 months.conclusion:combination therapy using mitomycin c and interferon-2b for pam with atypia may be recommended as an effective treatment modality , avoiding the ocular surface toxicity due to excessive mitomycin c exposure .

a 48-year - old presented to the emergency department with a 4-week history of inflamed scrotum . his scrotum was edematous and erythematous with well - delineated black necrotic areas ( fig.1 ) . the patient was given broad - spectrum antibiotics and iv fluids and referred to urology urgently . fournier gangrene is a rapidly progressive necrotizing infection of the perineal and genital fascia , often polymicrobial . in most infections a cutaneous , urethral or rectal source severity is increased in older patients , diabetics and the immunocompromised 1,2 . as in this case management options include resuscitative measures , broad - spectrum antibiotics , and surgical debridement 1,2 .

key clinical messagefournier gangrene is a rapidly progressive necrotizing infection of the perineal and genital fascia , often polymicrobial . severity is increased in older patients , diabetics and the immunocompromised . as in this case , the disease can have an indolent onset . early recognition and aggressive treatment is important .

chylothorax is a condition that results when the thoracic duct or its tributaries is disrupted along its course , leading to the accumulation of chyle within the pleural space . the etiology of chylothorax in adults is usually either traumatic or related to malignancy ( 1 ) . traumatic causes are most commonly iatrogenic ( post - surgical ) , but also have been reported after blunt or penetrating trauma , and only rarely requires surgical treatment ( 2,3 ) . we present a case of a high output chylothorax which resulted following a gunshot injury to the abdomen which required an unorthodox surgical approach to correct . an 18 year - old - male patient presented to our trauma center following a gun shot wound to the abdomen . on arrival , he was found to have a penetrating injury to the epigastrium with no apparent exit wound . he was hemodynamically stable and a cxr demonstrated a metallic foreign body in the upper abdomen . on physical exam , he was tender to palpation . the patient was taken to the operating room for an emergency laparotomy . during surgery , a laceration of the liver was identified and repaired with omental packing ( figure 1 ) . a large bullet fragment was palpable within the injured segment of liver and was removed . the patient had an uneventful recovery and was discharged home on post - op day 3 . six days later , the patient returned to the emergency department with complaints of right sided chest pain and worsening dyspnea . a plain chest film was performed ( figure 2 ) and demonstrated a large right pleural effusion . computerized tomography of the abdomen also demonstrated several tiny retained metallic fragments embedded within the liver and retroperitoneum ( figure 3 ) . a chest tube was inserted on the right side and immediately drained 2.2l of milky fluid ( figure 4 ) . fragment adjacent to aorta suspected of injuring retroperitoneal thoracic duct ( arrow ) . chest tube with classic appearance of milky white chyle . the diagnosis of chylothorax was confirmed after analysis of the fluid revealed elevated triglycerides and the presence of chylomicrons . the patient was initially treated with nothing via the oral route , parenteral nutrition and intravenous somatostatin analog . despite these conservative measures , having failed conservative therapy , the patient was taken to the operating room for a right - sided thoracotomy and attempted ligation of the thoracic duct . following this operation a few days later , the interventional radiologists attempted lymphangiography and embolization of the leaking duct ( figure 5 ) . this also proved unsuccessful and did not improve the output . the patient was subsequently taken to the operating room for a re - exploration and attempted ligation of the lymphatic leak transabdominally . during surgery , the right crus was divided and the retroperitoneal tissues to the right of the supraceliac aorta were ligated in an attempt to obstruct the lymphatics draining proximal to the transhepatic injury . following this operation , there was an immediate decrease in the output from the chest tube associated with an improved chest x - ray . the chest tube was removed on post - op day # 7 once the patient tolerated a regular diet without any increase or change in output . the patient was discharged home the next day and has since recovered well from his injury . lymphangiogram mapping through groin ( a ) with attempted embolization of thoracic duct utilizing fluoroscopy ( b ) . chylothorax has been reported with thoracic injuries but not previously reported with gunshot injuries to the abdomen . when diagnosed in a delayed fashion , chylothorax is typically managed conservatively with dietary restriction of fats , parenteral nutrition and chest tube drainage . somatostatin analog and embolization of injured lymphatic ducts has also been described with mixed results . thoracic duct ligation via a right thoracotomy is indicated for persistent drainage if conservative therapy fails to decrease the output . this approach is only successful if ligation occurs below the area of injury . in our case , we believe that a small metallic fragment injured the intrabdominal retroperitoneal portion of the cisterna chyli or thoracic duct , resulting in a persistent chylothorax despite attempted transthoracic ligation of the duct . continued output from the chest tube after thoracotomy could be explained by either failure to correctly identify and control the thoracic duct surgically , or an injury that was low enough to continue leaking chyle despite a more distally obstructed duct . we believe the later was the case since the output finally improved once we controlled the thoracic duct and lymphatics below the transhepatic injury . although less commonly used , the transabdominal approach has been previously described and thought to be advantageous due the thoracic ducts ' more consistent proximal anatomical location ( 4 ) . in summary , chylothorax is a rare complication following abdominal or retroperitoneal trauma and we believe that treatment may require an unorthodox approach , especially with suspected injuries below the thoracic cavity .

an 18 year - old - male sustained a gunshot wound to the abdomen which required an uneventful hepatorrhaphy . he later returned with a large right effusion and was diagnosed with a chylothorax . the output was persistent despite conservative measures . thoracotomy with attempted thoracic duct ligation was unsuccessful at decreasing the output . re - exploration and ligation of the thoracic duct was required thru an abdominal approach .

diabetes , as the most important medical condition of endocrinology , has impelled many initiatives to be organized by the federal government and professional associations . according to brazilian national data survey , diabetes is the fifth cause most frequent for hospitalization and is one of the 10 major causes of mortality in this country . for example , distribution to the entire population of oral antidiabetics and antihypertensive agents as well as human insulin is paid for by the government . as observed in other developing countries , prevalence of type 2 diabetes mellitus in brazil surveys showed a prevalence ranging from 6.7% to 9.2% in 1988 and from 9.0% to 10.2% in 2008 in general population . these figures are more astonishing in older population being 21.7% in age group between 60 and 69 years . chiefly among the younger age groups , increasing rates of overweight , obesity associated with lifestyle changes , and population aging are the main contributing factors to the increasing prevalence of type 2 diabetes . the observed changes in food consumption , with special emphasis on low consumption of fibre - rich foods and heavy consumption of free sugars , with or without saturated fatty acids , industrialized foods and beverages , constitute an important risk factor for developing diabetes in brazil , mainly in the low - income social classes , independently of body mass index . in brazil , a study in japanese brazilian community in bauru ( state of so paulo ) showed a higher intake of calories from lipids in relation to the dietary habits of japan 's population . in this population survey , the prevalence of type 2 diabetes , impaired glucose tolerance and impaired fasting glucose has risen dramatically in recent years , demonstrating the impact of environmental conditions interacting with a probable susceptibility gene on the risk of developing changes in glucose metabolism . regarding medical education , there are no special programs on diabetes in brazil , as it is taught as a subspecialty of clinical endocrinology . residence programs in endocrinology at university hospitals consist of an internship of 2 years at internal medicine and further 2 years at endocrinology service . after this training period , certifications are issued by brazilian society of endocrinology and metabolism together with brazilian medical association . all endocrinologists are encouraged to join brazilian society of diabetes , which comprises health care providers from all over brazil and abroad in great numbers : 3380 medical doctors , 205 nurses , 154 dietitians , 44 pharmacists , 40 psychologists , and 57 from other professional fields . the society is actively engaged in providing continuing medical education through regional and national scientific meetings and workshops . many guidelines regarding many aspects of diabetes management have been published and are easily available to all the society members . research in endocrinology is fostered by a number of organizations , which were created mostly during the 1950s , specifically for directly promoting and funding research and development in all science fields . among them , the national research council ( cnpq ) and the national agency for financing studies and researches ( finep ) , nowadays as part of the ministry of science and technology , are the most active . following the pioneering ( and highly successful ) example of so paulo research foundation ( fapesp ) , almost all federal states have their own public foundations for supporting research and development in general . usually these foundations are financially supported by state funds as settled in the constitutions of each federal state . brazilian scientific production exhibited a 4-fold increase in the last 2 decades because of investment in research and development activities , and changes in the policies of the main funding agencies mentioned above . most of this production is concentrated in public universities and research institutes located in the richest part of the country . among all areas of knowledge , the most productive are health and biological sciences . during the 19982002 period these areas presented heterogeneous growth ranging from 4.5% ( pharmacology ) to 191% ( psychiatry ) , with a median growth rate of 47.2% . the 20 most productive centers , during the 19982003 period , produced 78.7% of the scientific articles in these areas and are strongly concentrated in the southern part of the country , mainly in so paulo state . on the other hand , pubmed search on clinical articles about diabetes from authors affiliated to brazilian institutions resulted in a mean of 7.7 articles per year in the last 10 years . in comparison , for india , the same approach unveiled a mean of 33.9 articles per year . when compared with usa the difference is the greatest , as usa has produced a mean of 6223 articles per year in the last 10 years . capes foundation , a public organization within the brazilian ministry of higher education , released a ranking that puts brazil among the 25 countries with the greatest scientific productivity . this is due to the large proportion of the 10,000 phds released per year orbiting around academia and contributing to scientific production . however , in endocrine / diabetes fields , a more realistic and responsible policies in terms of medical traineeship , patient education , and mainly scientific production would be mandatory to raise brazil to a global competitive level .

brazil is a large , populous country in south america , which has one of the biggest concentrations of people with diabetes . this article reviews the current status of diabetes care , medical education and training , and diabetes / endocrine research in brazil . it highlights achievements in public health and research , which can be emulated by other countries . in endocrine / diabetes fields , a more realistic and responsible policies in terms of medical traineeship , patient education , and mainly scientific production would be mandatory to raise brazil to a global competitive level .

the diagnosis is usually made by demonstration of antigen in the urine , serum , relevant body fluids or pus and by fungal culture . herein we are presenting a rare case of genitourinary histoplasmosis where the diagnostic dilemma was resolved by fungal culture of prostatic abscess and histopathological examination of epidydimal tissue . a 37-year - old male , a renal transplant recipient , with the mother as donor , presented with complaints of pain and swelling in the scrotum and fever of one week duration , three years after the transplant . post transplantation , he was on triple immunosuppressive therapy with tacrolimus , azathioprim , and prednisolone , and his allograft function was stable . one - and - a - half years after his renal transplant , he had dysphagia and malena , and was diagnosed with esophageal candidiasis and colonic histoplasmosis on the basis of upper gastrointestinal endoscopy and colonoscopy , respectively . histoplasmosis was confirmed by demonstrating histoplasma capsulatum on histopathology of the biopsy from colonic ulcers and stool culture . he responded to itraconazole , which was continued for nine months and was asymptomatic on follow - up . at the time of this presentation , three years after the transplant , blood investigations revealed severe anemia , leucopenia , thrombocytopenia , and serum creatinine of 3.0 mg / dl . urinalysis showed proteinuria ( 300 mg / dl ) with numerous red blood cells ( rbcs ) and pus cells . digital rectal findings suggested bogginess on the anterior rectal wall with prostatic landmarks not well appreciable . the ultrasound showed enlarged , heterogenous epididymis , with fluid collection and a thick - walled collection in the prostate and retrotrigonal area . computerized tomography of the pelvis confirmed the ultrasound findings [ figures 1a , b ] . ct scan pelvis showing retrotrigonal ( a ) and prostatic location ( b ) of collection the epididymal swelling was explored and tissue biopsy was sent for polymerase chain reaction ( pcr ) for mycobacterium tuberculosis and histopathology . the prostatic and retrotrigonal collection was drained via the per - rectal route and sent for culture studies . histopathology diagnosed histoplasmosis [ figure 2 ] and culture of the pus ( obtained from the prostatic / retrotrigonal abscess ) confirmed the growth of histoplasma capsulatum [ figure 3 ] . a repeat colonoscopy was done , which did not reveal any ulcers or polyps or any other finding suggestive of intestinal histoplasmosis . the patient was restarted on itraconazole for nine months , to which he responded successfully . photomicrograph showing sheets of histiocytes with intracytoplasmic pas - positive yeast forms , suggestive of histoplasmosis : pas stain 400 magnification photomicrograph showing saprophytic phase of histoplasma capsulatum with spherical , tuberculate , macroconida ( 8 14 m ) : lactophenol cotton blue stain 40 magnification the risk of genitourinary tuberculosis is increased in transplant recipients , who are either born or live in areas where tuberculosis is endemic . the frequency of mycobacterium tuberculosis disease among the recipients of solid organ transplants in most developed countries is 1.2 6.4% , but among transplant recipients living in areas of high endemicity , it can reach 15% . it is worth noting that diverse , unsuspected , and elusive sites of tuberculosis infection have been described . in this patient , with enlargement of epididymis coupled with findings of an abscess in the prostatic and retrotrigonal region , tuberculosis was a strong probability . however , tissue pcr and culture for mycobacterium tuberculosis was negative and findings of histopathology and culture were conclusive for histoplasmosis . even as most cases of histoplasmosis were asymptomatic or self - limiting , they could rarely lead to severe and progressive disseminated infections . diagnosis of disseminated histoplasmosis required a high index of suspicion.[24 ] only four cases of epididymal histoplasmosis have been reported in literature so far . two cases were diagnosed from the united states in 1981 , one was presented by kahn dg , et al . , at a conference in the united states in 1992 ( international conference on acquired immunodeficiency syndrome ( aids ) 1992 jul 19 - 24 ; 8 : 93 ) , and the fourth case was reported from india in 1995 . three cases were diagnosed by histopathological examinations and pus cultures , and the fourth case was diagnosed with the help of a seminal culture . all the cases had a similar clinical presentation : fever , weight loss , and testicular pain and / or swelling . involvement of the prostate is very rare and only nine cases have been previously reported . reinfection can occur in patients who have had histoplasmosis in the past , like our patient who had colonic histoplasmosis . when there is a decline in immunity in such a host , residual host defenses can not combat invasion by histoplasma . diagnosis of histoplasmosis is made by the demonstration of antigen in the urine , serum , relevant body fluids or pus . histopathology of the suspected lesion can reveal yeast structures of h. capsulatum via the hematoxylin and eosin stain ; however , the organism is better visualized using methenamine silver or periodic acid schiff stains ( pas ) . confirmation can be obtained by setting up appropriate cultures , demonstrating the growth and dimorphic properties of the fungus . a high index of suspicion is needed for the diagnosis of histoplasmosis in immunosuppressed patients . the tissue , pus , and body fluid available for diagnosis must be subjected to investigations for uncommon infections , too .

genitourinary histoplasmosis is very rare and to our knowledge only four cases of epididymal histoplasmosis and nine cases of prostatic histoplasmosis have been reported in literature . we hereby report a case of a middle - aged male , who presented three years after renal transplant , with complaints of fever , pain , and swelling in the scrotum . imaging disclosed an enlarged right epididymis with prostatic and retrotrigonal abscess , suggesting tuberculosis infection . however , histopathology of the epididymal biopsy revealed histoplasmosis , and the drained pus on culture confirmed infection with histoplasma capsulatum .

in this issue of critical care , dale rublee and colleagues look beyond 28-day all - cause mortality , assessing the effects of antithrombin iii on quality of life in sepsis survivors . although many of us would like to think that the battle has been won when a patient leaves the intensive care unit ( icu ) after severe sepsis or acute respiratory distress syndrome ( ards ) , a growing body of literature indicates that survivors of intensive care have an increased risk of death and have a significantly impaired quality of life after they leave the icu . there are several possible mechanisms for these long - term effects , and many patients may be affected by several of them . survival may be impaired simply because people with severe co - morbid diseases are most likely to get critically ill . alternatively , critical illness may leave patients immunosuppressed and at risk for further complications that lead to early death . the trauma of critical illness and false memories may cause post - traumatic stress disorder and depression . tracheostomy scars , striae from anasarca , decubitus ulcers , and digits lost to ischemia may affect patients ' self image . the study by rublee and colleagues is an analysis of secondary endpoints in the kybersept ( antithrombin iii ) trial that failed to show a statistically significant effect on 28-day mortality . they compared the karnofsky performance scale and six domains of a visual analogue scale of quality of life : mobility , physical activity , communications the authors performed at least 63 statistical comparisons across variables , time points , and populations to identify several situations in which antithrombin iii seemed to have a statistically significant effect on quality of life . it is difficult to assess the importance of this effect because the clinically significant difference in percentage change in their visual analogue scale is unknown . because the results are all expressed as change from a baseline assessment when the patients were critically ill , we can not tell their actual status at each time point . despite these limitations , this study raises several important questions that critical care investigators must answer as we design studies of quality of life after critical illness . . we do not expect therapy for severe sepsis to improve patients ' quality of life ; at best we hope to return patients to their pre - morbid health status . ideally , we can find management techniques that minimize the effects of a critical illness on long - term quality of life . conflicts may arise between treatments that are life - saving and those that affect quality of life after critical illness . for example , it is possible that lung - protective ventilation for ards using increased sedation , lower oxygenation , and permissive hypercapnea saves lives but leads to worse cognitive function than a strategy using large tidal volume oriented toward normoxia and eucapnea . there are as yet insufficient data to assess this trade - off . functional status and quality of life are extremely important to patients ' decisions about life - sustaining treatment , and much better data are needed to allow us to predict future health status . when death competes with a non - mortality endpoint such as quality of life , a phenomenon called ' informative censoring ' can occur . a treatment that increases mortality may preferentially lead to the death of debilitated patients who would have had very poor quality of life if they had survived . similarly , a treatment that saves the lives of very ill debilitated patients may rescue these patients so that they are healthy enough to contribute very poor quality - of - life data to the study . life - saving therapy in the icu can seem to worsen quality of life , and harmful treatments can seem to improve quality of life . it is therefore problematic to adopt a therapy in the icu solely on the basis of its apparent effect on quality of life unless we know that this effect is not purchased by increasing mortality in the most debilitated . there are statistical techniques for studying a non - mortality endpoint in the face of competing mortality and these should be used by intensivists to evaluate the effects of interventions on quality of life after critical illness . intensivists are used to attempts in the complex body of literature in critical care to identify the ' best ' severity of illness measure . a similar , even more extensive , body of literature exists that explores the ' best ' quality - of - life instruments . experts debate the responsiveness , validity , clinical significance , and practicability of these instruments in various patient populations . selecting a general measure - of - health status that has been used extensively , such as the sf ( short form)-36 or euroqol , allows investigators to compare their results with a mature database of patients with many different diseases . studying measures of function , for example muscle strength , cognitive ability , and psychiatric status , can provide invaluable information for understanding the mechanisms of a reduced health - related quality of life . investigators interested in establishing the cost - utility of a therapy should use a health status measure that can be converted to patient utilities . reducing the long - term impairment associated with critical illness presents a set of new challenges to clinicians in the icu . we need association studies that link factors before , during , and after treatment in the icu with health status . continuing clinical trials in critical care can provide this information on risk factors by including long - term follow - up for survival and quality of life in their data collection . in the very near future we should expect clinical trials exploring the effects of icu and post - icu interventions targeted at improving quality of life after intensive care .

a growing body of evidence indicates that survivors of intensive care have an impaired quality of life . it is not entirely clear from the available literature whether this impairment is a complication of critical illness or a complication of therapy . there is little evidence to guide physicians to treatments in the intensive care unit that will minimize the effects of critical illness on these sequelae . although the study by rublee and colleagues in this issue of critical care provides little clinically useful information about the effects of antithrombin iii on quality of life , it provides some insight into the challenges that investigators will encounter as we try to incorporate these outcomes into studies of critical illness .

intravenous thrombolysis with tissue plasminogen activator ( tpa ) has been licensed for selected patients within 3 h after ischemic stroke in many countries [ 1 , 2 ] , and a recent clinical study demonstrated the efficacy and safety up to 4.5 h after stroke onset . however , the therapeutic guidelines of intravenous thrombolysis with tpa are very strict . because of potential higher risk of bleeding complications , the presence of an unruptured cerebral aneurysm is a contraindication for systemic thrombolysis with tpa [ 2 , 4,5,6,7 ] . in this case study , we report a patient receiving intravenous alteplase within 3 h after ischemic stroke according to the standard ct criteria , in whom post - tpa investigations detected an unruptured cerebral aneurysm . a 66-year - old woman without family history of subarachnoid hemorrhage ( sah ) developed aphasia and right hemiparesis , and arrived at our hospital 135 min after symptom onset . the baseline national institute of health stroke scale was 10 points and baseline ct showed an early ischemic sign in the left frontal lobe ( fig . 1 ) . the patient met the guidelines for systemic thrombolysis with tpa and received intravenous alteplase 177 min after onset . magnetic resonance ( mr ) imaging during tpa infusion detected a focal territorial early ischemia in the left frontal lobe on the diffusion image , and mr angiography showed no arterial occlusion and a suspected unruptured cerebral aneurysm at the anterior communicating artery . the patient had mild aphasia without hemiparesis at 90 days , indicating the modified rankin scale of 1 . delayed cerebral angiography for endovascular coiling after 3-month outcome follow - up confirmed an unruptured cerebral aneurysm with a diameter of about 7 mm at the anterior communicating artery ( fig . in this patient , the presence of an unruptured cerebral aneurysm was finally confirmed on cerebral angiography after 3-month clinical follow - up , although the presence of the aneurysm was suspected by mr angiography during tpa infusion . therefore , the off - label use of tpa was not intentional but accidental . if this patient had undergone mr angiography before tpa infusion , she would not have received intravenous tpa . however , according to the standard guidelines with the rigid 3-hour therapeutic time window , this patient received intravenous tpa . in clinical practice , standard ct - based thrombolysis with tpa for ischemic stroke may occasionally include non - stroke conditions . utilization of mri or vascular imaging before thrombolysis can provide a higher accuracy of stroke diagnosis and may detect the contra - indicated conditions such as cerebral aneurysm or other vascular anomalies . however , these additional imaging studies may delay the delivery of tpa . in reports on off - label thrombolysis for stroke [ 4 , 5 ] , intentional off - label thrombolysis , such as intravenous alteplase for elderly patients over 80 years in europe or for patients with seizure at onset , has been performed . in previous reports , only two patients in whom an incidental unruptured cerebral aneurysm was detected after intravenous tpa safely received systemic thrombolysis for stroke . among about 70,000 patients receiving systemic thrombolysis for myocardial infarction , none experienced an aneurysmal sah . to our knowledge , only one patient developed an aneurysmal sah after systemic thrombolysis for myocardial infarction . the decision against systemic thrombolysis in the presence of an unruptured cerebral aneurysm has no scientific basis [ 4 , 5 ] . in the general population , the incidence of an unruptured cerebral aneurysm has been reported to be 3.6 - 6% . therefore , many patients with an unruptured cerebral aneurysm might safely receive systemic thrombolysis with tpa , although unfavorable complications after off - label thrombolysis may have been underreported . the safety of systemic thrombolysis with tpa in stroke patients with an unruptured cerebral aneurysm should be further examined , especially with regard to the size and location of the aneurysm . in conclusion , data collection of both intentional and accidental off - label thrombolysis for stroke may contribute to modify the presently applied rigid guidelines .

therapeutic guidelines of intravenous thrombolysis with tissue plasminogen activator ( tpa ) for hyperacute ischemic stroke are very strict . because of potential higher risk of bleeding complications , the presence of unruptured cerebral aneurysm is a contraindication for systemic thrombolysis with tpa . according to the standard ct criteria , a 66-year - old woman who suddenly developed aphasia and hemiparesis received intravenous tpa within 3 h after ischemic stroke . magnetic resonance angiography during tpa infusion was performed and the presence of a small unruptured cerebral aneurysm was suspected at the anterior communicating artery . delayed cerebral angiography confirmed an aneurysm with a size of 7 mm . the patient did not experience any adverse complications associated with the aneurysm . clinical experiences of this kind of accidental off - label thrombolysis may contribute to modify the current rigid tpa guidelines for stroke .

a sixty - year - old man was admitted to the emergency department with sharp , stabbing chest pain . the symptom had first appeared a month ago , and had aggravated in the 5 days prior to presentation . the patient 's medical history showed that he had undergone a fixation operation using kirschner wire for treatment of a right pelvic bone fracture from a traffic accident about twenty years prior . he was diagnosed with diabetes and had been taking oral hypoglycemic agents for the past ten years , and also had spine and knee surgery due to lumbar spinal stenosis and degenerative arthritis of the left knee . during evaluation , his vital signs were stable and chest radiograph revealed a thin linear foreign body in thorax ( fig . electrocardiography showed a normal sinus rhythm and laboratory tests including cardiac enzymes were in the normal range . chest computerized tomography revealed a linear metallic foreign body from the pelvic bone that was penetrating the right ventricle of the heart with mild pericardial effusion ( fig . echocardiography showed a foreign body penetrating at the apex of the right ventricle , and mild ( grade i ) mitral , tricuspid regurgitation with normal valve morphology ( fig . one kirschner wire had disappeared from the pelvis radiograph compared with the previous image ( fig . after pericardiotomy , there was no hemopericardium and about 1 cm of protruding kirschner wire could be seen to the left of the distal left anterior descending coronary artery ( fig . 2b ) , without cardiopulmonary bypass or cardiotomy , we removed it after wrapping suture at myocardiumwith pledgeted 4 - 0 prolene suture . the patient had fully recovered without any complications and was discharged from the hospital on the fifth postoperative day . fixation instruments are frequently used in fracture and dislocation of bones , but their migration is uncommon . migration to the cardiovascular system is usually symptomatic , and can result in devastating complications . in an extensive review of case reports of wire migration from the shoulder region , the authors found 49 cases of migration in 47 patients , including 17 cases in which a pin had migrated to a major vascular system such as the heart ( 4 cases ) , the subclavian artery ( 2 ) , the ascending aorta ( 6 ) , and the pulmonary artery ( 5 ) . the causes of migration are known to be the action of muscles , the capillary phenomenon , bone resorption , gravity , and the range of motion . especially in the lower extremities , we found one report of migration of a wire used in patella fixation into the heart . the greater activity mass on the upper extremities , the range of motion , and gravity are likely to affect the incidence of migration . also , the distance between the instrument and major vessels should be considered in cardiovascular complications . in this case , we believe that the kirschner wire entered the vascular system penetrating into the external iliac vein and migrated through the inferior vena cava to reach the right ventricle . a doppler ultrasound or computed tomography can help to measure the distance , and more careful observation is necessary when the distance is close . because migration of a bone fixation device to the cardiovascular system can cause devastating complications , periodic radiographic follow up is necessary . in addition , even if there is no migration , surgeons may consider removing a fixation device depending on a patient 's age , activity level , and distance to major vessels .

a sixty - year - old man was admitted due to chest pain . he had a history of pelvic bone fracture fixation with kirschner wire about 20 years earlier . on examination , we detected a kirschner wire that had migrated into the right ventricle . without cardiopulmonary bypass , we removed the migrating kirschner wire via median sternotomy . the patient recovered without complications and was discharged on the 5th postoperative day .

a 9-year - old girl visited our hospital due to lateral knee pain that developed one week after a hyperextension injury of her left knee joint during exercise . physical examination revealed lateral joint line tenderness and positive results for the mcmurray test in both knee joints . the lachman test , anterior and posterior drawer and pivot shift tests , and maneuvers for rotator instability were all negative . the range of motion ( rom ) was no flexion contracture and a further flexion of 120 with mild pain in her left knee . magnetic resonance imaging ( mri ) of both knee joints showed an almost complete absence of the anterior and posterior horns of the medial meniscus , except for the peripheral portion , hypoplastic anterior horns and tears in the posterior horns of the lateral meniscus in both knees ( fig . 1 ) . the patient did not complain of pain or discomfort in her right knee , but did complain of pain and limited motion in her left knee . we , therefore , decided to perform arthroscopic surgery only on her left knee . at arthroscopy , the medial meniscus showed hypoplastic changes ( approximately 5 mm in width and 12 mm in height ) . the anterior horn of the lateral meniscus showed hypoplastic changes , while the posterior horn was connected to the meniscofemoral ligament with fibrous tissue . both the anterior and posterior cruciate ligaments were intact and there was no chondral pathology medially or laterally . arthroscopic partial meniscectomy was performed for the horizontal tear of the posterior horn of the lateral meniscus ( fig . postoperatively , the patient was allowed to bear weight and perform knee motions as tolerated . two weeks after the operation , she restored full active and passive rom without pain . at 4-year follow - up , she had no symptoms or abnormal objective findings for both knees and she had returned to full activity . on the follow - up mri for both knees at postoperative 4 years , there were no chondral lesions or tears of the remnant meniscus in the left knee and no significant changes in the right knee . the characteristic shapes of both the medial and lateral menisci are observed early in prenatal development , maturing along with the capsule and coronary and cruciate ligaments . rather , gradual changes occur , including a decrease in vascularity , progressing from the center to the peripheral margins ; a growth in size ; and changes in the configuration of the menisci1 ) . the association of other anomalies in the knee with hypoplastic menisci may be due to the common mesenchymal origin of these structures1,2 ) . however , in the patient described here , the only anomalies were in the menisci . the most common congenital abnormality of the meniscus is a discoid meniscus , which is more frequent in the lateral than the medial meniscus1,5 ) . other abnormalities have been reported , including abnormal attachments of the meniscus , hypoplasia and partial deficiency39 ) . these abnormalities were all incidentally diagnosed in patients with symptoms associated with trauma or other congenital anomalies . congenital hypoplasia of the medial meniscus has been reported in three patients4,6 ) , and bilateral hypoplasia of the medial meniscus with an outerbridge grade iii chondral lesion of the medial femoral condyle in one patient2 ) . one patient was reported to have partial deficiency of the lateral meniscus9 ) and another was found to have bilateral congenital absence of the anterior cruciate ligament and the internal menisci of the knee7 ) . another patient was found to have congenital absence of both menisci and both cruciate ligaments with radial club hands and congenital thrombocytopenia5 ) . bilateral hypoplastic lateral menisci with an osteochondral lesion in the lateral tibial plateau were reported in one patient3 ) , with another having congenital hypoplasia of the lateral meniscus and anterior cruciate ligament associated with osteochondritis dissecans of the medial femoral condyle8 ) . osteochondritis dissecans of the medial femoral condyle associated with hypoplastic medial and partially deficient lateral menisci was also reported10 ) . however , we have been unable to find a description of bilateral hypoplastic medial and lateral menisci . we think that future research efforts are necessary to detect the incidence and clinical course in patients with hypoplastic menisci .

hypoplasia of the meniscus is a very rare congenital abnormality , with only a few cases reported to date . a 9-year - old girl visited our hospital due to lateral knee pain following a hyperextension injury to the left knee . magnetic resonance imaging showed hypoplasia of the medial and lateral menisci , as well as a posterior horn tear of the lateral meniscus , in both knee joints . to our knowledge , this is the first report of a patient with hypoplasia of the medial and lateral menisci in both knee joints .

we report the cases of 3 patients with whim syndrome who are affected by tof ( figure ) . taking into consideration the relatively poor prevalence of the two disorders , in the present report the first patient is a 19-year - old man with a history of whim syndrome identified at age 2.5 years , manifesting as severe neutropenia and recurrent pneumonias , resulting in bronchiectases ( table ) . these hematologic and clinical findings led to the suspicion of whim syndrome and thereby to cxcr4 gene sequencing that revealed s338x mutation . the congenital heart disease was suspected at birth and was characterized by the anatomic variant of tof associated with pulmonary atresia and with anomalies in branch pulmonary arteries . of note , the patient presented with agenesis of the left - hand fingers with homolateral hypoplasia of the radius . the patient has been maintained on daily subcutaneously administered granulocyte - colony stimulating factor therapy since 2 years of age . in the second case , the patient had tof associated with the presence of patent ductus arteriosus documented at birth ; the heart disease was surgically corrected at the age of 2 years ( table ) . this patient is a 15-year - old girl who has had recurrent respiratory infections since early childhood . severe neutropenia was discovered at age of 2 years , and on that occasion , an analysis of the bone marrow revealed myelokathexis with mature neutrophils presenting morphologic abnormalities consistent with apoptosis . granulocyte - colony stimulating factor therapy was started at 5 years of age to maintain circulating neutrophil count in the normal range . because of the occurrence of repeated pneumonia episodes , she has been maintained on antibiotic prophylaxis . in the following years , the observation of hypogammaglobulinemia suggested whim syndrome and genetic confirmation of the diagnosis was obtained at age 5 by detection of the r334x mutation in the cxcr4 gene . the third patient is 7 years old and presented shortly after birth with a heart murmur that led to the discovery of a tof characterized by ventricular septal defect , overriding aorta , and pulmonary infundibular stenosis . neutropenia , lymphopenia , and hypogammaglobulinemia were present since infancy , and the analysis of bone marrow showed myelokathexis . three other family members have had neutropenia and recurrent infections , but none of them had a congenital heart defect . the genetic analysis of cxcr4 revealed the same mutation ( s338x ) in the 4 subjects . the child underwent treatment with intravenous immunoglobulins , obtaining a satisfactory control of infectious episodes . although tof is the most common form of cyanotic congenital heart disease , its occurrence in 3 unrelated patients with whim syndrome is much higher than expected ; the normal occurrence in the general population is 3 of every 10 000 live births . because of the low number of patients whom we have observed , we can not draw a correlation between the development of the heart defect with a specific mutation . the detection of the same mutation ( s338x ) in a family with 4 members affected by whim syndrome with only 1 showing tof rules out the hypothesis that the heterozygous gain - of - function mutation of cxcr4 directly leads to the development of the cardiac defect . instead , our observations suggest that the whim syndrome associated cxcr4 truncating mutation might increase the risk that this combination of cardiac defects may develop during the formation of the fetal heart . beyond a role for cxcl12 and cxcr4 in heart , nervous system , and blood vessel development , studies show that cxcr7 , the recently described second receptor for cxcl12 , has also a role in fetal endothelial biology and heart development in particular , ventricular septum and heart valve development . its germline deletion results in perinatal lethality , and its mutation affects semilunar valve development , contributing to aortic and pulmonary valve stenosis and , in some cases , septal defects . recent findings support the view that cxcr7 modulates cxcr4 function by acting as a scavenger for cxcl12 and forming heterodimers with cxcr4 . in vitro studies on endothelial progenitor cell function show a role for both cxcr4 and cxcr7 in regulating the response of the cells to cxcl12 and , thus , angiogenesis . this suggests that interactions between the 2 chemokine receptors may be required for proper valve morphogenesis . our observations demonstrate that tof can be a presenting manifestation of whim syndrome and that this rare inherited disease should be suspected in children with a congenital heart defect and neutropenia . the recognition of this manifestation of whim syndrome might help to prevent the diagnostic delay in the identification of this rare genetic disease . a prompt diagnosis should facilitate the management of leucopenia , which might include in the future cxcr4-targeted therapy as supported by 2 recent studies indicating that the specific cxcr4 antagonist plerixafor may be effective in restoring the cellular blood counts to normal .

warts , hypogammaglobulinemia , infections , and myelokathexis ( whim ) syndrome is a rare immunodeficiency disorder . we report three patients with whim syndrome who are affected by tetralogy of fallot ( tof ) . this observation suggests a possible increased risk of tof in whim syndrome and that birth presentation of tof and neutropenia should lead to suspect whim syndrome .

primary malignant melanoma of the esophagus ( pmme ) is one of the extremely rare tumor and the prognosis is very poor . this tumor has usually been reported as a pedunculated , polypoid lesion in the middle and lower third of the esophagus . according to the reports , hematogenic and lymphogenic metastases are common for pmme cases . adjuvant chemotherapy has been employed in some cases , but the effectiveness has not been proved . we report a case of pmme which was treated by surgical resection and post - operative chemotherapy . a 60-yr - old male presented with a 3-month history of dysphagia and upper abdominal discomfort . he was treated at a private clinic under the impression of gastritis without symptomatic improvement . there were no abnormalities noted on physical examination or within the laboratory reports , and a pre - operative simple chest radiography and electrocardiography showed no abnormal findings . barium - contrast esophagogram showed irregular luminal narrowing with a 4 cm sized filling defect at the distal esophagus . 1 ) . fiberoptic esophagoscopy showed a bell - shaped obstructing black mass with a 4 cm sized central ulcer located between 32 and 36 cm from the incisors . chest computerized tomogram showed the mass to be 5 cm in size at the posterior wall of the distal esophagus with evidence of lymph node enlargement in the lesser curvature of the stomach ( fig . 2 , 3 ) . pulmonary function tests , whole body bone scan with tc-99 m , and a computerized brain tomogram were all unremarkable , and therefore the pre - operative clinical stage was classified as iva ( ct3n0m1a ) . the patient underwent a total esophagectomy and cervical esophagogastric anastomosis via a right thoracotomy , laparotomy and cervical incision . reconstruction was performed by pulling the gastric tube up through a retro - sternal route . there was no evidence of mediastinal lymph node enlargement and the post - operative course was uneventful but the patient suffered from post - operative pain . the resected mass was 5.54.21.3 cm in size , polypoid and pigmented from the distal esophagus with lymph node enlargement along the lesser curvature of the stomach ( fig . microscopic examination revealed junctional activities of atypical melanocytes around base of the tumor and a definitive diagnosis of pmme was made ( fig . 5 , 6 ) . three lymph nodes at the lesser curvature of the stomach were infiltrated by tumor cells . this case was then classified as esophageal cancer , stage iva : t2-n0m1a , according to the tmn classification of ajcc . four months after discharge , at a follow - up , lymph node enlargements in the cervical area and celiac axis area were found and systemic chemotherapy was started . the patient received dacarbazine 800 mg / m intravenously ( iv ) on day 1 , cisplatin 30 mg / m iv on days 2 - 5 , and vinblastine 1.6 mg / m iv on days 1 - 5 . after the fifth cycle of chemotherapy , the size of the lymph nodes decreased significantly and at 35 months post - operative , the patient is doing well without evidence of recurrence . since baur made the first report of pmme in 1906 , scattered cases have been reported in the literature and the incidence of pmme is reported to be less than 0.1% of all esophageal malignancies ( 1 ) . chalkiadakis and colleagues , in a review of 110 patients diagnosed with pmme found that men predominated the cases by 2:1 , with an overall mean age of 60 yr ( 2 ) . the tumors were usually located in the middle and lower third of the esophagus ( 86% ) . the common symptoms were dysphagia ( 73% ) , retrosternal pain ( 24% ) , weight loss ( 16% ) , and regurgitation ( 12% ) . the diagnosis of pmme is difficult and best deduced by a combination of several examinations including upper gastrointestinal series , endoscopy , computed tomographic scan and biopsy with histopathologic assessment . macroscopically , the tumor usually presents as a polypoid mass , that is focally ulcerated , but covered for the most part by intact squamous mucosa . according to the diagnostic criteria of primary melanoma proposed by allen and spitz , which seems to be widely accepted , the tumor is considered as primary when ( a ) the mass has a characteristic structure of melanoma containing melanin , ( b ) the adjacent epithelium contains melanocytes , ( c ) the tumor is polypoid , and ( d ) it arises from the area of junctional changes in the squamous epithelium ( 3 ) . thus , the proposed criteria for the origin of a tumor in the esophagus depends on the finding of junctional activity adjacent to the tumor mass . recently , immunohistochemical staining utilizing antibodies specific to hmb-45 and/or s-100 cytoplasmic protein has been used to identify malignant melanoma of the esophagus ( 4 , 5 ) . hematogenic and lymphogenic metastasis are commonly associated with pmme and account for the poor prognosis . widespread metastases with a rapid deterioration in health , followed by an early death is the usual clinical course . the most common site of metastases reported is the liver , followed by the mediastinum , mediastinal lymph nodes , lung and brain ( 2 ) . the choice of therapy primarily depends on the functional status and the presence and extent of the metastatic disease at the time of diagnosis . surgical resection with re - establishment of gastrointestinal continuity is the proposed methods of treatment , and looks like to provide the best result . the median survival rate after surgical resection was 13.4 months and longest reported survival was 12 yr ( 2 , 6 ) . chemotherapy , radiation therapy , and immunotherapy have been reported , but with limited success ( 7 , 8) . recently , several authors have reported a therapeutic effect of combination chemotherapy including dacarbazine ( 9 , 10 ) . in this patient , this case supports the combination of surgical resection and post - operative adjuvant chemotherapy , with dacarbazine , and therefore should be considered as another treatment modality .

primary malignant melanoma of the esophagus ( pmme ) is an extremely rare tumor with only scattered cases reported . although surgical resection has been considered as the best possible option , the prognosis has been nonetheless poor . we report a case of pmme which was treated by surgical resection and additionally followed by chemotherapy . a 60-yr - old man underwent an esophagoscopy due to a 3-month history of dysphagia and upper abdominal discomfort . a pigmented polypoid mass in the lower third of the esophagus was discovered , and a biopsy identified the mass as a malignant melanoma . consequently , a subtotal esophagectomy and intrathoracic esophagogastrostomy was carried out . at follow - up four months after discharge , lymph node enlargements in the cervical area and celiac axis area were found . as a result , the patients was started on systemic chemotherapy treatment , which included dacarbazine . the patient has been doing well and is now 35 months post - operative .

in the previous issue of critical care , kamps and colleagues reported a surprising observation , one that at first glance contradicts perceived knowledge on therapeutic cooling . numerous studies have shown that hypothermia can prevent or mitigate ischaemia / reperfusion injuries , and can be used to treat brain oedema [ 2 - 4 ] . there is a growing list of potential indications , although many applications still await evaluation in rigorous clinical trials . in recent years one key mechanism is the inhibition of a harmful proinflammatory cascade that develops in injured organs following traumatic or ischaemic injuries . many animal experiments and clinical studies have shown that hypothermia can suppress these harmful inflammatory reactions , and block the release of proinflammatory cytokines [ 5 - 8 ] . hypothermia can also decrease production of leukotrienes and nitric oxide , prevent reperfusion - related dna injury and lipid peroxidation , and impair neutrophil and macrophage function . at temperatures < 32 to 33c , hypothermia can decrease the white blood cell count . the proinflammatory response , however , is not purely harmful . some reactions are helpful in tissue repair , and the good may be inhibited along with the bad [ 9 - 12 ] . moreover , suppression of inflammatory responses occurs in all organs , not just the injured ones , and inhibition of immune response can lead to an increased risk of infections . in this sense , hypothermia is a two - edged sword : the mechanism that provides tissue protection can simultaneously hamper the bodies ' ability to fight infections . this problem can be compounded by hypothermia - associated hyperglycaemia , which can further increase the infection risks . many clinical studies have indeed found higher infection rates in patients treated with prolonged ( > 24 hours ) therapeutic cooling . for example , in a recently published study in patients with ischaemic stroke , the rate of pneumonia was 50% in patients treated with hypothermia versus 10% in controls . nevertheless , out - comes were better in the hypothermia group , in spite of these adverse events . most studies on therapeutic cooling have reported trends or significantly higher infection rates in patients treated with cooling , with the risks appearing to increase with longer treatment periods . the increase in infection risk may be even greater with accidental hypothermia . based on the physiological data and the clinical studies discussed above , some increase in infection risk duration of cooling therapy is often limited because of the ( perceived ) risk of infections . few of the clinical trials performed so far , however , have used standard decontamination of the digestive tract ( sdd ) or other forms of antibiotic prophylaxis in their patients . in the previous issue of critical care , kamps and coworkers report on their use of prolonged therapeutic cooling to control intracranial pressure in patients with severe traumatic brain injury , in a setting where sdd was routinely used . they compared infection rates in 35 patients treated with hypothermia ( median duration 107 hours ) with 169 controls matched for severity of injury , age , and other relevant factors . the overall risk of any infection was 20% for hypothermia patients , versus 34.4% in controls . most notably , the risk of ventilator - associated pneumonia was the same in patients treated with hypothermia compared with matched controls . these findings may come as a surprise , but they are in line with two previous studies using prolonged hypothermia in combination with sdd . these studies also reported low infection rates in patients treated with hypothermia and sdd , and found that infection rates were the same or lower than in controls . many studies have shown that sdd can reduce gram - negative infection rates , and some have reported reductions in intensive care unit mortality . the findings of kamps and colleagues suggest that sdd could help avoid one of the most important complications of therapeutic cooling . their findings need to be confirmed in larger , prospective studies , and the efficacy of sdd in environments with a higher incidence of resistant micro - organisms needs to be determined . the study by kamps and colleagues provides an exciting starting point , and opens up possibilities for exploring long - term hypothermia treatments .

in vitro studies and clinical observations suggest that both accidental and controlled / therapeutic hypothermia have a strong immunosuppressive effect , and that hypothermia increases the risk of infections , especially wound infections and pneumonia . in the previous issue of critical care , kamps and colleagues report that when hypothermia was used for prolonged periods in patients with severe traumatic brain injury in conjunction with selective decontamination of the digestive tract , the risks of infection were the same or lower in patients treated with therapeutic cooling . the risk of infection is widely regarded as the most important danger of therapeutic cooling . the findings of kamps and colleagues need to be verified in prospective trials and in higher - resistance environments , but raise the possibility of cooling for prolonged periods with greatly reduced risk . we may be able to have our cake and eat it .

risk of low back pain in the human life span is 80% , while this probability for sciatica is about 50% . usually , when a patient presents with sciatalgia and positive straight leg raising test , most of the physicians referred it to a herniated lumbar disc . in rare cases , physicians are faced with some patients who have obvious clinical sciatica , but their imaging studies indicate normal intervertebral discs . this non - discogenic sciatica can be caused by any space - occupying lesion along the course of the lumbosacral nerve root and the sciatic nerve and therefore it can be arbitrary divided into two general categories : intraspinal and extraspinal non - discogenic sciatica . in this report , we present a rare case of refractory sciatica in an active otherwise healthy 25-year - old man that a diagnosis of varicose veins in epidural space was ultimately confirmed . a 25-year - old man presented to spine clinic with chronic left leg pain for about two years . he declared that analgesics were relatively useless for relieving his pain , but he could still do his ordinary activities . he could lift heavy weights without extra pain , but his maximum walking pain free distance was about 400 meters . he was obliged to take medicine to sleep well . on visual analogue scaling , he pointed the score of 10 for his leg pain , but zero for low back pain . in physical examination , he complained of paresthesis on the dorsal aspect of the foot . we found that his left extensor hallucis longus power was fair accompanying with limitation of straight leg rising to 40 associated with normal achilles deep tendon reflex . his previous conservative treatment consisted of modification in activities of daily living , 30 consecutive physiotherapy sessions along with plenty of anti - inflammatory and analgesic drugs for about two years . his blood laboratory test was normal and in electrodiagnostic study , a moderate degree of left l5 radiculopathy was reported . magnetic resonance imaging has been reported as normal , but we notice a space - occupying lesion in left side of l4-l5 level with a signal intensity that resembled disc material on t1 weighted slices ( figure 1 ) . on the other hand , intervertebral discs were appeared completely normal on t2 weighted images . with a preliminary diagnosis of a synovial cyst or perhaps an old degenerative free sequestrated disc , magnetic resonance images revealed a space occupying mass with high signal intensity on t2 ( left and middle images ) and low signal intensity on t1 ( right image ) weighted slices . the patient was given sufficient explanation about the disease and he signed the informed consent . with minimal longitudinal skin incision , left paravertebral muscles were retracted laterally . with limited laminotomy , vertebral canal was opened and explored with caution . compressed left l5 nerve root was retracted medially and beneath it , there was a bulging flexible mass . with some dissection , disc s appearance and consistency were completely normal and nerve root had a relax posture . the patient s symptoms completely disappeared even on the same day of the operation and he was discharged the day after . in postoperative study , we carried out an outpatient complete clinical and paraclinical examination accompanying with appropriate consultations to rule out any underlying disease ( like hepatobiliary diseases or alike ) but nothing could be found . in the last patient visit , performed 23 months after surgery , he was completely asymptomatic and oswestry disability index improved to 2% ( compared with 36% preoperatively ) . any peculiar pathology along the course of the sciatic nerve from its origin ( lumbosacral nerve roots ) to its bifurcation into tibial and common peroneal nerves can create sciatalgia similar to classic clinical presentation of a lumbar disc herniation . awareness of these possible causes of non - discogenic sciatica and careful attention to the patient s history , physical examination , and paraclinical details can assist the physician in making the correct diagnosis and treatment . some of them include herpes zoster , infection , lumbar nerve root tumor , spinal instability , excessive hypertrophy of the facet joint , ankylosing spondylitis , sacroiliitis , sciatic neuritis , piriformis syndrome , intrapelvic mass ( hypertrophic ossification ) , intraspinal ganglion cyst , endometriosis , adenomyosis , coxarthrosis , et cetera . literature reveals that the presence of varicose vein in the limited epidural space as an underlying cause of sciatica has been reported in some papers previously , but very rare reports of sciatica due to idiopathic epidural varicosis ( without any underlying disease ) have been ever done . secondary epidural varicosis may be due to the different types of inferior vena cava anomalies ( hypoplasia , aplasia ) , obstruction of this vein ( pregnancy or thrombosis ) , portocaval hypertension , compressive lesions in the spinal cord and herniated disc itself . the clinical presentation of both primary and secondary epidural varicosis might be completely similar to a typical lumbar disc herniation ( as in our patient ) . several mechanisms for secondary epidural varicosis have been proposed , but none of them confirmed for primary cases . according to the literature , ideal diagnostic tool for this entity is still magnetic resonance image , although its signal intensities based on its contents ( degree of thrombosis ) can be varied . patent large flowing vessels display decreased signal intensity on both t1 and t2 weighted images , whereas a totally thrombosed vein has an increased signal intensity on both weighted images . in the patient presented , the mass showed an increased signal intensity on t2 and decreased signal intensity on t1 weighted images . although in idiopathic and symptomatic epidural varicosis , some authors proposed coagulative ablation , the preferred approach is surgical resection of the varicose vein ( as we did ) and generally , long - term favourable outcomes have been reported . although the most common cause of sciatica , even in pediatric patients is lumbar disc herniation , with careful attention to the patient s history , physical examination , and paraclinical details , the possibility of non - discogenic sciatica should always be in mind .

non - discogenic sciatica can be caused by any lesion along the course of the lumbosacral nerve roots and sciatic nerve . we aim to present a rare case of refractory sciatica in an otherwise healthy 25-year - old man . he complained of left leg pain without significant back pain . extensor hallucis longus muscle was weak on the left side with limited straight leg rising . on magnetic resonance imaging , a space - occupying lesion resembling a sequestrated disc was noted that after surgical decompression , epidural varicosis was demonstrated .

dissection of the vertebral artery is an important but a rare cause of cerebrovascular accidents ( 1 ) . the annual incidence rate of vertebral artery dissection is estimated to be about 1 - 1.5 per 100,000 population . however it is responsible for 20% of cerebrovascular accidents in young adults and represents about 0.4 to 2.5% of all cerebrovascular events ( 2 , 3 ) . in recent years , this clinical entity has received more attention from neurologists and neurosurgeons , which has allowed its early and further diagnosis . we report a case of dissecting vertebral artery pseudoaneurysm , presenting itself with odontogenic pain principally . a 48-year - old man suffering from toothache and neck pain was referred to the emergency department . he was visited by dentists several times and taken different analgesics , without any improvement . the dental pain had exacerbated and neck pain was added , too . on arrival , he had normal vital signs and was conscious . three - dimensional ct angiography showed the presence of a dissecting pseudoaneurysm of the right vertebral artery ( figures 1 and 2 ) . his post - operative evolution was completely satisfactory and he was discharged after 5 days . atherosclerosis , connective tissue diseases , and cervical trauma are risk factors associated with development of this pathology ( 1 , 3 , 4 ) . the pathophysiology of dissection and pseudoaneurysm basically consist of an initial intimal tear , followed by entry of blood between the intima and the adventitia , which leads to growing and ballooning of the pseudoaneurysm ( 5 ) . therefore , the most prevalent clinical symptoms of vertebral artery dissection consist of neck pain and posterior headache that may follow cerebrovascular accidents related to posterior circulation entity . if extra - cranial dissection extends to the intracranial fossa , and also in cases of primary intracranial dissection , subarachnoid hemorrhage may occur ( 6 ) . some authors have declared that this should be considered as one of the differential diagnoses in patients referred with any cranio - cervical pain . but vertebral dissection presenting with facial pain has rarely been described in the literature ( 3 , 7 ) . modalities such as brain magnetic resonance imaging ( mri ) , computed tomography , and conventional angiography are frequently used for diagnosis . in spite of all that , conventional digital subtraction angiography has remained as the gold standard in terms of diagnostic modality to make the proper diagnosis and have a dynamic evaluation of the blood flow through the visualized defect ( 8) . a three - dimensional reconstruction shows important morphologic information of the lesion ( 9 ) . standard ct and mri with or without contrast enable evaluation of surrounding neurovascular and soft tissue structures and provide a better understanding of the surrounding anatomy ( 10 , 11 ) . surgery could be accomplished by either endovascular technique or surgical trapping with clips above and below . some advantages of the endovascular management are as follows : it is minimally invasive , needs less time for vascular occlusion during repair , avoids devitalized tissue , and can be performed in the presence of previously instituted anti - platelet or anti - coagulant therapies ( 12 - 15 ) . favorable outcome is assumed for younger patients , those who are diagnosed before appearing the stroke , and even in most ischemic strokes . on the other hand , late or missed diagnosis can lead to poor outcome ( 15 - 17 ) . dissection of the vertebral artery is a rare but important cause of cerebrovascular accidents in adults . becoming familiar with atypical manifestations , as in our case , all authors passed four criteria for authorship contribution based on recommendations of the international committee of medical journal editors .

dissection of the vertebral artery is an important but rare cause of cerebrovascular accidents . here we report a 48-year - old man with toothache since 4 days before who presented to the emergency department with neck pain and final diagnosis of dissecting right vertebral artery pseudoaneurysm . to our knowledge , this maybe the first report of odontogenic pain as the first manifestation of vertebral artery pseudoaneurysm in the literatures .

a spinal infection with tuberculosis may be associated with a pulmonary infection , which occurs in less than 1% of patients with tuberculosis or , more rarely , the condition may be encountered in the absence of a pulmonary infection8 ) . the most common locations for spinal tuberculosis , whether primary or secondary to a pulmonary tuberculosis , are the lower thoracic and lumbar spine . on the other hand , infection of the lumbosacral junction by tuberculosis is quite rare , and occurs in only 1 to 2% of all cases of spinal tuberculosis , moreover , isolated sacrococcygeal or coccygeal tuberculosis is much rarer10 ) . as far as the authors ' knowledge , there is only one reported isolated coccygeal tuberculosis5 ) . as uncommon presentations can be a cause of delay and error in diagnosis and management , we report a unique case of isolated tuberculosis of the coccyx . a 35-year - old man presented with a three month history of coccygeal and gluteal pain . furthermore , the pain was associated with numbness in the same region and difficulty walking . he reported no recent weight loss , reduced appetite , cough , or night sweats , and his respiratory , cardiovascular , and abdominal system examinations were normal . no paresthesia or muscle weakness was evident in the legs , which he was able to straighten and raise his legs . the mantoux test and radiographs of the chest were negative , and hematological studies , including erythrocyte sedimentation rate and c - reactive protein were normal . a computed tomography ( ct ) scan of the lumbosacral spine revealed lytic destruction of the coccyx with sporadic calcification ( fig . 1 ) . magnetic resonance images ( mri ) of the lumbosacral spine depicted a coccygeal mass involving the anterior and posterior extension ( fig . radionuclide bone scintigraphy ( 99 m tc mdp ) and positron emission tomography - ct were performed , but both failed to depict any lesion in addition to the lesion in the coccygeal area . a ct - guided biopsy of the sacral lesion was performed , and the patient was given anxiolytic and analgesic injection before and during the procedure . after infiltrating the skin and soft tissues with 2% lidocaine , a 14-gauge biopsy needle was advanced into the lesion and soft tissue and involved bone were aspirated . the solid material aspirated was dark brown in appearance and was forwarded for cytologic and microbiological examinations . the patient was given anti - tuberculosis treatment for 9 months based on four drugs ( isoniazid , rifampin , pyrazimide , and ethambutol ) , which is the standard anti - tubercular regimen used at our institute . patient made a rapid pain relief under this regimen and was asymptomatic at his last follow - up at 9 months . tuberculosis still remains one of the most pressing health problem in developing world , and tuberculosis of the spine occurs by hematogenous spread of infection from a pulmonary or extra - pulmonary site ; pulmonary infection is detected in around 50% of cases of spinal tuberculosis8 ) . spinal tuberculosis is mainly due to hematogenous spread of mycobacteria from primary foci in the lung and or genitourinary tract . it is widely believed that the paravertebral venous plexus of batson provides a primary pathway for the dissemination of tuberculous bacilli into the vertebral column , although it is also possible that lymphatic drainage of the pleura or kidney may involve the para - aortic lymph nodes and secondarily involve vertebrae1,4 ) . after reaching a vertebra , mycobacteria infect the anterior portion of the body near the intervertebral disc , and may subsequently reach the cortex , destroy the intervertebral disc , and fragment the adjacent vertebral body . however , the reasons for the low incidence of lumbosacral or sacrococcygeal tuberculosis have not been exactly elucidated . the sacrum is an uncommon site for tuberculosis involving the spine . in a review of 107 patients of tuberculous spondylitis by lifeso et al.6 ) , no patient had lumbosacral and sacrococcygeal involvement . . young individuals tend to present with discharging sinuses and abscesses , whereas backache is a dominant clinical feature in adults . because the sacral nerve roots are protected by bone our patient with isolated involvement of the coccyx is probably the second described in the literature . mri is the most sensitive diagnostic radiologic imaging , and the radiological spectrum of spinal tuberculosis has been well reported in the literature9,10 ) . however , the sparsity of reports on sacral tuberculosis continues to be a source of diagnostic delays and errors , which is in part due to the lack of specific symptoms and the fact that pathologies in the sacral region can easily be missed if a non - sensitive imaging method is used because of the large number of overlying soft tissue shadows . tuberculosis of the lumbosacral spine may also exhibit an uncommon presentation , where conventional simple radiographs do not reveal any obvious osseous lesion but mr images show the presence of an extradural granuloma or of peridural fibrosis compressing the spinal cord . nevertheless , the enhancement of epidural soft tissue by gadolinium contrast - enhanced mri suggests the possibility of an infection or tumor3 ) . plain radiographs are extremely insensitive , and do not detect vertebral involvement until at - least 50% of a vertebra is destroyed2 ) . similarly , bone scans are notably prone to miss this disease . in the described case , he presented with 3-month duration of coccygeal and gluteal pain . considering the patient 's clinical and radiological features , pyogenic osteomyelitis , tuberculosis , neoplasm such as chordoma or osteogenic sarcoma were included in the differential diagnosis . as tuberculosis lesions may be mistaken for other infectious diseases or neoplasms , fresh tissue should be obtained for culture and biopsy . diagnosis by culture usually takes a longer time , whereas diagnosis can be established rapidly by histopathologic analysis of tissues obtained by percutaneous biopsy . in such cases , it is imperative that such presentations of tuberculosis should be borne in mind because this ensures that correct diagnosis can be made promptly and appropriately . tuberculosis of the coccyx , though uncommon , should be considered when evaluating patients with a coccygeal mass lesion . the use of imaging - guided biopsy to provide tissue cores for histopathologic examination can be an essential component of diagnosis .

isolated tuberculosis of the coccyx is extremely rare . a 35-year - old man presented with a 3-month history of coccygeal and gluteal pain . computed tomography and magnetic resonance imaging revealed osseous destruction and a large enhancing mass involving the coccyx with anterior and posterior extension . pathologic examination of the surgical specimen revealed necrosis , chronic granulomatous inflammation , and multinucleated giant cells consistent with tuberculosis . this case highlights the importance of considering tuberculosis as a diagnosis even though unusual sites are involved .

the case described illustrates the correlation between copd and peptic ulcer disease . in a patient receiving corticosteroids for an acute exacerbation of copd , clinicians need to consider the complications of peptic ulcer disease , paying close attention to subtle changes in abdominal examination ( 1 ) . there have been reports that corticosteroid use and high cortisol levels could mask the peritoneal signs in a gastrointestinal perforation ( 26 ) . our patient presented without typical clinical signs of an acute abdomen , with only mild tenderness to palpation with no rebound or guarding that would classically be seen . theoretically , steroids , with their immunosuppressive effects , could turn down the systemic response to an acute perforation . numerous case control studies have found an association of perforated diverticular disease with corticosteroid use ( 79 ) . the largest study , involving 889 cases of diverticular perforation and 8,980 population controls from 1990 to 2005 , found a threefold increase in diverticular perforation risk with corticosteroid use ( 9 ) . although this patient did not have a formal diagnosis of peptic ulcer disease , his history of copd and treatment for a copd exacerbation placed him at risk for perforation . as mentioned previously , copd frequency in peptic ulcer disease while the duodenal sample was not tested for h. pylori , there is a high likelihood that the patient would have tested positive . in patients with endoscopically confirmed duodenal ulcers , h. pylori has been found to be seropositive in 7394% of patients ( 10 , 11 ) . there is an increase in positive h. pylori serology in patients with copd compared to patients without the disease ( 12 ) . it also appears that the more severe the copd , the higher the likelihood a patient is positive for h. pylori ( 13 ) . beyond being a risk factor , copd has been found to be an independent risk factor for peptic ulcer bleeding regardless of nsaid use or other medical illnesses ( 14 ) . in patients who have perforation or bleeding from their peptic ulcers , having copd peptic ulcer disease places significant economic strain on the healthcare system despite our knowledge of the disease ( 16 ) . understanding the contributory and aggravating factors to peptic ulcer disease is central to lowering costs . additional studies are needed to determine if the severity of copd correlates with increased risk of perforation . the authors have not received any funding or benefits from industry or elsewhere to conduct this study .

gastrointestinal perforations are a complication of 210% of duodenal ulcers . there are a variety of etiologies associated with duodenal ulcer formation and its complications . corticosteroid use is associated with an increased risk of duodenal ulcer perforation , with the first documented case in 1950 . other important medications associated with perforation include nsaids and opioids . beyond medication , one of the most common disease processes , chronic obstructive pulmonary disease ( copd ) , has been found to be associated with peptic ulcer disease . up to 30% of copd patients have been found to have peptic ulcers , and copd frequency in peptic ulcer disease is 23 times the general population . we herein present a case of an acute duodenal ulcer perforation in a patient receiving corticosteroid treatment for an acute copd exacerbation .

cytokines are an intriguing group of soluble protein mediators with a large number of described actions but short biological range . these molecules are produced by a variety of cells , including monocytes , macrophages , lymphocytes , and endothelial cells . such proteins include the pro - inflammatory cytokines interleukin ( il)-6 and soluble tumour necrosis factor receptor-1 ( stnfr-1 ) , as measured by jimenez and colleagues in their study . cytokines have been shown to be important in all biological processes , including inflammation in which the major pro - inflammatory cytokines are considered to be tumour necrosis factor ( tnf ) , il-1 , and interferon - gamma in addition to il-6 . they also have anti - inflammatory roles ( il-10 , transforming growth factor - beta , and il-4 ) . the control of this balance between pro- and anti - inflammatory cytokines is vital to the understanding of inflammation and the inflammatory response in human disease processes . cytokines are normally subject to tight homeostatic control and are produced in response to a variety of physiologic and pathologic stimuli . pro - inflammatory cytokines play a pivotal role in initiating and amplifying the inflammatory process . furthermore , the plasma concentrations of certain cytokines , such as il-1 and il-6 , have been shown to be predictive of outcome in specific subgroups of critically ill patients . in particular , tnf and il-1 are elevated early following cardiac surgery , with il-6 and il-8 peaking later . this damaging pro - inflammatory cytokine response is well documented in patients who develop systemic inflammatory response syndrome ( sirs ) after cardiac surgery . in this sirs patient group , il-8 and il-18 the association of elevated levels of pro - inflammatory cytokines with poor outcomes following cardiac surgery has been demonstrated by many studies , but a direct cause - and - effect relationship has not been demonstrated . the pro - inflammatory cytokine response to injury , including cardiac surgery , is countered by the release of anti - inflammatory cytokines . this anti - inflammatory response is not limited to cytokine proteins alone ( such as il-10 ) but also includes the release of soluble cytokine receptors ( stnfr-1 and -2 ) and cytokine receptor antagonists ( il-1 receptor antagonist ) . this complex balance between pro- and anti - inflammatory molecules is likely to influence outcome following many pathophysiological insults , including cardiac surgery . while in some respects the coagulation cascade and the inflammatory response are separate processes , they are closely interconnected in acute disease . the activation of coagulation is a key component of the acute inflammatory response and vice versa , with the endothelium intricately involved in both processes . pro - inflammatory cytokines are released at sites of local inflammation , leading to activation of the endothelium and initiation of the coagulation cascade . cardiac surgery ( in particular , cardiopulmonary bypass ) induces activation of the immune system by factors that include ( but are not limited to ) contact activation of immunological cells , ischaemia - reperfusion injury , and endotoxaemia . once activated , the immune system amplifies its response with activation of complement , cytokine production , coagulation / fibrinolysis , endothelium , and the cellular immune system . all of these processes , if unchecked , may lead to the development of sirs and a poorer outcome in this patient group . the use of tranexamic acid in the study by jimenez and colleagues to attenuate inflammation reveals a glimpse of how the fibrinolytic and pro - inflammatory responses are interlinked . the interplay of other major pro- and anti - inflammatory mediators is worthy of further study in this patient group ( in particular , tnf and il-10 ) to further elucidate the pathways involved and highlight the biological targets involved in this attenuation of the inflammatory response . their study also , perhaps more importantly , yields a practical , clinically applicable method of influencing these often catastrophic cascades that , once initiated , can lead to significant mortality and morbidity in subgroups of patients following cardiac surgery . in the last two decades , much scientific effort has led us closer to a definition of the molecular basis of the immunological response to biological insults and to the development of specific ' targeted therapies ' such as anti - tnf treatment for rheumatoid arthritis . each additional insight into the control of inflammation and its interaction with other biological responses , such as coagulation and fibrinolysis , has the potential to lead to more effective interventions for the clinician perioperatively and in the management of critically ill patients . il = interleukin ; sirs = systemic inflammatory response syndrome ; stnfr-1 = soluble tumour necrosis factor-1 ; tnf = tumour necrosis factor .

pro- and anti - inflammatory cytokines are elevated after cardiac surgery . the control of the release of these major paracrine proteins is becoming clearer and they have been shown to be involved in the activation of the coagulation / fibrinolysis pathway , among other cascades . the association of a predominance of pro - inflammatory cytokines with morbidity in some patients , particularly following cardiac surgery , is well described but still incompletely understood . clinical studies elucidating how clinicians may influence this cytokine release directly will improve our knowledge of the processes involved and could ultimately show benefit in better outcomes for patients .

the vast majority of urachal epithelial neoplasms are adenocarcinomas with several morphologic subtypes including enteric , signet ring , mucinous , mixed cell subtype as well as adenocarcinoma not otherwise specified . adenocarcinoma from several other primaries may mimic any of these urachal adenocarcinoma subtypes in the bladder or at distant sites . we present a case of metastatic urachal adenocarcinoma in bronchial brushings and review the use of immunohistochemistry in its diagnosis . a 47-year - old female was seen initially in august 2007 with urinary frequency and hematuria . she was treated by partial cystectomy with en bloc resection of the median umbilical ligament and umbilicus . the surgical specimen revealed a 1.1 cm moderately differentiated adenocarcinoma with focal mucin extravasation invading deep muscle . follow - up over the next 4 years were negative for signs of recurrence . in july 2011 , the patient presented with new onset of shortness of breath and stridor . thin prep slide of bronchial brushings showed small sheets of columnar cells with medium size nuclei , mildly irregular nuclear membranes , coarse chromatin pattern , and abundant vacuolated cytoplasm in a background of mucin . these cells lacked cilia and were the predominant population when compared with the sparse presence of ciliated bronchial cells . the lesional cells were positive for ck20 and cdx2 but negative for ck7 ( performed on thin prep slides , figure 1 ) . due to insufficient cytologic material bronchial brushings in thin prep ( a ) scanning view showing columnar cell clusters in mucoid background ( papanicolau stain 40 ) ( b ) rare cluster of bronchial epithelial cells with terminal bars and cilia ( papanicolau stain 400 ) ( c ) many clusters composed of disorganized columnar cells with abundant vacuolated cytoplasm and irregular nuclear contours ( papanicolau stain 400 ) ( d ) cdx2 nuclear positive stain ( 400 ) ( e ) ck20 positive stain ( 400 ) ( f ) ck7 negative stain ( 200 ) the concurrent biopsy and the previous resection specimens showed similar cytomorphologic features and immunohistochemical profiles [ figure 2 ] . based on these findings concurrent bronchial biopsy specimen showing well differentiated mucin secreting adenocarcinoma with vacuolated cytoplasm and irregular nuclear outlines ( a ) h and e , 100 ( b ) cdx2 and ( c ) ck20 are positive ( 400 ) less stringent criteria than previously suggested have been proposed and include location of tumor in the dome / anterior wall , epicenter of carcinoma in the bladder wall , absence of widespread cystitis cystica / glandularis beyond the dome / anterior wall , and absence of a known primary elsewhere . metastases from urachal carcinoma have been reported to meninges , brain , maxilla , ovary , and abdominal wall . the most common sites of metastases were regional lymph nodes and lungs followed by peritoneum , anterior abdominal wall , bone , soft - tissue , and ovary . the time interval between initial diagnosis and metastasis was up to 132.8 months with a mean of 28.7 months . our patient presented 4 years after diagnosis with a lung mass , mediastinal adenopathy , and an endobronchial lesion . at this time , the diagnosis of a second primary was a distinct possibility given the time lapse and lack of regional disease or metastases . the tumor cells were immunoreactive with ck20 and cdx2 and were negative for ck7 and ttf-1 . lack of ttf-1 staining in a well differentiated adenocarcinoma is unusual for a primary pulmonary neoplasm . our differential diagnosis at this stage included metastasis from the patient 's known urachal carcinoma or a second primary from the colorectum . gopalan et al . , noted that all 15 urachal adenocarcinomas were positive for ck20 and variably positive for ck7 and 34be12 . the majority showed cytoplasmic membranous staining pattern for beta - catenin and in one case , there was focal nuclear immunoreactivity . paner et al . , studied 34 urachal adenocarcinomas using a panel of markers ( p63 , ck7 , ck20 , cdx2 , nuclear beta - catenin , claudin-18 and reg iv ) and found that nuclear beta - catenin was the only one that had some value in differentiating urachal adenocarcinoma of enteric morphology from colonic adenocarcinoma . all urachal adenocarcinomas had membrano cytoplasmic staining and only 6% had focal to moderate nuclear staining . the authors caution that moderate nuclear staining can rarely occur in urachal adenocarcinoma and that the frequent membrano - cytoplasmic staining pattern may make nuclear staining evaluation difficult . from these two studies however , the presence of nuclear staining in up to 6% of urachal adenocarcinomas and somewhat problematic evaluation of the stain compromise its specificity . in conclusion , this is the first case report of urachal adenocarcinoma presenting in bronchial brushings . the differential diagnosis in this patient included metastasis from a second primary and metastatic urachal carcinoma . the diagnosis in this case would have been difficult to make and required knowledge of the patient 's history of urachal carcinoma , comparison with the patient 's previous material , and lack of gastrointestinal pathology . as this is case report without identifiers , our institution does not require approval from institutional review board ( irb ) ( or its equivalent ) . to ensure the integrity and highest quality of cytojournal publications , the review process of this manuscript was conducted under a double blind model ( authors are blinded for reviewers and vice versa ) through automatic online system .

urachal carcinoma is rare comprising less than 1% of all bladder carcinomas . metastases of urachal carcinoma have been reported to meninges , brain , ovary , lung , and maxilla . cytologic features of metastatic urachal carcinoma have not been previously reported . we present a case of metastatic urachal adenocarcinoma in bronchial brushings and review the use of immunohistochemistry in its diagnosis . a 47-year - old female was seen initially in 2007 with adenocarcinoma of the bladder dome for which she underwent partial cystectomy . she presented in 2011 with a left lung mass and mediastinal adenopathy . bronchoscopy showed an endobronchial lesion from which brushings were obtained . these showed numerous groups of columnar cells with medium sized nuclei and abundant cytoplasm . the cells were positive for ck20 and cdx2 and negative for ck7 . the cytomorphological findings were similar to those in the previous resection specimen and concurrent biopsy . this is the first case report of bronchial brushings containing metastatic urachal carcinoma . no specific immunohistochemical profile is available for its diagnosis . the consideration of a second primary was a distinct possibility in this case due to the lapse of time from primary resection , absence of local disease , and lack of regional metastases .

they have poor communication with the normal lymph systems and therefore collect lymph.[15 ] the most common location in the mouth is the dorsum of tongue , followed by lips , buccal mucosa , soft palate , and floor of the mouth . these tumors typically present in children younger than 2 years and in a significant number of cases are present at birth.[19 ] this article describes a case of lymphangioma of the tongue and its management . a 5-year - old girl reported to the opd of kothiwal dental college , moradabad with chief complaint of growth on the right lateral border of tongue , associated with biting of the tongue on mastication . her parents noticed it increasing in size along with the growth of the child . on inspection , there was a marked soft tissue growth with numerous papillary and vesicle - like projections which made it appear irregular and granular [ figure 1 ] . the mouth opening was normal and there was no restriction of functions of the tongue . on the basis of history and clinical features a provisional diagnosis of lymphangioma was made and an excisional biopsy under general anaesthesia was planned . right lateral border of tongue showing the soft tissue growth under general anesthesia , a deep suture was placed on the tip of the tongue [ figure 2 ] for retraction . single linear incision was outlined on the base of the elevated mass on the lateral surface of the tongue with methylene blue . initially , the incision was mucosal all around the elevated mass and subsequently it was deepened to excise it completely [ figure 3 ] . retraction of tongue and localization of lesion tongue after excision of the lesion following excision , the tongue was approximated in two layers [ figure 4 ] . on histopathological examination it was confirmed to be a lymphangiomatous lesion [ figure 6 ] . there was no evidence of recurrence of the lesion on follow - up of 4 years . they are believed to arise from lymph sac sequestration and enlarge due to inadequate drainage , from lack of communication with the central lymphatic channels or excessive secretion of lining cells . goetsch in 1938 noted that this sequestered lymphatic tissue forms cysts which enlarge from accumulation of lymph caused by projection of endothelial sprouts from cystic walls . the sprouts further destroy tissue and force the lesion into areas of least resistance , between muscles and vessels , invading tissue planes and causing atrophy , fibrosis and hyalinization of the engulfed tissue . lymphangiomas can be classified histopathologically as : lymphangioma simplex ( composed of small thin - walled lymphatics).cavernous lymphangioma ( comprised of dilated lymphatic vessels with surrounding adventitia).cystic lymphangioma ( consisting of huge , macroscopic lymphatic spaces surrounded by fibrovascular tissues and smooth muscles).benign lymphangioendothelioma ( lymphatic channels appear to be dissecting through dense collagenic bundles ) . cystic lymphangioma ( consisting of huge , macroscopic lymphatic spaces surrounded by fibrovascular tissues and smooth muscles ) . lymphangiomas are classified according to their clinical presentation into macrocystic ( cavities larger than about 2 cm ) , microcystic ( cavities smaller than about 2 cm ) , and mixed ( combining these two types ) . they are always present at birth , but they may go unnoticed until after dentition erupts or even after puberty . however , there have been reports of its occurrence in adults . the frequent site is the anterior two - thirds of the tongue and may enlarge to a great extent that results in macroglossia , which may impair speech and eating and may become life - threatening due to their size or secondary infection . their limited extent usually permits complete surgical excision with various techniques of tongue reduction described by a number of authors . the treatment of lymphangioma depends upon their type , size , involvement of anatomical structures and infiltration to the surrounding tissues . microcystic lesions do not respect tissue planes , are diffuse and difficult to eradicate , whereas macrocystic lesions are localized and easily excised . partial surgical excision , injection of sclerosing solutions ( ok432 ) , electrocoagulation , cryotherapy , embolization , steroid administration , radiation and laser surgery may be the other modalities of treatment of diffuse lymphangioma of the tongue.[179 ] since they do not respond to sclerosing agents , pressure therapy , radiotherapy or any known chemotherapy , they are either tolerated by the patient or treated surgically .

lymphangiomas are developmental malformations and have a marked predilection for the head and neck region . they are benign proliferation of lymphatic vessels and represents hamartomas of malformed lymphatics . the most common location in the mouth is the dorsum of tongue , followed by lips , buccal mucosa , soft palate , and floor of the mouth . in the tongue , they may present as a localized or a diffused growth which may enlarge to cause macroglossia , impaired speech and difficulty in mastication . these tumors are typically present at birth , but may go unnoticed until after dentition erupts or even after puberty . presenting here is a case of localized lymphangioma of the tongue in a five year old child and its surgical management .

it was not until the year 1900 , when karl landsteiner at the university of vienna , discovered why some blood transfusions were successful while others could be deadly . landsteiner discovered the abo blood group system by mixing the red cells and serum of each of his staff . he demonstrated that the serum of some people agglutinated the red cells of other . from these early experiments , he identified three types , called a , b and c ( c was later to be re - named o for the german the fourth less frequent blood group ab , was discovered a year later . in 1930 , landsteiner received the nobel prize in physiology and medicine for his work ( 1 ) . the gene that determines human abo blood type is located on chromosome 9 ( 9q34.1 ) and is called abo glycosyltransferase . the abo locus has three main allelic forms : a , b , and o , as mentioned above and each of them is responsible for the production of its glycoprotein . it is therefore the combination of alleles that are inherited from parents that determines which glycoproteins ( antigens ) are found on persons blood cells and thereby their abo blood type ( 1 ) . as investigations have demonstrated on monkeys ( table 1 ) , human blood groups are very old genetic indicators which have evolved during several million years ( 2 ) . based on the primary races hypothesis , it was thought that in the three major races of man , blood groups a in europe , b in asian , and finally o in south america have been emerged and gradually due to the migration and mixing of the races , became the present situation . but we know that in each continent , the isolated populations are seen that have completely different blood groups . for example , there is relatively high prevalence of blood group o in siberian inhabitants ; also this blood group is very common in some areas of switzerland ( 3 ) . according to another hypothesis , the emergence of all blood groups a and b and their subgroups , are resulted from successive mutations , from a basic and common blood group , which is the o group , and have been branched over millions of years ( fig . the old races have o blood group , such as red indians of south america , and eskimos that among them the frequency of o blood group is between 75100% . while in most of recent ethnic groups a and b blood groups are dominant . in another hypothesis , the first blood group had been ab blood group , which gradually and over the time due to genetic mutations was resulted in a and b and finally o blood groups ( fig . 2 ) . base on this theory , perhaps a few million years ago all people have had type o blood only , which is more resistant against many infectious diseases . geographic distribution and racial blood groups a and b and o in the world ( according to the mourant design 1958 ) are shown in figures 3 to 5(4 ) . the geographical spread not only is a result of the above assumptions , but the current process of natural selection against environmental factors such as diseases , climate , humidity , altitude and etc after discovery of the first human blood groups ( abo ) by karl landsteiner in 1901 ( 5 ) , gradually from 1927 , other blood groups were also discovered and reported which its collection is given in table 2 . it is important to mention that landsteiner together with his american colleague alexander wiener discovered the rh blood group and reported it in 1940 , 1941 . karl landsteiner was born on 14 june 1868 , in vienna , austria ; he died on 26 june 1943 ad , at 75 years old , in the united states . landsteiner in his 17 scientific paper in 1901 reported blood group abo which was displayed at the beginning with the letters abc . in 1930 in addition to the known blood groups ( table 2 ) , nearly twenty public antigens and also sixty - specific antigen or family antigen ( private antigens ) have been reported ( 3 ) . moreover , the main blood groups abo , gradually discovered and reported ( 3 ) which the most notably of them are as follows : a subgroups , including a1 , a2 , a3 , and also rare types a4 , a5 , a6 , z , x , end , boutu , g , i.b subgroups , including b1 , b2 , b3 , and rare types w , x , v , m.subgroups , including o1 , o2 , o3 , and other types such as yy , hh , xx , and bombay . a subgroups , including a1 , a2 , a3 , and also rare types a4 , a5 , a6 , z , x , end , boutu , g , i. b subgroups , including b1 , b2 , b3 , and rare types w , x , v , m. subgroups , including o1 , o2 , o3 , and other types such as yy , hh , xx , and bombay . in a compilation by mourant in 1958 ( 4 ) , referring to a limited and small sampling from iran ( tehran ) by a.ajir was seen , but there was no systematic and comprehensive research about types and frequencies of blood groups , serum proteins , and red blood cell enzymes , found in iran . the first report about the frequency of lutheran blood group in iran was published in 1979 ( 23 ) . after a long study and targeted collection , detailed reports of the frequency of abo blood groups in different iranian ethnic groups was released ( 24 ) . in another study , the frequency of blood groups , serum proteins and red cells enzymes in various iranian populations furthermore , a collection of valuable and extensive cooperation with iran blood transfusion organization , different types of blood groups in various population of iran , was reported . this report included the study of abo and rh blood groups phenotype and genotype frequencies among 291857 individuals and their geographical spread in different provinces of iran ( 26 ) . in this report , in addition to the abo blood groups and rh , genotype and phenotype frequencies of rare blood groups , including kell ( n=5522 ) , daffy ( n=3764 ) , kidd ( n=3650 ) , lutheran ( n=3199 ) , kp ( n=1489 ) , xg ( n=3227 ) , were also presented ( 26 ) . since over 2030 years have passed from that sampling in different provinces of iran , population displacement , and various environmental factors , diseases , immigration , exogamous marriages within different ethnic groups , no doubt that provincial prevalence of blood groups distribution , at this time has changed , too . however , over time , case reports and local frequencies of blood groups in different regions of iran , were prepared and published , including abo and rh blood groups report in population of larestan and lamerd , fars ( 27 ) . ethical issues ( including plagiarism , informed consent , misconduct , data fabrication and/or falsification , double publication and/or submission , redundancy , etc ) have been completely observed by the authors .

the evolution of human blood groups , without doubt , has a history as old as man himself . there are at least three hypotheses about the emergence and mutation of human blood groups . global distribution pattern of blood groups depends on various environmental factors , such as disease , climate , altitude , humidity etc . in this survey , the collection of main blood groups abo and rh , along with some minor groups , are presented . several investigations of blood groups from iran , particularly a large sampling on 291857 individuals from iran , including the main blood groups abo and rh , as well as minor blood groups such as duffy , lutheran , kell , kp , kidd , and xg , have been reviewed .

guinea worm disease ( gwd ) also known as dracunculiasis , is a preventable waterborne parasitic disease affecting rural parts of developing nations of south asia and africa which do not have access to safe drinking water . banwari lal , a 25-year - old man from the jodhpur district in rajasthan , was india 's last reported case of guinea worm disease in july 1996 . annual number of reported cases worldwide has been declined from approximately 900,000 in 1989 to only about 3000 in the year 2009 and about 1000 in the year 2011.[24 ] asia is considered as free of disease . an 8-year - old girl from rural area came with chief complaints of pain in left leg and ulcer on left shin of 15 days duration . she was given anti - tubercular therapy and then multidrug therapy ( mdt ) for leprosy but the ulcer recurred . on examination , there was an ulcer of size 3 3 cm on shin anterior aspect and a healed ulcer just above the present ulcer . a x - ray of left leg showed no evidence of osteomyelitis however , it revealed a linear calcification ending in the ulcer . a provisional diagnosis of gwd was made , on the basis or radiological features long linear chain mail type of calcification typical of calcified gunieworm . as the lesion was chronic and recurrent , we decided to surgically remove the worm . when it was opened , a long linear calcified worm was found which was removed from the subcutaneous plain [ figures 24 ] . x - ray showing linear calcification intra operative demonstration of worm intra operative demonstration of worm case 2 clinical presentation a 25 year old woman came with complaints of nodular lesion left side abdomen for 5 to 7 years [ figure 6 ] . dermatological examination showed large nodular lesion of size 5 cm in diameter left side abdomen , not adherent to the deeper structures . we decided to rule out squamous cell carcinoma ( scc ) and a biopsy was done . lesion was excised and the whole specimen was again sent for histopathology which showed calcified worm . an 8-year - old girl from rural area came with chief complaints of pain in left leg and ulcer on left shin of 15 days duration . she was given anti - tubercular therapy and then multidrug therapy ( mdt ) for leprosy but the ulcer recurred . on examination , there was an ulcer of size 3 3 cm on shin anterior aspect and a healed ulcer just above the present ulcer . a x - ray of left leg showed no evidence of osteomyelitis however , it revealed a linear calcification ending in the ulcer . a provisional diagnosis of gwd was made , on the basis or radiological features long linear chain mail type of calcification typical of calcified gunieworm . as the lesion was chronic and recurrent , we decided to surgically remove the worm . when it was opened , a long linear calcified worm was found which was removed from the subcutaneous plain [ figures 24 ] . x - ray showing linear calcification intra operative demonstration of worm intra operative demonstration of worm case 2 clinical presentation a 25 year old woman came with complaints of nodular lesion left side abdomen for 5 to 7 years [ figure 6 ] . dermatological examination showed large nodular lesion of size 5 cm in diameter left side abdomen , not adherent to the deeper structures . we decided to rule out squamous cell carcinoma ( scc ) and a biopsy was done . lesion was excised and the whole specimen was again sent for histopathology which showed calcified worm . the cases presented here are rare and were difficult to diagnose and physcians may not have seen such case for many years . dracunculiasis ( gwd ) is a parasitic disease caused by the parasite worm dracunculus medinensis commonly known as guinea worm . the mode of transmission is by drinking stagnant water containing copepods that carry gunieworm larvae . guinea worm larvae are eaten by the copepods that live in these stagnant water sources . after ingestion , the copepods die and release the larvae , which penetrate the host stomach and intestinal wall and enter the abdominal cavity and retroperitoneal space . after maturation into adults and copulation , the male worms die and the females migrate in the subcutaneous tissues towards the skin surface . about a year after infection , the female worm induces a blister on the skin , generally on the distal lower extremity , which ruptures when it comes into contact with water , a contact that the patient seeks to relieve the local discomfort . the clinical features include slight fever , itchy rash , nausea , vomiting , diarrhoea , and dizziness . it enlarges and cause severe pain and burning sensation , which relieves on submerging the part in water . acute stage complications include cellulitis , abscesses , septic shock , septic arthritis , while in late stage calcification of worm and joint deformities can occur . acute stage is treated with local care of ulcer , combined with slowly removal of the worm . in chronic stage , rarely the calcified worm may cause recurrent problems as in our case and need surgical removal . the radiological diagnosis can be made easily when the characteristic long linear , serpiginous or coiled , whorled chain mail types of calcification are present in the soft tissues.[46 ] several filarial worms , particularly loa loa and onchocerca volvulus , may calcify but these are much smaller and almost always seen in the hands and feet . either may cause small , coiled masses of calcification and occasionally may be linear , but neither is as large or extensive as the guinea worm . in summary , although considered eradicated long ago physicians should be aware of this rare diagnosis and should have high index of suspicion for early diagnosis and treatment .

dracunculiasis ( guinea worm disease ) is a debilitating disease caused by parasite dracunculus medinensis . the condition is not life threatening , but causes significant morbidity . patients may be sick for several months . dracunculiasis has been claimed to be eradicated in india since 1999 . we report two patients with calcified guinea worm . both patient required surgical removal of calcified worm . the cases document that the parasite can cause chronic recurrent nodules and ulcers .

intravesical administration of bacillus calmette - guerin ( bcg ) has been used in the treatment of non - muscle invasive bladder cancer for nearly 40 years and is one of the most successful immunotherapies for this condition ( 1 ) . however , since it contains a live attenuated strain of mycobacterium bovis and is based on an immune reaction to it , this therapy can occasionally cause serious side effects , such as bcg infection and autoimmunity . we herein report a recent case of henoch schnlein purpura ( hsp ) nephritis associated with this therapy . an 80-year - old man was hospitalized for acute kidney injury ( aki ) presenting with systemic edema , diarrhea , and purpura on lower legs . approximately 1 year prior to this presentation , he had been diagnosed with transitional carcinoma of the bladder and underwent endoscopic resection 8 months before admission , and bi - weekly administrations of connaught strain bcg ( 80 mg ) had been initiated 4 months before admission ( fig . no history of an upper respiratory infection , visible hematuria , pain on urination during bcg therapy , or intake of new medications was noted . bcg : bacillus calmette - guerin , aki : acute kidney injury , scr : serum creatinine he had no fever , arthralgia , or abdominal pain . a physical examination revealed moderate pitting edema and palpable purpura on both lower legs ( fig . blood test results showed a white blood cell count of 17,800/mm , a c - reactive protein ( crp ) concentration of 7.9 mg / dl , and a platelet count of 33.310/mm . the plasma coagulation factor xiii ( f xiii ) activity was 47% ( normal 70 - 140% ) . his creatinine level had risen to 3.6 mg / dl from 0.9 mg / dl 1 month prior . his serum albumin level was 2.6 g / dl , and his electrolytes were normal . the urine protein - to - creatinine ratio was 4.1 g / gcr with several red blood cells ( rbcs ) visible on urine microscopy . anti - nuclear antibody and anti - neutrophil cytoplasmic antibodies were negative , and serum complement levels were normal , while iga was 654 mg / dl ( normal 110 - 410 mg / dl ) . 3a - c ) , and immunohistochemistry revealed iga deposits mainly in the mesangium ( fig . we made a diagnosis of hsp nephritis in conjunction with the clinical course , which was classified as iskdc iiib according to the classification criteria of the international study of kidney disease in children . ( a ) hematoxylin and eosin staining ( 25 ) , showing a crescentic glomerulonephritis in 20% of glomeruli . ( b ) pas stain ( 400 ) and ( c ) silver stain ( 400 ) , showing a crescentic and fibrinoid necrotizing glomerulonephritis with focal rupture of the capillary walls . ( d ) immunohistochemstry ( 400 ) , demonstrating granular staining for iga deposits mainly in the mesangium . scale bars : 200 m ( a ) and 20 m ( b , c , d ) . based on the clinical and pathological severity , he was started on 3 sessions of plasmapheresis and hemodialysis , with pulsed methyl prednisolone ( 500 mg / day for 3 days ) followed by oral prednisolone ( 30 mg / day ) . after that , his symptoms and renal function gradually improved , with his creatinine level decreasing to 2.8 mg / dl . however , he developed catheter - related blood stream infection due to methicillin - resistant staphylococcus aureus ( mrsa ) after 30 days and died of mrsa septicemia after 32 days despite aggressive antibiotic treatment . henoch schnlein purpura ( hsp ) , also termed iga vasculitis ( igav ) , is a systemic small - vessel vasculitis characterized by immune complexes containing iga . the clinical features of adult case are often atypical , and significant renal involvement is more likely to develop than in children , thereby leading to a poorer prognosis ( 3 ) . it is now believed that individuals who have high levels of underglycosylated iga1 with an uncertain second hit aggregate iga1 accompanied by anti - glycan autoantibody , which is then deposited in the small vessels and mesangium , causing inflammation . what constitutes this second hit is unclear , but more than 50% of cases are preceded by infectious disease ( 4 ) . in the present case , the evidence for bcg - induced hsp nephritis is circumstantial but strengthened by the following : first , from a clinical perspective , there was no obvious exposure to an infection other than bcg administration . second , our patient was undergoing intravesical bcg therapy and developed hsp nephritis 4 months after starting the treatment . this clinical course and the temporal relationship between the therapy and the onset of hsp suggested that bcg treatment might be the cause of the hsp . while hsp is occasionally reported after bcg therapy ( 5,6 ) or associated with mycobacterium avium - intracellilare complex ( mac ) ( 7 ) , our case differs from these previous reports in that this case of hsp nephritis after intravesical bcg therapy was diagnosed by renal biopsy . in addition , the pathological findings and decreased f xiii activity of this patient implied strong renal involvement . with respect to the etiology of aki , we speculated that aki was caused by hypersensitivity vasculitis , i.e. , hsp nephritis , rather than direct lesions of bcg . this is because the lesions indicating acid - fast bacillus infection were not detected by renal biopsy , such as acute interstitial nephritis and granulomatous inflammation . the most disappointing point in this case was the outcome , namely the patient 's death due to catheter - related blood stream infection . in a randomized , double - blind , placebo - controlled trial for children with hsp in 2013 , the authors found no evidence to suggest that early treatment with prednisolone reduces the prevalence of proteinuria 12 months after disease onset ; they therefore do not support the routine use of prednisolone to prevent nephropathy in early hsp ( 8) . considering this report , the indication of immunosuppressive therapy should be carefully judged in cases of hsp to prevent renal involvement , particularly in patients who have risk factors for infection . in conclusion , clinicians should be aware of hsp nephritis as a potential complication associated with bcg therapy , since a considerable number of patients receive intravesical bcg administration . conversely , in adult cases of hsp nephritis , clinician should take a thorough history regarding recent bcg therapy , paying attention to renal involvement .

henoch schnlein purpura ( hsp ) , also known as iga vasculitis ( igav ) , is a systemic small - vessel vasculitis that predominantly affects adolescents and is rare in adults . in many cases , the onset of hsp has been causally linked to an infectious disease . we encountered a case of hsp with severe renal involvement diagnosed by renal biopsy following bacillus calmette - guerin ( bcg ) therapy for bladder cancer . this is of clinical relevance , as intravesical bcg administration is becoming an established therapy for superficial bladder cancer and is supposed to be safe . it is important for all clinicians to recognize that bcg therapy has this rare but potentially serious systemic complication .

shear has estimated about 9% while donath about 4% of dentigerous cysts to occur in the first decade of life . developmental type of cyst develops in a mature tooth as a result of fluid accumulation . inflammatory type develops in an immature permanent tooth as a result of spread of inflammation from an overlying non - vital primary tooth . larger cysts may cause springiness of the bone , expansion of cortical plates and mild sensitivity without any pain . radiographically , the cyst presents as a well - defined unilocular radiolucency surrounding a crown of unerupted tooth . histologically , the cyst consists of a fibrous wall lined by non - keratinized stratified squamous epithelium of myxoid tissue , odontogenic remnants and rarely , sebaceous cells . the latter approach is preferred for larger cysts and is especially helpful in pediatric patients to conserve the unerupted permanent successors . a 10-year - old female patient was referred to our department in january 2011 with the chief complaint of painless swelling of one - month duration in the left side of the lower jaw . on extra - oral examination , a slight buccal bulge was present in the mandibular left posterior region [ figure 1 ] which was felt hard on palpation . intra - oral examination revealed the presence of grossly carious mandibular primary left canine ( 73 ) and mandibular primary left first molar ( 74 ) with distally tipped mandibular permanent left lateral incisor ( 32 ) . we advised an orthopantomogram x - ray to the patient which revealed the presence of a well - defined unilocular radiolucency in relation to the roots of 73 , 74 and mandibular primary left second molar ( 75 ) . the radiolucency also involved unerupted mandibular permanent left canine ( 33 ) , first premolar ( 34 ) and second premolar ( 35 ) . we advised the patient to have a computed tomography ( ct ) scan of the region . the ct scan showed a well - defined radiolucency involving 33 , 34 and 35 with expansion of buccal and lingual cortical plates [ figures 3 and 4 ] . the dimensions of the radiolucent lesion were approximately 25.5 36.0 mm . based on clinical and radiological findings , a provisional diagnosis of dentigerous cyst was made . initial preoperative photograph with buccal swelling clinical view showing swelling in relation to 74 and 75 preoperative ct scan showing a large well - defined radiolucent lesion ct scan ( occlusal view ) showing expansion of buccal and lingual cortical plates extractions of 73 , 74 and 75 were done under local anesthesia which led to opening of the cavity . we also got the tissue from the cavity which was sent for histopathological examination showing wall of the dentigerous cyst lined by stratified squamous epithelium having features of inflammation including numerous proliferating blood vessels and mixed inflammatory cells [ figure 5 ] . microphotographs showing ( a ) wall of dentigerous cyst lined by stratified squamous epithelium ( shown by arrow h and e , 100 ) , ( b ) other area of cyst having features of inflammation including numerous proliferating blood vessels and mixed inflammatory cells ( h and e , 100 ) the cortical plates were compressed and the cavity was left open for continuous drainage . a space maintainer was given with an acrylic and wire extension into the cystic cavity to keep the cavity open . space maintainer was removed after six months as there was closure of the cystic cavity as the permanent teeth moved upwards . nine - month follow - up examination in october 2011 revealed the clinical presence of 34 and 35 in the oral cavity [ figure 6 ] and almost complete bone healing [ figure 7 ] . the extra - oral swelling caused by expansion of the buccal cortical plate also disappeared completely [ figure 8 ] . nine months follow - up clinical view with erupted 34 and 35 nine months follow - up ct scan showing almost complete bone healing nine months follow - up photograph with no buccal swelling inflammatory dentigerous cyst ( idc ) is a type of dentigerous cyst which is found in mixed dentition only . it develops when the inflammation present at the root apex of a non - vital primary tooth spreads to involve the follicle of the unerupted immature permanent successor . in our case the infection present at the root apex of a grossly carious and non - vital 74 spread to involve the follicle of 34 resulting in formation of idc . in the differential diagnosis of idc , an odontogenic keratocyst , unicystic ameloblastoma odontogenic keratocyst and unicystic ameloblastoma occur in the second and third decade of life and are found in the molar region of the lower jaw . radiograph alone can not differentiate the above mentioned lesions so a histopathological examination should be performed wherever possible . however , as suggested by kozelj and sotosek in 1999 , leaking out of cystic fluid during an extraction of a primary tooth or during a decompression , respectively , confirm the clinical impression of the cyst . in our case , histopathological examination as well leaking out of the fluid during the extraction of primary teeth confirmed the diagnosis of idc . marsupialization or decompression technique has been advocated by several authors for treating dentigerous cyst in young patients . in this conservative technique , creation of an accessory cavity helps to relieve intra - cystic pressure and accelerate the healing of the cystic lesion . the permanent teeth generally erupt in the oral cavity with or without the need of orthodontic correction . however , the patient should be followed up until the complete eruption of permanent teeth in their designated location . from the foregoing discussion , it is concluded that marsupialization technique is an ideal approach to treat large dentigerous cysts in pediatric patients . however , the follow - up of the patient should be done until the complete eruption of permanent teeth in their right location in the oral cavity . it is observed that , if permanent teeth are left undisturbed then as the cyst heals in due course of time , these permanent teeth erupt in the oral cavity .

dentigerous cyst may be developmental or inflammatory in origin . the latter is found only in mixed dentition with a low frequency . treatment of inflammatory type of dentigerous cyst in children should be done with the aim of saving developing permanent teeth which should not be sacrificed as far as possible . this is a case report of a large inflammatory dentigerous cyst in a 10-year - old female patient treated conservatively by marsupialization method saving all teeth ( mandibular permanent left canine , first and second premolars ) in relation to the cyst .

spinal cord ependymoma comprises less than 2% of all central nervous system neoplasms , 15% of spinal cord tumors , and up to 60% of spinal cord gliomas20 ) . it is an intramedullary lesion and there have been reports of only 17 cases of ependymomas arising in idem locations worldwide1,2,3,4,5,6,7,8,9,11,12,13,15,16,18,19 ) . this is the fourth - reported case of primary idem ependymoma in south korea10,14,17 ) . however , it is the first cervico - thoracic - involved case of idem ependymoma with spinal root attachment . a 36-year - old woman visited the hospital with complaints of progressive paraplegia , occurring for one month , and left shoulder pain of a year 's duration . on the initial neurologic examination , the patient showed lower limb weakness ( grade iii / iii ) in both limbs , hypoesthesia below t5 , and bladder disturbance . magnetic resonance imaging of the whole spine revealed an idem heterogeneously enhanced fusiform mass , about 9.8 cm in length , causing compression of the cord from the level of c6 upper body to t4 mid body ( fig . complete imaging of the rest of the neuroaxis revealed an arachnoid cyst in the right middle cranial fossa . surgeons performed a posterior c6 through t4 laminectomy , which allowed intradural exploration and removal of the idem mass , and a midline durotomy . the tumor was grossly yellowish in color and markedly distorted the spinal cord to the right . histological examination revealed a who grade ii ependymoma with a proliferation index of < 1% measured by ki-67 ( fig . spinal cord ependymoma is an intradural intramedullary lesion most frequently located at the conus medullaris or filum terminale . only rare reports exist of spinal cord ependymomas in idem locations ; only 17 cases of spinal cord ependymomas in idem locations exist in the relevant literature1,2,3,4,5,6,7,8,9,11,12,13,15,16,18,19 ) . the overall prevalence of the majority of spinal ependymomas among males and females is equal but this neoplasm is more common in women ; there were only 4 reported cases of male patients1,3,8,11 ) . although duffau et al.5 ) has postulated the insufficiency of hormonal theory , there are no reports of hormonal assessment . typical clinical features include a slowly progressive myelopathy with weakness and pain according to tumor location . compared with almost all other idem ependymoma patients in the literature who presented with myelopathy and axial pain , the patient in the current case showed a unilateral , radicular symptom(shoulder pain ) in addition to myelopathy , probably caused by root compression from the root attachment . in most previous reports , doctors often diagnosed as schwannomas or meningiomas due to similarities in mri findings and in clinical courses . distinct from the other cases , the current case showed heterogenous enhancement , but intraoperative findings and pathologic findings were undistinguished16 ) . more case reports are needed to clarify the pathogenesis and clinical manifestations of this type of tumor . in most previous reports , there were thin stalks that connected the tumor to the spinal cord . in the case presented here , and in one reported by bonfield et al.2 ) there were spinal nerve root attachments in the intraoperative findings . in the current case , the tumor was removed safely during surgery with an ultrasonic aspirator because of the loose attachment . it appears that this case is the second reported idem ependymoma involving a spinal nerve root2 ) . distinct from the current case , bonfield et al.2 ) decided on the resection of nerve root because of the dense attachment . it is probable that the separated ectopic ependymal tissue during neural tube closure was the origin of the tumor on the nerve root11 ) . since prognosis is directly related to the extent of surgical resection , complete resection of the tumor is the optimal therapy for idem ependymoma . in this case , despite the nerve attachment , no residual tumor appeared in surgical findings or on postoperative magnetic resonance imaging ; therefore , no adjuvant radiotherapy was warranted . if there are tumor remnants or malignant cases , adjuvant radiotherapy should be considered . there were no signs of local recurrence or distant dissemination on mri at the 6-month follow - up and the patient had no neurologic deficits . however , since anaplastic transformation15 ) and recurrence1 ) have been reported in previous cases , regular follow - up evaluations are required . this paper reports a rare case of intradural extramedullary ependymoma with spinal root attachment , which was successfully treated with gross total resection .

a 36-year - old female patient presented with shoulder pain experienced over a period of one year and progressive weakness in both legs for one month . a magnetic resonance imaging scan revealed an intradural extramedullary ( idem ) fusiform mass about 9.8 cm in length , heterogeneously enhanced at the level of c6-t4 with spinal cord compression . at the time of surgery , the surgeon found an encapsulated idem tumor with spinal root attachment . the tumor was completely resected and the histologic diagnosis revealed ependymoma . the patient showed a favorable outcome with no recurrence at the 6-month follow - up . this paper reports a rare case of intradural extramedullary ependymoma with spinal root attachment .

a 60-year - old man was presented to an orthopedician in the operating theatre as a case of difficult urinary catheterization . the patient was going for a lengthy orthopedic operation that required a drained urinary bladder beforehand . the urologist was called and he found the patient anesthetized , having circumcised penis and a pin - hole external urethral meatus ( eum ) at the corona penis level . the tight meatus was negotiated first with a lubricated 4-french ( fr ) bougie urethral dilator followed by subsequent sized dilators up to 14- fr caliber when a proper foley catheter was introduced easily into the bladder . the patient was carefully interviewed to reveal that circumcision was performed neontally as a religious rite , and he has no history of urologic diseases or trouble voiding , as this was the first time for him to meet a urologist . the patient , who is unaware of his condition , is a father of 12 children and never underwent urinary catheterization . hypospadias is a highly prevalent congenital anomaly of the male genitalia . in comprehensive review , sorensen ( 1953 ) credited rennes in 1831 with reporting a prevalence of hypospadias of 1 in 300 recruits and also reported the same figure for live male births in denmark . there is a plethora of hypospadias cases of different degrees of severity repaired in childhood that resulted in different psychological , social , and sexual outcomes . psychosexual effects of hypospadias repair are endured in adulthood although affected men maintain satisfaction in their sexual life . it is unusual in the sense that the elderly man should have had his hypospadias deformity repaired decades ago in order to pass urine freely and to impregnate his spouse with a wider , and properly located meatus . the tight meatus in this case was severe , hardly admitting a lubricated 4-fr bougie dilator . litvak et al . , reported that the meatus in children younger than one year accepts a lubricated 5-fr feeding tube . they also reported that , in children aging 1 - 6 years , an 8-fr feeding tube could pass without difficulty . the social and sexual life of adults operated for hypospadias during childhood has been studied by a few authors . a few publications reported patient and partner dissatisfaction with the appearance of genitalia , as sexual dissatisfaction is often attributed to penile size and curvature . noted that self - reported strength of libido was slightly better for controls compared to patients with hypospadias , but without a statistically significant difference . noted that only about 10% of both patients and controls reported that their libido was low . problems reported include weak or dribbling ejaculation , having to milk out ejaculate after orgasm , quantity of semen passing after intercourse , anejaculation with or without orgasm . liu et al . observed that the rates of ejaculation problems in the distal and proximal groups were 19.5% ( 8/41 ) and 48.6% ( 17/30 ) , respectively [ 5 , 6 ] . the erectile problems in repaired hypospadias may be attributed to surgically correctable and non - correctable causes . more commonly encountered correctable causes include persistent chordee , penile torsion , fistula formation , acquired meatal stenosis , and inadequate cosmetic outcome . similarly , in unrepaired cases , as the eum is distal enough to deposit the seminal fluid high in the posterior vagina with no difficulty , fertility is supposed to be preserved . furthermore , aho et al . found that men who had hypospadias during childhood were less likely to live with a partner , and that they had fewer children ( 0.8 vs. 1.1 ) . figure 1 extremely tight external meatus in a fairly asymptomatic 60 yrs , old man with distal hypospadias . the literature is scant about the urinary , social , sexual , and fertility aspects of unrepaired hypospadias cases . to our knowledge , no other case has been reported for a circumcised uncorrected hypospadias with extremely tight eum in a the finding in this treatise supports the previously published data surrounding the unaffected sexual and fertility aspects of patients having distal hypospadias , and limits repair to cosmetic reasons .

hypospadias is a highly prevalent congenital anomaly . english articles , indexed in pubmed , published the long - term sexual and reproductive outcome following hypospadias repair . almost all repairable cases of hypospadias are operated in childhood . although distal hypospadias does not interfere with fertility , it is worthy reporting a case of an elderly man , who fathers 12 children and has no urologic complaint , presented with unrepaired coronal hypospadias and severely tight external urethral meatus .

a 44-year - old woman was admitted to the hospital for evaluation of pulmonary nodule detected on computed tomography ( ct ) . she had a history of total thyroidectomy for ptc , which was performed 8 years ago . the primary ptc measured 2.41.3 cm in dimension with invasion into adjacent muscle and had metastasized to several regional lymph nodes ( pt4n1 ) ( fig . the patient had taken neck and chest ct scan at an interval of one year . a follow - up ct scan taken after postoperative 6 years demonstrated the presence of two nodules in both the lower lobes of lungs . at the 2 years follow - up ct scan after detection of the pulmonary nodules , there was a slight increase in the size of right lobe nodule from 0.6 to 0.8 cm ( fig . 2 ) . consequently , the patient underwent wedge resections of both lobes . the resected right lower lobe measured 5.83.3 cm in dimension and it contained a small nodule measuring 0.80.5 cm in dimension . microscopically , the main part of the nodule showed typical histologic features of nonmucinous ais . single layer of cuboidal cells grew along the thick - ened alveolar walls in a lepidic pattern . , there was a 0.20.1 cm sized lesion , which exhibited papillary structures with typical nuclear features of ptc . the papillary carcinoma was composed of enlarged cells with clear nuclei and occasional intranuclear inclusions ( fig . , multiple microscopic foci of metastatic ptc were also observed in the surrounding normal lung tissue . immunohistochemical staining for thyroid transcription factor-1 ( ttf-1 ) and thyroglobulin was performed on both thyroid and lung tumor . the tumor cells of the ais of the lung showed immunoreactivity for ttf-1 antibody but were negative for thyroglobulin . the tumor cells in the papillary carcinoma in the ais displayed diffuse positivity for thyroglobulin ( fig . after 11 months of lung resection , no newly developed lesion was observed in the lung . the lung is one of the most frequent site of metastasis in the case of extrathoracic cancers . this phenomenon can be explained in terms of presence of certain features like : 1 ) they continuosly receive the entire right - sided cardiac output with every heartbeat ; 2 ) they are the first capillary plexus met by cells after most of the lymphatic drainage enters the venous system ; and 3 ) they consist of delicate membranes that may be beneficial for initially drawing on nearby oxygenated air for early sustenance.8 however , lung carcinoma is one of the rarest recipients for tumor - to - tumor metastasis . rcc is the most common recipient tumor in tumor - to - tumor metastasis.5 the reason for the relatively high incidence of rcc as a recipient for tumor - to - tumor metastases remains unclear . however , two major mechanisms have been considered to account for the possible explanations for this observed behavior . one is the rich regular network of thin - walled blood vessels of rcc , which renders them more accessible to metastatic tumor cells in the circulating blood , and the other is the high lipid and glycogen content in rcc , which may provide a nutrient - rich microenvironment for the growth of metastatic tumor cells.9,10 based on the above explanations , presence of different vascular structures between normal lung and lung cancer may be the possible reason for the different potential as a site of metastasis . unlike normal lung , lung cancer does n't contain a rich network of small thin - walled blood vessels . the other possible explanation which we considered was that lung cancer is rapidly growing tumor regarding the reason for the relatively high incidence of lung cancer as a donor in tumor - to - tumor metastasis , horie and nomura11 proposed that the rich vascularity of lung carcinoma in association with the probable shedding of tumor cells during respiratory movement might increase the frequency of invasion into the pulmonary vein and subsequently into the general circulation . the tumor emboli from lung cancer have a better chance to reach all the body organs through the left - sided cardiac output than cancers of the other organs . despite the incidence of multiple primary malignant tumors being between 4.2 to 8% regarding the reason for the rarity of these occurrences , rabson et al.2 suggested that neoplasms produce substances , which are locally antagonistic to the other new growth . kikuchi et al.12 considered the biological differences between cancer cells and normal ones and experimentally confirmed the antigenic difference between their membranous structures.5 furthermore , the nutritional requirement of a rapidly growing tumor may be extensive to the point where a nutritive competitive cellular growth ensues.13,14 it is still unexplained whether tumor - to - tumor metastasis is merely of a chance occurrence or is a selective growth within the recipient tumor . our case showed that the metastatic ptc foci were not limited to the ais and multiple microscopic foci of ptc were observed in the surrounding normal lung tissue . tumors that have metastasized to the other tumors are generally widely disseminated in the other regions of the body . till date , no specific type of tumor with a predilection to metastasize to other tumors has been identified . together , we suggest that our case of tumor - to - tumor metastasis is an incidental condition rather than a specific selective growth . such a combination of tumor - to - tumor metastasis ( ptc within ais ) should be differentiated from primary mixed adenocarcinoma , which is composed of ais and papillary adenocarcinoma of lung . awareness about this phenomenon of tumor - to - tumor metastasis and proper immunohistochemical staining such as thyroglobulin are important for correct diagnosis . disease - specific survival of ptc patients with distant metastases at 5 and 10 years was reported to be 65 and 45% , respectively.15 however , if the distant metastases was confined to the lung with small - sized nodule , it showed relatively favorable outcomes.15 in summary , we report a rare case of tumor - to - tumor metastasis to ais of the lung . we believe that rich network of thin - walled blood vessels , good circulation , and less aggressiveness of recipient tumor than donor tumor may be the associated conditions in recipient tumor in tumor - to - tumor metastasis .

the tumor - to - tumor metastasis is a rare event . the lung tumors are the most common donor tumors in tumor - to - tumor metastasis , but are exceedingly rare as a recipient . here , we report a case of papillary thyroid carcinoma ( ptc ) metastasizing to adenocarcinoma in situ ( ais , formerly bronchioloalveolar carcinoma ) of the lung in a 44-year - old woman who underwent total thyroidectomy for ptc 8 years ago . to the best of our knowledge , the present case is the first case reporting on ptc metastasized to ais . a review of the relevant literature is presented .

hibernoma is a rare , benign , slow - growing tumor of brown fat origin the majority occurring in the extremities and the neck . the presacral location is rare , although a few cases in the retroperitoneum have been described in the literature.[25 ] we describe a case of presacral hibernoma with radiologic - pathologic correlation . the imaging findings are nonspecific but , given the rarity of this tumor , when there is a large well - defined heterogeneous retroperitoneal mass with dilated intratumoral vessels , the possibility of a hibernoma should be considered in the appropriate clinical setting . a 36-year - old female presented with abdominal distention and dull , vague , left lower quadrant pain since 2 years . a large solid pelvic mass was found and pelvic ultrasound was requested . on ultrasound , there was an ill - defined large heterogenous mass with increased vascularity seen posterior and superior to the uterus , with poor demarcation from the uterus and the left ovary . the patient being unwilling for mri , ct scan of the pelvis was performed for further evaluation . ct scan revealed a large presacral , heterogeneous , hypervascular mass , measuring approximately 10.6 10.0 5.8 cm and causing compression and displacement of the rectum anteriorly [ figure 1a c ] . this mass had multiple tortuous arterial and venous channels , with the dominant arterial supply being from branches of the inferior mesenteric artery ( i m a ) . on microscopy , there were numerous multivacuolated adipose cells with eosinophilic cytoplasm and eccentric nuclei , consistent with hibernoma [ figure 3 ] . noncontrast ( a ) and contrast - enhanced ( b ) axial ct scans through the pelvis demonstrate a large well - defined , heterogeneous , enhancing presacral mass ( arrows ) , with dilated tortuous vessels ( arrowheads ) . sagittal reformatted contrast - enhanced image ( c ) demonstrates the mass pushing the rectum ( r ) and uterus ( ut ) anteriorly . ub = urinary bladder prone , axial , plain ct scan through the pelvis , with the biopsy needle ( arrow ) in the presacral mass photomicroscopic image of the biopsy specimen shows numerous multivacuolated adipose cells , with eosinophilic cytoplasm and eccentric nuclei as the mass was hypervascular , preoperative embolization of this mass was clinically requested . on selective i m a catheter angiogram , there were dilated vascular branches feeding this mass , with an early tumor blush , but no arteriovenous communication was seen [ figure 4a ] . selective catheter angiogram of the inferior mesenteric artery ( i m a ) , ( a ) shows a feeding vessel ( arrow ) and early tumor blush ( arrowhead ) . selective i m a angiogram , ( b ) after particle embolization of the feeding branch of the i m a shows the embolized feeding branch ( arrow ) with near complete loss of tumor blush ( arrowhead ) on gross examination , the mass was lobulated and well encapsulated , measuring 13.1 12.6 4.2 cm . the cut surface was brownish yellow in color , with multiple coursing vessels and without any areas of hemorrhage or necrosis [ figure 5 ] . cut surface of the gross specimen shows the brownish - yellow color of the mass , with multiple vessels but without any areas of hemorrhage or necrosis hibernoma is a rare , benign , slow - growing tumor of brown fat origin . the tumor is named hibernoma due to its similarity to brown fat , which has a thermogenic role in hibernating animals . the tumor was first termed as pseudolipoma by merkel in 1906 and subsequently renamed hibernoma by gery in 1914 . the imaging features of this rare tumor have been mostly described for lesions occurring the extremities and neck region , with only six cases of retroperitoneal hibernoma described in the english literature.[125 ] to the best of our knowledge , this is the first case report of a presacral hibernoma , describing cross - sectional ct imaging and conventional catheter angiographic feature of this tumor with preoperative vascular embolization . hibernoma is seen in the 20- to 50-year age - group , with a female preponderance . however , a large series by the armed forces institute of pathology ( afip ) did show a slight male preponderance . the intra - abdominal hibernoma , by itself , does not produce symptoms and as a result the tumor is often large at initial presentation , the large size of the tumor causing abdominal discomfort and mass effect on adjacent structures . the usg findings of intra - abdominal hibernoma have not been described , but subcutaneous and intramuscular hibernomas are seen as well - defined heterogenous masses with variable echogenicity ; some may appear as hypoechoic masses , with increased vascularity on color doppler flow . on ct scan , the hibernoma is seen as a large heterogeneous , well - circumscribed mass , with attenuation ranging between fat and muscle . the heterogeneity of the hibernoma is attributed to the presence of varying amount of fat , muscle , and vessels . in our case , the mass demonstrated soft - tissue attenuation , without any definite fat attenuation or necrosis . following intravenous contrast administration , the hibernoma shows heterogeneous enhancement , with hypertrophic branching intratumoral vessels as was seen in our case . retroperitoneal hibernoma causes displacement of the adjacent organs and structures but no infiltration . on mri , hibernoma demonstrates high signal intensity on t1w and t2w images ; however , the intensity is less than that of subcutaneous fat and there is incomplete fat suppression on fat - saturated pulse sequences . the differential diagnosis of a large well - defined heterogenous retroperitoneal and/or presacral mass containing large intratumoral vessels could include liposarcoma , angiolipoma , and hemangiopericytoma . liposarcoma are heterogenous soft - tissue masses with areas of necrosis , hemorrhage , and/or calcification , but without large intratumoral vessels . also , in contrast to the slow - growing well - defined hibernoma , the liposarcoma has a poor margin , with infiltration into adjoining structures . a mature teratoma classically presents as a complex mass containing fat , calcification , rarely a fat - fluid level , without intralesional vascularity . angiolipoma and hemangiopericytoma may have increased vascularity ; however , none of these lesions show both increased signal intensity on t1w and t2w images and large branching intratumoral vessels . on angiography , hibernoma demonstrates dilated feeding vessels , typical tumor blush , and early venous return , as was seen in our case . in the past , angiography was performed for evaluating presurgical vascular anatomy and also to differentiate hibernoma from hypovascular lesions like lipomas and fibromas . these days , ct and mri have superseded the role of conventional angiography in the diagnosis of such lesions . given the nonspecific features on imaging , histological diagnosis is usually required for appropriate planning before surgery . despite the increased vascularity of this tumor , image - guided core biopsy microscopically , hibernoma is composed of multivacuolated adipocytes with granular eosinophilic cytoplasm and eccentric nuclei , univacuolated cells with peripheral nuclei , and abundant capillaries .

hibernoma is a rare benign tumor of brown fat origin . it is found in locations where brown adipose tissue is present in the fetus . these locations include the neck , axilla , thorax , and extremities . retroperitoneal hibernoma is an extremely rare site of presentation . we present a rare case of presacral hibernoma . preoperative embolization of this highly vascular tumor was performed . to the best of our knowledge , this is the first reported case of a presacral hibernoma in the english literature .

being an invasive procedure , it is associated with complications that can be managed conservatively or require surgical intervention . in our case report , we discuss a patient who developed surgical emphysema following a therapeutic colonoscopy without any associated abdominal or chest symptoms and was managed conservatively . this case report highlights one of the rare presentations of colonoscopic perforations and provides a feasible management option for clinicians . a 60-year - old female presented to our endoscopy suite after being referred by her general practitioner for investigation of bleeding and passage of mucus per rectum . her past medical history included being diagnosed with ulcerative colitis 26 years ago during her first pregnancy for which she was treated with steroids and has been relatively healthy since then . this colonoscopy showed quiescent colitis confirmed by biopsy . during the present colonoscopy , she had biopsies taken from her rectum , sigmoid , descending and ascending colon given the findings of pseudopolyps . this was not accompanied by shortness of breath , chest pain or abdominal pain . on examination , her oxygen saturation was 99% on room air , with normal respiratory and heart rate . she had marked crepitus over her clavicles , supraclavicular fossa and most of her neck . her erect chest x - ray revealed air under both hemidiaphragms and significant pneumomediastinum ( fig . 1 ) . a computed tomography ( ct ) scan of her chest , abdomen and pelvis was requested . this ct - scan revealed very extensive spreading of surgical emphysema in the retroperitoneum , extending into the mediastinum with small volumes of free intraperitoneal gas ( figs 24 ) . figure 2.ct scan showing an extensive pneumoretroperitoneum . figure 3.ct scan of abdomen showing free intraperitoneal gas compressing the splenic flexure . this consisted of keeping her nil by mouth for 24 h , intravenous fluids and antibiotics . her cervical emphysema gradually resolved and she was discharged 72 h after presentation without any surgical intervention . the rate of perforation following colonoscopy ranges from 0.03 to 0.12% [ 1 , 2 ] . first , it can result from mechanical perforation with the colonoscope and is usually recognized at the time of endoscopy . reported that advanced age , significant comorbidity , obstruction as an indication for colonoscopy and performance of invasive interventions increased the risk of perforation . also reported that older age , male sex , having a polypectomy and a low volume endoscopic service increased the risk of perforation . our patient presented after a therapeutic colonoscopy with cervical emphysema as the initial complaint without any abdominal or chest complaints even though her imaging revealed both pneumoperitoneum and pneumomediastinum . fortunately , she did not exhibit signs of the fourth pneumothorax and did not require a chest drain . this is in stark contrast to previously published case reports where patients developed pneumothoraces requiring chest tube drainage [ 6 , 7 ] . briefly , air in the retroperitoneum can track upwards entering the mediastinum via the hiatus for the aorta and ivc . the air can then continue along fascial planes into the neck resulting in cervical emphysema . additionally , air within the peritoneum can enter the mediastinum via the oesophageal hiatus or can pass through openings in the diaphragm to enter the pleural cavity resulting in pneumothorax . conservative management of perforation following colonoscopy is a safe and acceptable option in patients not exhibiting signs of peritonitis , adequate bowel preparation or silent perforations . additionally , perforations resulting from therapeutic procedures are usually small and are best managed conservatively .

colonoscopy is an invasive procedure used in the detection of colon cancer , inflammatory bowel disease and investigation of bleeding from the rectum . in addition to diagnostic procedures , colonoscopy also has therapeutic indications such as polypectomy and dilation of strictures . we present a case of a patient who presented with cervical emphysema following a therapeutic colonoscopy . the patient had no abdominal or chest pain , shortness of breath and was managed conservatively . perforation following colonoscopy is a rare complication ; however , it is essential that doctors recognize and are aware of the different presentations and management options for this complication .

a 35-year - old female patient presented with bilateral visual loss of two days duration ( on 16.12.2009 ) with history of consumption of about 150 ml neem oil five days back ( 12.12.2009 ) in an attempted suicide , for which she was taken to the nearby government hospital . she was admitted and treated with induced vomiting after about 2 - 3 hours of consumption . later patient noticed sudden bilateral visual loss and was referred to us for further ophthalmic evaluation . her fundus examination showed bilateral , hyperemic and edematous discs with extension of edema along the superior and inferior temporal arcuate fibers for about 2 - 3 disc diopters from the disc margins . the veins around the disc were mildly dilated and tortuous [ fig . 1 and 2 ] . right eye fundus picture showing disc edema , venous tortuosity with extension of edema along the superior and inferior arcades for about 2 - 3 disc diopters from the disc margins left eye fundus picture showing disc edema , venous tortuosity with extension of edema along the superior and inferior temporal arcades for about 2 - 3 disc diopters from the disc margins complete hemogram was within normal limits , venereal disease research laboratory ( vdrl ) , hiv1 and 2 were non - reactive . a magnetic resonance imaging ( mri ) scan showed bilateral putaminal regions with altered signal , hypointensities in t1-weighted images , hyperintensities on t2-weighted images and hyperintense on fluid attenuation inversion recovery ( flair ) images suggestive of cytotoxic edema due to tissue hypoxia [ fig . 3 and 4 ] . medical therapy was initiated which included methyl prednisolone 1 g intravenously ( iv ) for three days followed by oral prednisolone 50 mg /day for 11 days along with 1000 g vitamin b12 ( injection methyl cobalamin 1000 g intramuscularly ) every five days for eight weeks . bilateral putaminal regions showed altered signal hypointensities in t1w images hyperintensities on t2w images patient did not show any visual improvement after one week of treatment but pupillary reactions improved . during her subsequent follow - up after about 15 days her vision in both eyes improved to counting fingers 1/2 meters and at one month follow - up vision was 20/200 in both eyes and pupillary reactions were normal in both eyes . . 5 and 6 ] right eye fundus picture showing reduced disc edema left eye fundus picture showing reduced disc edema at two months follow - up her vision remained 20/200 in both eyes and injection methyl cobalamin 1000 g intramuscular was continued . neem oil , also known as margosa oil is obtained from the neem plant ( azadirachta indica a. juss ) . neem oil is used as a traditional medicine in india for various diseases like headache , gastrointestinal disorders , as a male contraceptive , menstrual disorders , asthma etc . neem oil poisoning causes vomiting which occurs within minutes to hours following ingestion of the oil . drowsiness and tachypnea with acidotic respiration followed by recurrent generalized seizures the seizures are usually associated with loss of consciousness and coma . in our case , patient had consumed 150 ml of neem oil which had the following contents : neem oil kernel extract in solvent methanol containing 0.15% , azadirachtin she had bilateral toxic optic neuropathy with exaggerated deep tendon reflexes and extensor plantar reflex . mri scan findings were suggestive of acute toxic encephalopathy and it can be considered as a possible etiology for bilateral toxic optic neuropathy . we treated her with injection iv methyl prednisolone 1 g per kg body weight for three days based on case reports on methanol poisoning .

a 35-year - old female was referred to our hospital with bilateral loss of vision of two days duration . she gave history of consumption of about 150 ml of neem oil five days back . examination revealed no perception of light in both eyes . both pupils were dilated and sluggishly reacting to light . her fundus examination showed bilateral hyperemic , edematous discs and also edema extending along the superior and inferior temporal vascular arcade . magnetic resonance imaging ( mri ) scan showed bilateral putaminal regions with altered signal , hypointensities in t1-weighted images , hyperintensities on t2-weighted , images and hyperintense on fluid attenuation inversion recovery ( flair ) images suggestive of cytotoxic edema due to tissue hypoxia . her vision improved to 20/200 in both eyes with treatment after two months . this is the first case report of such nature in the literature to the best of our knowledge .

gastrojejunocolic fistula ( gjf ) is a rare and late complication of the surgical technique of gastrectomy and gastrojejunostomy applied for recurrent peptic ulcer disease . generally gjf is considered to be induced by a stomal ulcer due to inadequate gastric resection , incompleteness of vagotomy and long afferent loop . the most frequent symptoms of such a fistula are upper abdominal pain , severe weight loss , diarrhea , gastrointestinal bleeding and sometimes fecal vomiting . the diagnosis is most reliably and frequently made by barium enema and gastroscopy . in the management of the gjf , the historical approach was 2 - 3-phased operations which included colostomy in order to ameliorate the nutritional status and minimize the mortality of the patient . however today , because of improved parenteral and enteral support treatments and the developments in intensive care conditions , single - stage procedures can be applied and these have been favored to minimize mortality . although the occurence of gjf has decreased remarkably as a result of better medical treatment , the modern management of this condition and the generally accepted surgical treatment strategies must be discussed . a 51-year - old man was admitted to the surgical unit of yuksek ihtisas hospital in october 2006 with complaints of chronic diarrhea , epigastric pain and discomfort , severe weight loss and weakness . his medical history was marked by a gastroenterostomy due to duodenal ulcer disease in 1992 . the patient was cachectic and dehydrated . at physical examination , liquidated intestine loops were sensed in the epigastrium and upper right quadrant . a complete blood count revealed a hemoglobin level of 10.3 g / dl ( normal range 10.016.0 ) and an albumin level of 2.1 g / dl ( normal range 3.44.8 ) in the electrolyte values , the blood sodium level was 132 upper gastrointestinal series assessment confirmed the passage between the corpus of the stomach and the proximal jejunum ( fig . 1 ) . transabdominal ultrasonography and tomography showed liquidated small intestine with edema and liquid between the intestine loops ( fig . 2 ) . gastroscopy showed a previous gastrojejunostomy and a fecaloid secretion from the anastomotic region of the lumen . colonoscopy showed an ulcer mass obstructing the transverse colon lumen and preventing the endoscope from passing to the proximal region . total parenteral nutrition ( tpn ) was started immediately and continued in the postoperative period till the albumin level returned to normal . after improving his state of malnutrition , the patient was taken into the operation room and an exploratory laparotomy was undertaken . at surgery , a radical en bloc resection was performed involving a subtotal gastrectomy , posterior vagectomy , partial transverse colon and jejunum resection . recovery was uneventful and the patient remained well at follow - up ; once his general status had improved , he was taken into the normal service . today , the surgical operation necessity for peptic ulcer disease has decreased dramatically . especially developments in the medical treatments with the usage of h2 receptor blockers , proton pump inhibitors and the eradication regimes for helicobacter pylori play an active role . so the incidence rate of these fistulas has been remarkably lowered because of the decrease in surgery . since fistula formation needs a 20 - 30-year latent period after the initial surgery , this severe and rare complication may occur presently . therefore , gjf is still an important and current complication [ 2 , 3 , 9 ] . the symptoms of gjf are diarrhea with lienteric stools , epigastric pain and discomfort , gastrointestinal bleeding , feculent eructation , fecal vomiting , weight loss and weakness . the most frequently used diagnostic tools are barium enema and endoscopy [ 1 , 11 , 12 , 13 ] . the success rate of barium enema in correctly diagnosing the fistula is approximately 95 - 100% [ 10 , 12 , 13 ] . however , its determination success in the phenomenons which are related to malignancy is low . computerized axial tomography is used to determine this complex fistula and to exlude extraluminal diseases that may define underlying etiology . especially the fistula orifice can be followed by upper gastrointestinal system endoscopy and when necessary , preoperative histological confirmation can be done . because of the fading effect of the disease , in the postoperative period , careful clinical and laboratory evaluation must be carried out . as specified before , in a large majority of these patients , the physical examinations and laboratory studies reveal low to medium malnutrition , anemia and electrolyte imbalance . as soon as possible therefore , tpn or total enteral nutrition ( ten ) should be administered in patients who are suffering from malnutrition and this treatment protocol should continue in the postoperative period . because of the physiologic effects of ten over tpn , it should be the first choice if a nasoenteric tube can be placed into the jejunum . the surgical options and treatments of gastrocolic fistula / gjf have changed over the years . the historical approach was 2 - 3-staged operations even involving a preliminary diversion colostomy in order to ameliorate the nutritional status of the patient and to decrease mortality [ 4 , 6 , 7 ] . in the late 1930s , the three - staged procedures included ( 1 ) colostomy , ( 2 ) resection of the fistula and ( 3 ) colostomy closure . this operation , which is known as lahey 's procedure , was very popular because it was done with lower morbidity and mortality . in the 1960s , it was stated that single - stage procedures could be applied . today , because of the parenteral and enteral support treatments and the developments in intensive care conditions , one - stage resection can be applied , and the mortality rates are getting lower [ 2 , 3 , 8 , 21 , 22 ] . in conclusion , as a result of developments of the agents used in peptic ulcer treatment , gjf incidence has decreased dramatically . in the ancient times , recovering the medical status and staged operations were applied in gjf treatment , but today one - stage resections are preferred if the general status of the patient is suitable . tpn or ten should be administered in patients suffering from malnutrition . in the preoperative diagnosis stage , a possible malignancy must be eliminated .

we herein report the case of a 51-year - old man with gastrojejunocolic fistula . it is one of the late severe complications of gastrectomy and gastrojejunostomy and is considered to be induced by a stomal ulcer due to inadequate resection of the stomach and incompleteness of vagotomy . the main clinical presentation of this condition is chronic abdominal pain , weight loss , diarrhea , gastrointestinal bleeding and fecal vomiting . the diagnostic workup should include barium enema , gastroscopy and sometimes colonoscopy and abdominal tomography for excluding and ruling out the possibility of malignant extraluminal disease . the historical approach of the treatment of this rare entity was 23-phased operations which included colostomy . however today , medical management has recently been recommended as the first - line therapy , with parenteral and enteral support treatments . the preferred surgical approach is single - stage gastrocolic resection and anastomosis and this has been favored to minimize mortality .

cystic echinococcosis of the bone is rare with an incidence of 0.5 to 2.5% , and as compared to other sites , osseous hydatidosis presents late , as it is usually not suspected clinically because of its rare occurrence at this site . bone hydatidosis is difficult to treat and carries high morbidity due to frequent recurrences , especially in certain locations , like ilium and hip , where radical surgery is difficult . we herein report a case of cystic echinococcosis of the hip bone and pelvis for its uncommon site and frequent recurrences depicting a poor prognosis and high morbidity at this site . only a few case reports of long term follow - up of such cases are available in literature . a 31-year - old asian indian female presented to the outpatient department of sir ganga ram hospital , new delhi , india with a history of increasing pain in the right hip and pelvis for the last 3 years . pain was progressively increasing in intensity , radiating to the right lower limb and back , increased on movements and associated with tingling sensation since last 3 months . her past history showed that at 22 years of age , she had presented with a large abdominal mass . her ct at that time showed a multicystic lesion measuring 18.09.87.6 cm in the right abdomen , which appeared to be arising from the right ovary . the liver and other organs , including the uterus , kidneys , and left ovary , were normal . thus , with a clinical diagnosis of a right ovarian tumour , she underwent surgery which showed a hydatid cyst which was confirmed on histopathology . peroperatively her ovaries were normal with presence of paraovarian cystic masses which were densely adherent to the right ovary . after remaining asymptomatic for 3 years , in 2007 , she again gradually developed pain in the right pelvic region . this time mri showed a large solid cystic mass measuring 106.3 cm in the right iliac fossa and right gluteal region . it was arising from the right iliac bone , involving right sacroiliac joint and adjacent marrow of the sacrum . all other pelvic organs were unremarkable . with a clinical possibility of tumour or tuberculosis , two cystic pieces were removed which were diagnosed as hydatid cyst . in view of the complicated nature of cysts and location it could not be completely excised . the patient continued with the pelvic pain although postoperative mri showed reduction in size of the lesion along the right iliac bone and sacroiliac joint . the x - ray of the pelvis and hips revealed cortical irregularity with ill defined lytic defects and linear areas of patchy sclerosis along the right iliac blade ( fig . she continued with pain and follow - up ct reviewed after 8 months revealed extension of the disease . the patient was then referred to our hospital . on physical examination , 2 ) involving the medullary cavity of the right iliac bone resulting in irregular lytic destruction and extending across the right sacroiliac joint into the right half of the sacrum , right sacral foramina , and adjacent soft tissues . resection of the hydatid cyst of sacroiliac region with allograft and autograft ( rib graft ) with lumbosacroiliac fixation was done . fragments of the osseous tissue with hyaline and laminated membranes of hydatid cysts were seen admixed with lymphocytes and monocytes ( fig . focal areas of necrosis , calcification , palisading histiocytes , and numerous foreign body granulomas were observed in the intertrabecular spaces . the serological and molecular identification of the parasite , however , could not be performed due to non - availability of the facility in the hospital . the postoperative period was uneventful and patient was discharged on the 10th day of surgery . the follow - up of the patient at 6 months showed no detectable abnormality on radiology , and she was doing well . cystic echinococcosis is caused by a parasitic tapeworm , echinococcus . in humans , 3 forms of echinococcosis are known to occur ; cystic echinococcosis ( ce ) , caused by echinococcus granulosus , alveolar echinococcosis ( ae ) , caused by echinococcus multilocularis , and polycystic echinococcosis ( pe ) , due to echinococcus vogeli or echinococcus oligarthrus . other organs , which may rarely be infected , are the kidney , spleen , muscles and skin , and bones . bone involvement of echinococcosis rarely occurs . occasional cases have been reported in the rib and humerus.the disease in the bone starts when scolices settle there . most of the patients become symptomatic at an advanced stage with symptoms of pain and tenderness at the affected site and rarely with fracture . mri is the most helpful of all the imaging modalities and should be done to evaluate the extent of the disease and planning of the treatment . immunological studies are not much helpful . at times , however , radiology may mimic a variety of benign and malignant lesions of the bone . in all these cases the principal treatment of cystic echinococcosis of the bone is surgery with wide excision followed by mebendazole therapy . bone hydatidosis commonly present with recurrences as was seen in the present case leading to high morbidity . the results of treatment of osseous hydatidosis are satisfactory only in locations where complete and wide excision is possible . in the pelvis and hip , where radical surgery is almost impossible , the results are disappointing even after aggressive treatments , hence , they present with frequent recurrences and a long term follow - up is therefore recommended . the purpose of this study is to be aware of this disease at this rare location . it also highlights that a more aggressive treatment is needed to minimize recurrences in bone involvement by cystic echinococcosis and a long - term follow - up of these patients needs to be done .

hydatid cysts commonly affect the liver and the lung . however , they rarely involve bones with vertebral column . we hereby report a case of a female patient with cystic echinococcosis of the hip bone and ilium . she presented with a long history of frequent recurrences highlighting the dismal prognosis at this rare site . resection of the hydatid cyst from the sacroiliac region was done with allograft and autograft ( rib graft ) with lumbosacroiliac fixation . follow - up of the patient at 6 months showed no detectable abnormality on radiology and the patient was doing well .

mitral regurgitation ( mr ) represents more than 30% of native valve disease [ 1 - 3 ] . the good results of surgery , particularly valve repair , set a high standard for future percutaneous valve intervention [ 4 - 6 ] . however , in current practice worldwide , surgery is denied in many patients with severe valvular heart disease and symptoms and mitral valve repair is not always used or feasible . in addition , surgical risk remains high in a significant percentage of contemporary patients who are elderly and have comorbidities . two main techniques can be used for percutaneous mitral valve repair : the edge - to - edge technique and prosthetic ring annuloplasty . the edge - to - edge technique mimics the surgical alfieri procedure , which creates a double mitral orifice by means of a few stitches securing the two leaflets together in their midpart . this technique can be performed percutaneously using a clip ( mitraclip ; evalve , inc . , menlo park , ca , usa ) made of cobalt - chromium alloy and covered with polyethylene terephthalate . another design involved the use of one or more sutures deployed via a catheter - based device . the technique requires general anaesthesia and a transseptal approach with continuous fluoroscopic and transoesophageal echographic guidance to catch the leaflets at their coaptation point ( figure 1 ) . these constraints mean that the technique is initially difficult to learn and will limit its dissemination . left panel : right anterior oblique view . the clip is opened in the left ventricle , just below the mitral valves under transoesophageal echocardiographic guidance . the clip is closed at the coaptation point of the mitral valves and then released . current clinical experience is focused almost exclusively on the clip technique ( mitraclip ) in 500 patients because the development of the other , suture - based device has been abandoned . in the initial everest ( endovascular valve edge - to - edge repair study ) registry including 107 patients , severe mr was of degenerative origin in around 80% of patients . sixty - four percent of patients were discharged from hospital with a clip and mr of less than grade 2/4 , and the procedure did not induce mitral stenosis [ 14 - 17 ] . the procedure was well tolerated and 98% were free from any complication in the expert centres involved in the study . after 36 months , 66% of patients who underwent a successful procedure were free from death , mitral valve surgery , and mr greater than grade 2/4 . preliminary studies also report satisfactory outcomes in patients at high risk or in functional mr subsets . limitations of the edge - to - edge technique can be anticipated : this technique , in isolation , does not address the problem of annulus dilatation , which has been shown to increase the risk of secondary residual regurgitation . in degenerative mr , its use will probably be restricted to a limited percentage of patients . prosthetic ring annuloplasty is key in most cases of surgical valve repair ; data show that the diameter of the mitral annulus can be reduced by inserting a constraining device in the proximate coronary sinus . several devices are under investigation : the carillon device ( cardiac dimensions inc . , kirkland , wa , usa ) uses a nitinol wire with proximal and distal anchors ; the viacor device ( viacor , inc . , wilmington , ma , usa ) leads to a decrease in anteroposterior diameter through the application of pressure on the midportion of the posterior leaflet by using several rigid elements placed in a plastic sleeve . these two devices can be withdrawn if efficacy is not satisfactory or if the circumflex artery is compromised . in the monarc device ( edwards lifesciences llc , irvine , ca , usa ) , prosthetic ring annuloplasty is easier to perform than the edge - to - edge technique and requires a catheterisation of the coronary sinus through the jugular vein and the delivery of the devices under fluoroscopic guidance ( figure 2 ) . a left anterior oblique view of a clinical case of coronary sinus prosthetic percutaneous annuloplasty is shown . a proximal stent is deployed in the proximal part of the coronary sinus , and the distal stent is deployed in the distal part of the coronary sinus . the preliminary results obtained in around 200 patients with functional mr suggest that the feasibility ranges from 40% to 80% and that safety is satisfactory ( 83% of implanted patients are event - free at 90 days ) . after 2 years , preliminary studies show that 80% of the patients survive and most of them experience functional improvement . data on efficacy are more limited and show a trend toward reduction of the degree of mr . in addition , up to 30% of patients had some degree of circumflex cinching , but this led to adverse events in only a few cases [ 19 - 21 ] . the ring inserted into the coronary sinus is localised to the posterior half of the annulus , where surgeons usually use undersized and complete rings . the coronary sinus is 1 or 2 cm away from the atrioventricular groove on the atrial side , and this may also decrease the efficacy . finally , there is a potential risk of cinching the circumflex artery whilst crossing the coronary sinus . several other devices and techniques are currently being investigated experimentally or tested in humans for the first time : direct ' annuloplasty ( such as transventricular suture - based annuloplasty or subannular annuloplasty ) , and mitral cerclage ' annuloplasty , in which ventricular remodelling using either transpericardial ventricular remodelling with anchoring pads on either side of the ventricle or a combination of anchors placed in the coronary sinus and the atrial septum allows a tensioning cable to be placed across the left atrium , applying tension to reduce the septal lateral dimension . the data we have on these new techniques are still limited and suffer from a number of methodological limitations . a lot of questions are still pending : more clinical data are needed to accurately assess efficacy , safety , and durability . the everest ii randomised study comparing the percutaneous edge - to - edge technique with surgery will be presented soon and further safety and efficacy studies are to be conducted with percutaneous annuloplasty . it is essential to know whether secondary intervention , either percutaneous ( including resynchronisation ) or surgical , will be feasible . imaging guidance using three - dimensional transoesophageal echocardiography will improve the performance of the edge - to - edge technique . overall , it is unlikely that these new percutaneous techniques will reproduce the current results of surgical valve repair ; however , they may play a role in patients with contraindications to or high risk for surgery .

the new percutaneous mitral valve repair techniques are at an early stage . preliminary series show that they are feasible ; however , they need to be further evaluated in comparison with contemporary treatment to accurately assess their efficiency . potential applications may benefit high - risk patients after thorough evaluation .

a 27-year - old male patient presented to us with painless hard mass in superonasal quadrant ( snq ) of left orbit for 3 months which had progressively increased in size during 1-month associated with drooping of upper lid of left eye for 20 days [ fig . 1 ] . photograph of patient showing visible mass at superonasal quadrant of left orbit on examination , best corrected visual acuity was 20/20 in both eyes . anterior segment and posterior segment of both eyes were normal except for approximately 2 mm downward and 2 mm outward dystopia of left eyeball . exophthalmometry of both eyeballs measured by hertel 's exophthalmometer were od-14 mm and os-15 mm and interpuppilary distance was 62 mm . computerized tomography scan report showed approximately 13 mm 14 mm sized well defined minimally enhancing soft tissue density lesion in snq of left orbit . the lesion was found in extraconal compartment causing downward and outward displacement of the left eyeball . scalloping and erosion of the lateral wall of left frontal sinus were noticed [ fig . 2 ] . approximately 13 mm 14 mm sized minimally enhancing soft tissue density lesion in superonasal quadrant of left orbit on computerized tomography plates . note scalloping and erosion of lateral wall of left frontal sinus patient was operated for left - sided anteromedial orbitotomy through lid crease approach under general anesthesia . though mass was found to be adherent to the bone , it was separated in toto by blunt dissection and sent for histopathological examination . mass was a well - circumscribed nodule measuring 1.5 cm 1.8 cm , having glistening white multilayered appearance [ fig . 3 ] . the tissue was composed of round to ovoid nuclei in lacunar spaces suggesting cartilaginous differentiation with little pleomorphism and no mitotic figures [ fig . 4 ] . immunohistochemical studies were positive for s-100 protein consistent with a cartilaginous tumor and for vimentin , consistent with a mesenchymal lesion , suggesting benign cartilaginous tumor - chondroma . gross specimen of resected mass hematoxylin and eosin stained slide photograph showing mature chondrocytes in lobulated manner photograph of patient after 1-month patient was advised to follow - up after 6 months or in case of recurrence of symptoms . chondromas of head and neck are extremely rare with an estimated 10% occurrence in this region . the sites of predilection in the head and neck region include ethmoid sinus ( 50% ) , maxilla ( 18% ) , nasal septum ( 17% ) , hard palate and nasopharynx including sphenoid sinus ( 6% each ) , and alar cartilage ( 3% ) . though chondroma belongs to cartilaginous structure , it can develop from the nest of growth plate cartilage that have become entrapped in the medullary canal and lead to hamartomatous proliferation later on . orbital chondromas are fairly distributed throughout all age groups ; however , incidence occurs most frequently in the third and fourth decade with no sex predilection . had reported only one case out of 1373 and 627 total mesenchymal orbital tumors respectively . study of the incidence of primary orbital bone tumors conducted by rootman and connellet al . revealed only one case of chondroma out of 62 primary orbital bone tumors over a period of 24 years . overall , the incidence of chondroma peaks during the sixth decade of life . however , in the facial skeleton , chondroma generally occurs during adolescence and early adulthood as seen in our case . pasternak et al . also reported orbital chondromas in 19-year - old male and 25-year - old woman respectively . however , harrison et al . has also reported an orbital chondroma in 9-year - old boy . recognizing a chondroma as a benign lesion can be challenging . according to batsakis et al . , histopathological distinction between a chondroma and a low grade chondrosarcoma is notoriously difficult as many of the fine structural features of low - grade chondrosarcoma cells are also found in cells of normal hyaline cartilage . ( 1970 ) pointed out that because of many of the well - differentiated tumors were erroneously diagnosed as benign cartilaginous neoplasms , multiple blocks from cartilaginous tumors should be examined since areas diagnostic of chondrosarcoma may be noted only focally . because of the discrepancy between the histological picture and biologic behavior , chondrogenic neoplasm should be considered potentially malignant . benign cartilaginous tumors are radio resistant , but radiotherapy may be offered for the treatment of primary and recurrent malignant cartilaginous tumors . as chondroma has a tendency to sarcomatous change , even in histopathologically proven benign tumors a long - term follow - up is strongly advised . the prognosis of such tumors is good , and recurrence is uncommon with appropriate treatment .

while relatively common in the skeletal system , cartilaginous tumors are rarely seen originating from the orbit . here , we report a rare case of an orbital chondroma . a 27-year - old male patient presented with a painless hard mass in the superonasal quadrant ( snq ) of left orbit since 3 months . on examination , best - corrected visual acuity of both eyes was 20/20 , with normal anterior and posterior segment with full movements of eyeballs and normal intraocular pressure . computerized tomography scan revealed well defined soft tissue density lesion in snq of left orbit . patient was operated for anteromedial orbitotomy under general anesthesia . mass was excised intact and sent for histopathological examination ( hpe ) . hpe report showed lobular aggregates of benign cartilaginous cells with mild atypia suggesting of benign cartilaginous tumor - chondroma . very few cases of orbital chondroma have been reported in literature so far .

a uterine manipulator is normally inserted through the cervix into the uterus before surgery , which sometimes is not possible in patients with a myoma bulging out of the external os of the uterus into the vagina . the primary 10 mm trocar may be inserted at a point 2 - 3 cm above the umbilicus or in the palmer 's area in such cases . we prefer three 5 mm trocars ( one midline and two lateral ) in the lower abdominal region . a step worth considering in huge cervical myomas is bilateral uterine artery ligation before myomectomy or hysterectomy . the uterine can be approached by opening the broad ligament at round ligament and identification and ligation at its origin from the internal iliac artery through retrograde umbilical ligament tracking . an average of 20 u / ml diluted in 200 ml of saline can be laparoscopically injected into the myoma in the presence of a posterior uterine wall myoma and in the layer between the myoma and the serosa , in an attempt to reduce operative blood loss . the course of the ureter is confirmed with special attention given to the sacral uterine ligament attachments , to determine the displacement in the relative position of the uterine arteries or ureter . at times when the myoma is large and boundaries difficult to confirm , the broad ligament is opened with an anterior approach to delineate the myoma . in case of an anterior wall type , a transverse incision is made in the uterovesical pouch and peritoneum , to perform a blunt dissection of the bladder . when performing traction and enucleation with a laparoscopic myomectomy screw , the base of the wound is held with a bipolar grasping forceps and simultaneous complete homeostasis is achieved along with dissection to avoid post enucleation difficulties due to retracted capillaries . in case of posterior myomas we prefer a midline vertical incision to avoid injuring the vessels and to stay at a safe distance from the ureters [ figure 1 ] . posterior myomas we prefer a midline vertical incision to avoid injuring the vessels and to stay at a safe distance from the ureters after the cervical myoma is completely enucleated , morcellation is performed . if complete enucleation is difficult with large myomas and there is limited pelvic space for traction , the myoma is sometimes morcellated when it is still attached to the uterus . the most difficult part of cervical myomectomy is suturing the base of the wound following enucleation . complete dissection of the surrounding organs such as the bladder and ureter , near the base of the wound , is difficult . bleeding makes it difficult to maintain the visual field , thereby increasing the possibility of damage during suturing . a bottom - up procedure makes it possible to perform suturing safely by providing a complete visual field [ figure 2 ] . saturing of resultant defect the resultant defect is sutured at intervals of about 1 cm with 1/0 or 2/0 absorbable suture material . for the second layer , continuous sutures of the muscle layer can be made with 1/0 sutures for the first layer of cervical mucosa . it is recommended to close the peritoneum also with sutures , to restore the anatomy . an additional difficulty may be the introduction of conventional manipulators during total laparoscopic hysterectomy , where a myoma screw with additional port may be utilized , to manipulate the uterus . a reasonable option is to perform myomectomy and correct the distortion of the anatomy before proceeding to the hysterectomy , in selected cases . when performing hysterectomy with myoma in situ , approach the broad ligament on the side with less distortion first and try to tackle the uterine simultaneously on that side . after release of the uterosacrals on the less affected side , normally , reasonable mobility of the uterus is gained so as to be at a safe distance from the vital pelvic organs on the contralateral side ( where myoma is projecting ) when performing the dissection and coagulation . prior identification of the ureters with either retroperitoneal dissection or pre - procedure cystoscopic ureteric stenting may be of help in selected cases of very large cervical myomas with lateral projection . in cases of anterior myomas thus , after considering the location of the myoma , cervical myomectomy or hysterectomy can be performed safely by developing a uniform strategy that uses a fixed operative procedure , if sufficient attention is paid to the following points [ figure 3a and b ] : ( a ) uterus with cervical myoma , ( b ) bladder dissection reducing the size of the myoma with preoperative gnrhdetermining the positional relationship between the myoma and the surrounding organsoptional blocking of the uterine artery blood flow temporarilysuppressing bleeding during myomectomy with the use of vasopressinminimizing the risk of damaging the surrounding organs by properly positioning the incision in the myometriumsuturing the bottom of the wound after grasping with the forceps , to avoid making a dead spaceprior myomectomy before attempting definitive steps for hysterectomy reducing the size of the myoma with preoperative gnrh determining the positional relationship between the myoma and the surrounding organs optional blocking of the uterine artery blood flow temporarily suppressing bleeding during myomectomy with the use of vasopressin minimizing the risk of damaging the surrounding organs by properly positioning the incision in the myometrium suturing the bottom of the wound after grasping with the forceps , to avoid making a dead space prior myomectomy before attempting definitive steps for hysterectomy

compared to myomas that occur in the uterine corpus , cervical myomas are closer to other organs such as the bladder , ureter , and rectum , and the approach needs to be modified , as the organs that have to be considered differ depending on the location of the myoma . surgical difficulties associated with these cases are , poor access to the operative field , difficulty in suturing the repairs , increased blood loss , and distortion of the anatomy of the vital neighboring structures in the pelvic cavity .

replantations for major amputations of the upper limb have been widely reported and performed at many centres around the world . we report a case of successful replantation of complete avulsion amputation of the right upper limb at scapulothoracic level in a 3-year - old boy . in our literature search a 3-year - old boy was brought to the emergency room around 3 h after sustaining an injury while playing in the fields . his right upper limb accidentally got caught in the belt of a thresher machine and was avulsed from the chest wall . the child was taken to a nearby hospital with the amputated part [ figures 1 and 2 ] from where he was referred to our hospital . amputated limb ( dorsal aspect ) amputated limb ( ventralaspect ) on arrival , the child was stable and the amputated limb was well preserved . the patient was evaluated to rule out other injuries and prepared for surgery while an x - ray was done for the amputated part [ figures 3 and 4 ] . x - ray of the amputated part on examination , the skin disruption was found to be at axillary level while the upper limb had avulsed from the chest wall along with the scapula and its attached muscles ( scapulothoracic dissociation ) . the deltoid muscle was disrupted from its origin and found in the amputated stump with the skin retracted distally over it . the cut ends of brachial artery and its accompanying vein were found retracted under the biceps muscle . the brachial plexus was disrupted at cord level with distal cut ends of medial , lateral and posterior cords found alongside the transected vessels . the posterior cord was found to be of longest length suggesting higher level of avulsion . after thorough cleaning of the amputated limb , the brachial artery and its accompanying vein were dissected out and tagged . the brachial artery was cannulated , and infusion of cold heparinised saline ( 5000 units in 500 ml ) started . meanwhile , the patient was shifted to the operating room and anaesthetised . on exploration of the amputation stump , the axillary artery was found to be transected after the branch to the latissimus dorsi muscle . the posterior cord was not found in the wound , and an incision was given in the supraclavicular region for exploration of the brachial plexus . however , the proximal end of the posterior cord could not be found . since the level of transection of the axillary artery was after the take - off of the thoracodorsal and circumflex scapular arteries , the anticipated blood supply to the scapular muscles after replantation would have been doubtful . furthermore , the approach to debride the subscapularis muscle in the event of necrosis following replantation would have been cumbersome and dangerous . the supraspinatus and infraspinatus muscles were left attached to the scapula as even in the scenario of necrosis of these muscles following replantation , they would potentially be easily approached from the dorsal side . the amputated limb was then brought into the operative field [ figure 5 ] . the dislocation of shoulder joint was reduced and fixed with a 1.5 mm k - wire . the fracture of glenoid neck was reduced and stabilised with two 1.5 mm k - wires . acromioclavicular joint was fixed by a k - wire , and acromioclavicular joint capsular repair was performed by figure of 8 non - absorbable sutures . the associated elbow dislocation was managed with closed reduction , and the fracture of ulna was managed by splinting . amputated limb following fasciotomy and prepared amputation stump axillary vessels were dissected to the healthy end . since no bone shortening was feasible in this case , an end - to - end vascular anastomosis was not possible . an 18 cm long saphenous vein graft was harvested from the left lower leg to reconstruct the segmental defects of the axillary artery and vein ( 8 cm each ) . the total ischaemia time was nearly 8 h ( 1 h warm/7 h cold ) . the distal end of the posterior cord was anastomosed end to side with the proximal medial cord . there was persistent ooze from the scapular side , which was packed with laparotomy sponges . intraoperatively , the patient was given unfractionated heparin and 3 paediatric units of blood were transfused . after 48 h , the sponge packs were removed from scapular site in the operating room and no further oozing was encountered . post - fasciotomy raw area was covered with split - thickness skin graft on day 20 . the k - wires were removed on day 90 in the outpatient department , and the arm supported in a sling , and physiotherapy started [ figures 6 and 7 ] . the last follow - up at 18 months post - operative showed evidence of recovery of crude touch sensations up to the digits . one year post - operative ( frontal view ) one year post - operative ( oblique view ) nonetheless , they are worthwhile as reported in a large series by sabapathy et al . in 2007 as patients with successful replants put the hand to greater use . furthermore , as reported by otto et al . in 2015 , replantation of a traumatically amputated arm leads to good function and higher satisfaction rates than a prosthesis . although few reports are available of successful shoulder level replantation , there is none for scapulothoracic level . although venkatarama et al . in 2015 have reported a case of scapulothoracic avulsion amputation in a 18-year - old male , the replanted limb had to be amputated due to ensuing sepsis . hence , to our knowledge , this is the first reported case of a successful replantation of a limb separated at scapulothoracic level . literature is rife with reports of cases with scapulothoracic dissociation which is a closed avulsion injury caused by a severe traction injury and associated with massive soft tissue swelling of the shoulder , lateral displacement of the scapula and severe neurovascular injury . scapulothoracic muscles are devascularised and have to be debrided . with no muscles being available for scapulothoracic fixation bone shortening is not possible , and therefore , vascular reconstruction would always involve vein grafts . however , with the patient being a child , a reasonable degree of neural recovery is expected . furthermore , the replanted upper limb at this level would be better than any available prosthesis . the child and family have been saved the stigma of an amputation which can have serious psychosocial issues . although the child is doing fine at present , there is still a long road ahead for a reasonable functional recovery , and the secondary reconstructive procedures may be required . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed .

replantations for major amputations of upper extremity have been widely performed . we report a unique case of successful replantation of scapulothoracic avulsion amputation in a child . in this manuscript , we discuss the various challenges faced during the procedure and chances of neural recovery .

internal hernias are an uncommon cause of bowel obstruction , accounting for less than 1% of cases . it can be asymptomatic , causes chronic abdominal pain or presents with acute intestinal obstruction . the diagnosis of this congenital malformation is often made during surgical intraoperative exploration for intestinal obstruction . it 's about a 77 year - old woman consulting for an acute bowel obstruction evolving since two days . physical examination showed a discrete abdominal distension and slight tenderness in the epigastric and the left upper quadrant . the computed tomography ( ct ) showed the presence of a jejunal loop contained in a retroperitoneal sac interposed between the stomach pushed forward , the tail of the pancreas to the right and back and the left colon to the left and back [ figure 1 ] . the mesenteric vessels of these loops were stretched with displacement of the inferior mesenteric artery to the left . the diagnosis of a left ph complicated with acute bowel obstruction has been well discussed . surgical exploration found about 40 cm of proximal healthy small bowel in an internal left paradoudenal hernia . abdominal ct showing the presence of dilated loops in the back cavity of the omentum interposed between the pancreas ( 1 ) stomach ( 2 ) and the descending colon ( 3 ) an internal hernia is a protrusion of bowel through abnormal orifice in the peritoneum or mesentery , it account for only 0.25 - 0.9% of all patients with small intestinal obstructions . based on the former definition , the most commonly seen hernia types are as follows : paraduedonal , transmesenteric , pericecal , intersigmoid , supravesical , foramen winslow , retroanastomotic , and omental hernias . ph is the most common form of congenital internal hernia , making up 53% of all reported cases . the left sided hernias are more common than right sided ones , representing 75% of cases [ figure 2 ] . peroperative view : the neck of the hernia ( arrow ) on the left side of the duodeno - jejunal junction ( adj ) paraduedonal hernias result from abnormal rotation of the midgut during embryonic development [ figure 3 ] . left parduodenal hernia : landzert 's parduodenal fossa ( arrow ) , ( 1 ) the fourth portion of the duodenum , ( 2 ) inferior mesenteric vein , ( 3 ) left colic artery although ph is congenital , most cases are discovered between the 4 and 6 decades of life with mean age about 38.5 years , they are exceptionally manifested in the elderly patients over 75 years old . some of internal hernia may remain asymptomatic during the whole life time . about 10% to 15% of cases are discovered preoperatively . often , the clinical signs are dominated by periombilical cramps or postprandial epigastric pain , nausea , vomiting and so rarely the presence of an abdominal mass in the left side of the abdomen . regarding the risk of incarceration of ph witch mortality as higher as 20 - 50% , it is recommended that all cases of ph , including asymptomatic ones , should be surgically corrected . the classic radiological findings of left ph were originally described from the small bowel follow - through examination . abdominal ct has now become the study of choice to provide the correct diagnosis during symptomatic periods . left ph have a characteristic appearance of a formation include clustering of small bowel loops , a saclike mass with encapsulation at the ligament of treitz , duodenojejunal junction depression , mass effect on the posterior stomach wall , engorgement and crowding of the mesentery vessels with frequent right displacement of the main mesenteric trunk , and depression of the transverse colon . anterior and upward displacement of the inferior mesenteric vein that lies in the ventral circumference of the hernia orifice has been postulated as an additional diagnostic feature on ct . the preoperative diagnosis of these internal hernias could be often unknown even after ct well guided , its discovery sometimes occurs during surgery . if it 's difficult to reduce because of its bulky size or adhesions within the sac , an incision in the avascular portion of the hernia sac to the right side of the inferior mesenteric vessels , can be made so to enlarge the neck hernia . during this procedure , it 's important to consider the relationship with the inferior mesenteric vessels to avoid injury of these structures . the defect is dealt with in two ways , either by simple closure or by wide opening of the sac by making it continuous with the peritoneal cavity . , uematsu reported the first laparoscopic repair of ph , the laparoscopic approach has been reported as a way of diagnosis and repair of ph in many literature reports . as with open repair , laparoscopic repair of ph can be performed either by wide extension or by surgical closure of the hernia defect . although laparoscopic repair would be expected to reduce postoperative pain and the hospital stay , the paucity of data precludes any final conclusions regarding these issues as well as rates of recurrence . the current data suggest that acute small bowel obstruction can be managed by laparoscopy in selected cases . although relatively uncommon , left ph should be included in the differential diagnosis of small bowel obstruction despite any history of previous abdominal surgery . the combination of a high index of suspicion , familiarity with this disease entity , and modern imaging technology make preoperative diagnosis easier today .

internal hernias of the abdomen are uncommon . they represent less than 1% of bowel obstruction cases . the left paraduodenal hernia ( ph ) is the most frequent type of internal hernias . we report a case of 77 year- old woman consulting for bowel obstruction evolving since two days . the abdominal computed tomography revealed a retroperitoneal small bowel contained in a peritoneal sac . the surgical exploration confirmed the diagnosis of a left internal ph by showing incarcerated jejunal loops in a ph through a narrow opening to the left of the angle of treitz . a surgical reduction of the hernia and closure of the hernia neck were performed . the follow - ups were uncomplicated . through this observation and a literature review , we try to recall the clinical and radiological characteristics of this disease and to clarify the therapeutic modalities .

autosomal dominant polycystic kidney disease ( adpkd ) is characterized by progressive cyst formation in the kidneys leading to massive kidney enlargement . over time renal cysts may rupture leading to hematuria and pain . a pyogenic infection may also be located in these cysts . adpkd patients with chronic pain related to the enlarged kidneys , recurrent hematuria or therapy - resistant cyst infection may benefit from nephrectomy . in addition , in some patients , the cystic kidneys are exceptionally enlarged leaving too little abdominal space to allow for kidney transplantation . here , we report nephrectomy of an extremely enlarged adpkd kidney prior to transplantation in a 42-year - old man . a 42-year - old male patient with adpkd visited our outpatient clinic while preparing for renal replacement therapy . his medical history included hypertension and myocardial infarction , complicated with aorta dissection , classified as a stanford type a. he had experienced multiple periods with hematuria and kidney pain , most likely caused by recurrent cyst bleedings . he inherited adpkd from his paternal side ; his father started renal replacement therapy at 42 years of age and died at 46 years due to complications during a second kidney transplantation . his 35-year - old sister also has adpkd with a 24-hour urine creatinine clearance of 137 ml / min and a total kidney volume of 1,974 ml . physical examination of the patient revealed a man weighing 99 kg ( 218 lb ) and measuring 189 cm ( 6 feet 2.4 inch ) , with a body mass index of 27.7 . his residual 24-hour urine creatinine clearance had decreased to 8 ml / min , and his serum creatinine was 812 mol ( 9.2 mg / dl ) . since a potential living unrelated donor allograft magnetic resonance imaging showed exceptionally enlarged kidneys in a small abdominal space ( total kidney volume is 10,280 ml , left kidney 5,628 ml and right kidney 4,652 ml ) ( fig . the total kidney volume was measured on t2-weighted coronal images using the commercially available software analyze direct 8.0 ( analyze direct , inc . , overland park , kans . postoperatively , no complications were reported and he was discharged from the hospital in a good condition 7 days later . the removed kidney weighed 8.7 kg ( 19.3 lb ) and had a maximal length of 48 cm ( 19 inch ) ( fig . material , see www.karger.com/doi/10.1159/000363378 ) revealed almost complete replacement of cortical as well as medullary kidney tissue by numerous cysts . some of these cysts were filled with clear pre - urine , others with hemorrhagic fluid and again others with a chocolate - like substance . after the nephrectomy , the patient was on temporary hemodialysis until he received a living unrelated renal allograft 3 months later . one week after the transplantation , he experienced an episode of vascular allograft rejection type iia , which was successfully treated with antithymocyte globulin and plasmapheresis . five months after the transplantation , he was doing well with mildly reduced but stable kidney function [ creatinine level 124 most affected subjects have progressive renal function decline and need renal replacement therapy between their 40th and 70th year of age . the weight of the removed kidney was 8.7 kg ( 19.3 lb ) and its volume was 5,925 ml . this indicates that the volumetric mass density ( mass per unit volume ) of the removed kidney was much higher than water . to our knowledge , only one case reported a patient with adpkd that had kidneys with higher total weight than the present case . transection of the kidney revealed the presence of clear and cloudy fluid , to chocolate - brown semisolid contents in the cysts , consistent with the variable aspects of the cysts in the magnetic resonance image ( fig . the dark contents of some cysts probably reflect passed bleeding events that may have been associated with a sudden increase of tension on the kidney capsule , a cause for pain and discomfort reported by the patient , although symptomatic episodes probably underestimate the true frequency of cyst hemorrhage . in the removed kidney , most renal parenchyma in both the cortex and , in general , both kidneys are usually affected to a similar degree by adpkd , it seems remarkable that the patient 's preoperative creatinine clearance was still 8 ml / min .

autosomal dominant polycystic kidney disease ( adpkd ) is the most common hereditary kidney disease . it is characterized by progressive cyst formation in both kidneys , often leading to end - stage kidney disease . indications for surgical removal of an adpkd kidney include intractable pain , hematuria , infection , or exceptional enlargement and small abdominal cavity hampering implantation of a donor kidney . we report the case of an extraordinarily large adpkd kidney weighing 8.7 kg ( 19.3 lb ) with a maximal length of 48 cm ( 19 inch ) , and with cysts filled with both clear and bloody fluid .

some cases of ch have been reported among patients with turner syndrome ( 1 , 2 ) . in these cases , ch resulted from congenital vascular abnormality and hypertension . there is a relatively high prevalence of hypertension in turner syndrome patients , compared with age - matched control groups ( 3,4,5 ) , and most of these are renovascular hypertension cases ( 6 , 7 ) . based on these findings , it is suspected that turner syndrome patients possess some risk factors for ch . in this paper , we report a case of turner syndrome associated with ch and discuss its causes . a 21-yr - old female with turner syndrome ( 45,x ) was transferred to our hospital due to loss of consciousness . her consciousness level was classified as comatose ( japan coma scale , ii-30 ) . left hemiplegia and hypertension ( 212/96 mm hg ) were observed . a computed tomography scan revealed ch in the right putaminal region ( fig . cerebral angiography revealed shifting of the right median cerebral artery due to the hematoma ; however , no vascular malformation or aneurysm was found . urinalysis , peripheral blood counts , and coagulation function tests were within the normal limits , ( data not shown ) . emergency surgery was performed in order to excise the hematoma , and the patient regained consciousness 12 h after the surgery . the left hemiplegia and hypertension persisted ; therefore , a calcium antagonist was administered . magnetic resonance imaging angiography revealed absence of vascular anomaly or narrowing in the aortic arch , renal arteries , or other branches of the aorta . the optic fundi showed a slight hypertensive change , indicating that the patient was suffering from hypertension prior to this episode . forty days after the surgery , her plasma renin activity ( pra ) and plasma aldosterone concentration ( pac ) were high , i.e. , greater than 20.0 ng / ml / h and 331 pg / ml , respectively . thereafter , her basal pra persisted at a high level from 4.0 to 10 ng / ml / h at rest in the supine position . eight months after ch , a renin stimulation test with furosemide ( 20 mg i.v . ) was performed with 2 h upright . her basal renin level was 4.60 ng / ml / h , and after the stimulation , it was 34.6 ng / ml / h . we administered an angiotensin ii receptor blocker and a calcium antagonist , and her hypertension was well controlled . after one and a half years , her left hemiplegia was completely resolved , but the left side paresthesia persisted . she was short in stature in infancy and was referred to our hospital at the age of 7 yr for evaluation of her stature ( 106.0 cm : 3 sd ) . a chromosomal study revealed the karyotype 45,x , and she was diagnosed as turner syndrome . recombinant human gh ( rhgh ) therapy was initiated from the age of 11 yr and was completed at the age of 15 yr . following the rhgh therapy , female sex steroid replacement therapy was started . hypertension ( 154/88 mm hg ) was incidentally detected at the age of 16 yr when the patient was hospitalized for a nasal conchotomy to correct a nasal obstruction . some cases of ch have been reported among patients with turner syndrome ( 1 , 2 ) . in these cases , ch resulted from cerebral vascular abnormalities or hypertension . turner syndrome is occasionally accompanied by aortic dissection , aortic coarctation , aneurysms , arteriovenous malformation , and teleangiectasias . it is suspected that these vascular abnormalities occur due to vascular connective tissue defects ( 8 , 9 ) caused by the chromosomal abnormalities in turner syndrome . there is a high incidence of hypertension in turner syndrome cases in comparison with age - matched female groups ( 3,4,5 ) . however , sex hormone replacement therapy is not recognized as elevating the blood pressure ( 10 ) . with regard to hypertension , pra is higher in turner syndrome cases than in the control group , and it is more elevated in hypertensive turner syndrome patients than in normotensive turner syndrome patients ( 11 ) . this high pra may be due to the renal vascular abnormality that accompanies turner syndrome . we should recognize the fact that turner syndrome cases are susceptible to renovascular hypertension . we could not ascertain the cause of pra elevation , and her hypertension must have resulted from hyperreninemic hypertension . a possible mechanism of ch in our turner syndrome patient may be the occurrence of some vascular abnormality in the renal artery and cerebral artery caused by a chromosomal abnormality . both renovascular hypertension and cerebral vascular abnormality may play roles in the occurrence of the cerebral vascular incidents . this case emphasizes the necessity to carefully monitor the blood pressure in turner syndrome cases , even during childhood , in order to prevent the risk of ch .

we report the case of a 21-yr - old female with turner syndrome associated with cerebral hemorrhage ( ch ) . she was transferred to our hospital for loss of consciousness and was diagnosed with right putaminal hemorrhage . following surgical removal of the hematoma , she regained consciousness , and her left hemiplegia gradually improved after surgery . angiography revealed absence of vascular abnormality of the cerebral artery , aorta , and renal arteries . hypertension was noted on arrival at the hospital and persisted after surgery . a slight hypertensive change was observed in her retinas . plasma renin activity was elevated ( 20 ng / ml / h ) and renovascular hypertension was suspected . in this patient , ch was suspected to have occurred due to hypertension . this case emphasizes the necessity to carefully monitor the blood pressure in turner syndrome cases , even during childhood .

cleaning tasks such as washing cookware entail high workloads involving repetitive hand and wrist movements1 . typically , many dishes are washed in a large , deep sink . during a busy work shift , dishwashers bend the trunk at an angle of 1045 from an upright position and maintain this fixed posture for long periods without a break2 . studies have evaluated the effects of load during dishwashing on the low back and legs3 . although most studies have focused on low back pain in dishwashers , dishwashing also puts excessive load and tension on the forearm and shoulder muscles3 , 4 . a prolonged poor posture with a forward head position and protracted shoulders could contribute to neck and shoulder pain5 . the use of rubber gloves is a common practice in many industries , including meat packing , construction , and warehousing . wearing gloves can alter the efficiency of tasks that require manual dexterity , although few studies have investigated this . therefore , this study examined the effects of wearing rubber gloves on the activities of the forearm and shoulder muscles during two dishwashing stages . ten female students ( mean age , 33.1 2.2 years ; height , 158.9 3.0 cm ; weight , 57.2 5.7 kg ) participated in this study . all of the participants were right - hand dominant and typically spent approximately 1 hour per day washing dishes at home . the purpose and methods of the study were explained to the participants before their inclusion in the study , and they provided informed consent according to the principles of the declaration of helsinki . the activities of the left wrist flexor and upper trapezius muscles were measured using a wireless electromyography ( emg ) system ( delsys , boston , ma , usa ) . the emg data are expressed as a percentage of the maximum voluntary isometric contraction . the participants performed two dishwashing stages ( washing and rinsing ) with and without rubber gloves , washing five 20-cm - diameter dishes at their usual speed . the height of the dishwashing table was adjusted such that the top was level with the anterior superior iliac spines of the participants . 18.0 ( spss , chicago il , usa ) with the level of statistical significance set at p < 0.05 . a paired t - test was used to assess differences in the activities of the wrist flxor and upper trapezius muscles between conditions with and those without rubber gloves during the two dishwashing stages . during the washing stage , the activities of the wrist flexor and upper trapezius muscles were significantly ( p < 0.05 ) greater without gloves than with gloves ( 51.4 22.0% vs. 40.5 10.9% and 32.1 13.8% vs. 26.6 9.0% , respectively ) . during the rinsing stage , the activities of the wrist flexor and upper trapezius muscles did not differ significantly ( p > 0.05 ) between conditions with and those without gloves ( 37.4 19.1% vs. 35.5 17.9% and 20.1 13.8% vs. 22.6 12.0% , respectively ) . research can help industries select the types of rubber gloves that enable the most efficient performance while providing suitable protection for a specific job . this study showed that during the dish washing stage , the activities of the wrist flexor and upper trapezius muscles were significantly higher without gloves than with gloves . in the present study , the activities of the wrist flexor and upper trapezius muscles during the rinsing stage did not differ significantly according to the use of glove . recent reports have suggested that the use of rubber gloves increases the incidence of cumulative traumatic disorders and interferes with the wearer s ability to perform fine motor activities and object manipulations5 , 6 . information gained from studies on gloves can help set guidelines for designing workplaces and equipment . a decrease in forearm muscle activation was shown to lead to a compensatory increase in shoulder movements5 , 6 . shin and yoo found that manual material handling or light manual precision work depended on exerting effective grip force7 . the grip force exerted was reduced by impaired recruitment of the extensor digitorum and flexor digitorum superficialis , resulting in an increased incidence of wrist or shoulder pain8 . known risk factors for upper - extremity muscle injury include repetitive and continuous detailed work9 . typically , most dishwashers wear poorly fitting gloves that allow their hands to move inside the gloves , impairing fine - motor performance10 . wearing the wrong gloves can affect manual dexterity , which is key to performing many tasks efficiently10 . however , working bare handed or wearing well - fitting rubber gloves was shown to result in effective forearm muscle activation , which decreased inefficient shoulder movements8 . wearing well fitting rubber gloves can improve safety and comfort in the workplace , helping manual workers to perform their tasks both rapidly and efficiently8 . in conclusion , dishwashers should wear gloves during the washing stage to prevent wrist and shoulder pain .

[ purpose ] the present study examined the effects of wearing rubber gloves on the activities of the forearm and shoulder muscles during two dishwashing stages . [ subjects ] this study included 10 young females . [ methods ] the participants performed two dishwashing stages ( washing and rinsing ) with and without rubber gloves . the activities of the wrist flexor and upper trapezius muscles were measured using wireless electromyography . [ results ] during the washing stage , the activities of the wrist flexor and upper trapezius muscles were significantly greater without gloves than with gloves when performing the same tasks . however , during the rinsing stage , the activities of these muscles did not differ significantly according to the use of gloves . [ conclusion ] dishwashers should wear gloves during the washing stage to prevent wrist and shoulder pain .

in this issue , lopes and colleagues add to the list of studies investigating the concept of goal directed haemodynamic therapy ( gdht ) . a variety of strategies and monitoring modalities have been applied and in general have resulted in improved patient outcomes . we have worked through pulmonary artery catheters , doppler probes , and less invasive methods of cardiac output measurement , but the recent paper is the first to use a truly minimally invasive technique to assess the requirement for further fluid infusions above normal perioperative care . in their study of goal directed fluid management based on pulse pressure variation monitoring during high risk surgery , they demonstrate a spectacular improvement in outcome using their monitoring and fluid management strategy . pulse pressure variation in mechanically ventilated patients has been shown to be a good predictor of fluid responsiveness and by targeting this parameter lopes and colleagues increased the mean volume of intra - operative fluid infused from 1,694 ml in the control arm to 4,618 ml in the treatment arm . despite comparable pre - operative demographics , improvements were seen in post - operative complication rates , duration of mechanical ventilation and length of hospital and intensive care unit ( icu ) stay . it is the dramatic outcome improvement that will be the talking point in this study and questions will be raised about the nature of treatment given to the control group were they undertreated , what protocols were used for them and is this baseline mortality comparable to experience in my institution ? on this last point it is noteworthy that other studies from south america have shown similar control outcomes . despite the quantity of evidence in support of the principle of gdht there are a number of reasons for this including a lack of familiarity with preventative medicine in the perioperative setting , confusing terminology , problems with identifying patients who might benefit , doubts about the evidence , little peer pressure to undertake such protocols , a confusion with the debate on efficacy of pulmonary artery catheterisation and the use of gdht in the situation of sepsis , and implementation issues such as requirement for investment , identifying suitable clinical areas and personnel . on these last points the current study may be very influential as the advantage of the approach used by lopes and colleagues is that the technique is simple and requires very little extra investment . however , another reason for the slow uptake of this concept is that the evidence for gdht loses some of its strength when closely examined . the meta - analysis by poeze and colleagues demonstrated that small , ' poor quality ' studies generally produce much larger treatment effects than bigger , higher quality studies . in this meta - analysis there was only one trial with a smaller sample size than the trial by lopes and colleagues , and when only higher quality trials were included in the analysis there was no statistically significant improvement in outcome from gdht . one reason that the study was so small is that it was stopped early because marked clinical benefit was observed . while one can sympathize with the trialists ' desire to move as soon as possible to treatment that they observe improving patient outcome , the practice of stopping trials early due to benefit has been seriously questioned . in the analysis by montori and colleagues it is unusual in medical care to have proposed a relatively simple treatment that has received considerable positive support from randomised clinical trials over a number of years , in different clinical settings ; and in economic analyses has proved to be cost effective ; which has not been adopted . parallels can be seen in the failure of widespread adoption of selective decontamination of the digestive tract . it seems unlikely that further small trials will result in the breakthrough to widespread implementation that the evidence seems to warrant and it seems quite clear that what is required is a large , multicentre , randomised trial of a gdht in high risk surgical patients . if the strategy suggested by shoemaker and investigated now by lopes and colleagues and resulting in 20 or so original trials in the intervening period continues to deliver the observed reductions in complications and length of stay in a larger trial setting then it may truly revolutionise perioperative care for all patients .

the literature concerning the use of goal directed haemodynamic therapy ( gdht ) in high risk surgical patients has been importantly increased by the study of lopes and colleagues . using a minimally invasive assessment of fluid status and pulse pressure variation monitoring during mechanical ventilation , improvements were seen in post - operative complications , duration of mechanical ventilation , and length of hospital and intensive care unit ( icu ) stay . many small studies have shown improved outcome using various gdht techniques but widespread implementation has not occurred . those caring for perioperative patients need to accept the published evidence base or undertake a larger , multi - centre study .

a 56-year - old indian male , hypertensive , with uncontrolled diabetes mellitus presented with blurring of vision in both eyes for 1 week . on examination , his best - corrected visual acuity ( bcva ) was 20/60 , 20/200 for near in the right eye and 20/30 , 20/20 for near in the left eye . anterior segment was normal in both eyes but left eye had 1 + vitreous cells . he was investigated to rule out tuberculosis as an etiology and mantoux test , quantiferon tb gold test and chest x - ray were found to be normal in this patient . fundus revealed healed scars of sc involving macula in the right eye and active lesions involving the peripapillary area and encroaching the fovea with hyperemic disc in the left eye [ fig . on fluorescein angiography ( fa ) , active lesions were hypofluorescent in early phase [ fig . 1b ] with late hyper - fluorescence with disc staining in late phase [ fig . the patient was not given clearance for intravenous methylprednisolone or oral steroids as his fasting , and postprandial blood sugar were 233 and 338 mg / dl , respectively . the patient was given an injection of 2 mg of triamcinolone acetonide in the left eye under aseptic precaution and was put on oral azathioprine . ( a ) the fundus photograph of the left eye with active lesions of serpiginous choroiditis ( white arrowhead ) threatening the fovea with disc hyperemia ( white curved arrow ) . ( b and c ) the fluorescein angiographic picture in early and late phase respectively with early hypofluorescence ( white thick arrow ) and late hyperfluorescence ( white thin arrow ) and disc staining ( white arrowhead ) three days postinjection , patient was reviewed , and the lesions were found to be regressing [ fig . 2 ] . bcva in the left eye improved to 20/20 , 20/20 for near at that time and right eye was stable . fundus examination showed complete healing of the lesions in the left eye and was confirmed on fa . ( a ) the fundus photograph of the left eye showing the resolving lesions ( white long arrow ) with persistent disc hyperemia ( white arrowhead ) , ( b ) the montage picture of the left eye with triamcinolone acetonide deposit in the vitreous ( white short arrow ) ( a ) the fundus photograph of the left eye showing healed lesions of serpiginous choroiditis leading to retinal pigment epithelium atrophy ( white short arrow ) and normal optic disc , ( b ) the montage picture of the left eye with triamcinolone acetonide deposits in vitreous ( c ) hyperfluorescence due to window defect in late phase sc is a chronic inflammatory chorioretinitis which starts from the peripapillary area and spreads to macula and peripheral retina in a geographic fashion . vision loss occurs due to the involvement of the macula or due to choroidal neovascularization developing later in the course of the disease . treatment in active stage is immunosuppression using monotherapy with steroids or cyclosporine to triple therapy . however , it can show the effect on prolonged use due to long duration required for onset of action . in this case , the regression of the lesions were noted as early as 3 days which can be due to ivta . use of 2 mg ivta has been found to be associated with lesser duration of the requirement of anti - glaucoma medications than 4 mg ivta . use of 2 mg ivta can be a safer alternative in terms of iop spike . in our patient , 2 mg of triamcinolone acetonide was injected intravitreally . however , a large - scale study is required to prove the efficacy of using reduced dose ivta in such situations . few reports are available showing the benefit of 4 mg of ivta in treating active sc . however , to our knowledge , this is the first report showing the efficacy of a reduced dose ( 2 mg ) of ivta in treating active sc .

active serpiginous choroiditis ( sc ) is a vision - threatening condition which requires intensive treatment using corticosteroids and/or immunosuppressives , especially if the lesions are involving or encroaching on the macula . use of oral and intravenous high - dose steroids are contraindicated in uncontrolled diabetics . intravitreal steroid delivers a localized dose in such situations . this case report highlights the efficacy of reduced dose of intravitreal triamcinolone acetonide ( 2 mg ) in the treatment of active sc .

one hundred and twelve eyes of 112 patients with cataract and oht ( 60 males , 52 females ) who were operated between january 2010 and november 2011 were evaluated retrospectively . their mean age was 61.32 11.12 ( standard deviation [ sd ] ) ( 4588 ) years . full ophthalmological examinations including uncorrected visual acuity , best corrected visual acuity ( bcva ) , iop measurements , acd , ica , and cct measurements , slit - lamp biomicroscopy , and fundus examination , were performed preoperatively and postoperatively . iop measurements were made with goldmann applanation tonometer ( haag - streit , switzerland ) , acd with iol master optical biometer ( zeiss , germany ) , ica with goldmann 3-mirror lens ( haag - streit , switzerland ) and cct was measured with ultrasonic pachymeter ( meda , usa ) . the iop levels of the patients were higher than normal , but they had no visual field defects and neither used any antiglaucomatous medications , that 's why classified as oht . the cases with primary open angle glaucoma , acute or chronic primary angle closure glaucoma ( acg ) , pseudoexfoliation ( pex ) syndrome , pigmentary dispersion syndrome , history of ocular surgery , and intraoperative and/or postoperative complications were excluded . preoperative mean bcva of the patients was 0.54 0.23 ( sd ) ( 0.31.00 ) , postoperatively it was 0.03 0.04 ( sd ) ( 0.000.10 ) according to the logarithm of the minimum angle of resolution scores . preoperative mean iop value was 24.67 2.14 ( sd ) ( 2129 ) mmhg , the decrease in iop values of the 1 week , 1 month , 3 month , 6 month , and 1 year was statistically significant but that of the 2 year was not significant . preoperative mean acd value was 2.98 0.33 ( sd ) ( 2.43.6 ) mm . the increase in acd values of the 1 week , 1 month , 3 month , 6 month , and 1 year was statistically significant , but that of the 2 year was not significant . preoperative mean ica value was grade 2.85 0.75 ( 24 ) according to shaffer classification . the increase in ica values of the 1 week , 1 month , 3 month , 6 month , and 1 year was statistically significant , but that of the 2 year was not significant . preoperative mean cct value was 550.89 20.07 ( sd ) ( 520595 ) mm . the increase in cct values of 1 week and 1 month was statistically significant , but those of 3 month , 6 month , 1 year and 2 year were not significant . postoperatively , visual field tests were performed every 6 months from a 1 postoperative month on . preoperative and postoperative bcva , iop , acd , ica , and cct values are summarized . in fig . pre- and post - operative bcva , iop , acd , ica and cct values postoperative intraocular pressure changes postoperative anterior chamber depth changes postoperative ridocorneal angle changes postoperative central corneal thickness changes it is known that the cataract extraction causes iop reduction , but the mechanism is not known completely . after cataract surgery , iop reduction is more prominent in patients with acute or chronic acg . the more shallow anterior chamber is preoperative , the more deepening will occur postoperatively , and ica widens more and as a result of these , mean iop reduction occurs more . showed that in patients with primary acg , cataract surgery resulted in the dissolution of lens volume and pupillary block and also attenuation of the anterior positioning of the ciliary process , which contributed to the postoperative widening of the angle . huang et al . reported that postoperative reduction in iop was proportional to the increase in angle after cataract surgery and postoperative iop reduction was greater in eyes with a narrower angle . besides , after cataract surgery , iop reduction is prominent in eyes with pseudoexfoliative glaucoma . this is due to increase blood - aqueous barrier permeability which is impaired in pexsyndrome and intraoperative aspiration of the pseudoexfoliative material on anterior lens capsule and trabecular meshwork leading to increased outflow . the most important parameter affecting postoperative iop is the preoperative iop level , meaning the higher preoperative iop is , the more postoperative reduction is , the lower preoperative iop is , the less postoperative reduction is . reported that cataract surgery decreased iop in patients with oht over a long period of time , at least 36 months . chang et al . reported that , in ocular hypertensive and glaucoma patients , uncomplicated phacoemulsification had no significant iop - lowering effect compared with the phakic fellow eye for up to 3 years postoperatively and also there was no difference between the mean number of postoperative iop - lowering medications used in the surgical and fellow eyes . in our study , we observed that , in patients with cataract and oht , after phacoemulsification , reduction in iop and increase in both acd and ica were statistically significant at postoperative 1 week , 1 month , 3 month , 6 month , and 1 year , but these changes were not significant at the end of 2 year . phacoemulsification surgery decreases iop and increases acd and ica in the short - term , but in the long - term , it does not cause any significant changes .

we evaluated the effect of phacoemulsification surgery on intraocular pressure ( iop ) , anterior chamber depth ( acd ) , iridocorneal angle ( ica ) , and central corneal thickness ( cct ) of the patients with cataract and ocular hypertension . the decrease in iop values of the 1st week , 1st month , 3rd month , 6th month , and 1st year was statistically significant , but that of the 2nd year was not significant . the increase in acd and ica values of the 1st week , 1st month , 3rd month , 6th month , and 1st year was statistically significant , but that of the 2nd year was not significant . the increase in cct values of 1st week and 1st month was statistically significant , but those of 3rd month , 6th month , 1st year , and 2nd year were not significant . in conclusion , phacoemulsification surgery decreases iop and increases acd and ica in the short - term . however , in the long - term it does not cause any significant changes .

intramedullary tuberculoma ( imt)are extremely rare lesions should be distinguished from other space occupying lesions like neoplasms . although it is a treatable condition delaying diagnosis leads to significant morbidity . we report a case of intramedullary thoracic tuberculoma with paraparesis showing deterioration of neurological status during medical treatment . the possibility of imt should be seriously considered when an intraspinal mass is found with evidence of tuberculosis . anti - tuberculous therapy ( att ) is the mainstay of treatment and had shown good results . surgical removal is to be considered in cases of worsening neurological status while the patient is on att . intramedullary spinal tuberculosis ( tb ) is a rare disease entity , which was first described by abercrombie in 1828 . it can be seen at any level of the cord but usually involves dorsal spinal cord . a 25-year - old male presented with low back pain , progressive weakness in lower limbs since 1-month and bladder incontinence since 2 weeks . neurological examination revealed grade 3 power in both lower limbs and hypoesthesia below d8 level . plain radiograph of the spine showed paradiscal lesion l2 - 3 level , while rest of the spine was normal . magnetic resonance imaging ( mri ) of the spine demonstrated paradiscal lesion with paravertebral abscess at l2 - 3 . at the level of d8 vertebra , there was an intramedullary lesion of 12 mm 9 mm which was hypointense on t1 , isointense lesion in t2 , and demonstrated ring enhancement on gadolinium contrast ( fig . 1 ) . based on this clinico - radiological picture , a diagnosis of l2-l3 tb with intramedullary tuberculoma ( imt ) at d8 level was made and patient was started on multidrug treatment ( isoniazid , rifampicin , ethambutol , and pyrazinamide ) and a short course of dexamethasone . at 6 weeks , patient was deteriorated neurologically with grade 2 power in both lower limbs . at 12 weeks patient did not show any signs of improvement . at this point , we found a grayish mass with a good plane of cleavage from the surrounding cord ( fig . a grey , irregular , multilobulated soft mass measuring 12 mm 10 mm 6 mm was removed completely ( fig . histopathology examination showed diffuse & dense infiltrate compromising of lymphocytes , plasma cells , and neutrophils with large areas of caseous necrosis surrounded by sheets of the epithelioid cell and langerhans type of giant cells . z - n stain showed no acid - fast bacillus , while tissue culture was negative . patient slowly showed signs of neurological recovery after 6 weeks however his bladder disturbance persisted . at 3 months after surgery , his lower limb power in hip and knee was completely recovered , with recovered bowel and bladder retention . post - operative after 6 months patient was walking without support and had functional power in both lower limbs . intraoperative picture showing grayish mass with a good plane of cleavage from the surrounding cord . intracranial tuberculomas are still a common cause of space occupying lesion ( sol ) in india . imt are seen in only 2 out of 100,000 cases of tb and 2 out of 1000 cases of central nervous system tb . imt most commonly is hematogenous , secondary to the tb elsewhere in the body , commonly pulmonary tb , but some cases may present only with isolated extrapulmonary forms . imts tend to occur predominantly in young people , most commonly found in thoracic region . the commonest symptom is subacute spinal cord compression with the appropriate motor and sensory findings , depending on the level of the lesion . mri is gold standard diagnosis modality , as it accurately shows the site , size , number in such lesions . , there is an abundance of giant cells and poor collagenous capsule with variable surrounding edema . at this stage , tuberculoma is isointense on both t1w1 and t2w1 and shows homogenous enhancement with gadolinium contrast . later on surrounding capsule become richer in collagen and surrounding inflammatory reaction starts disappearing . this results in an isointense lesion on t1w1 and isointense or hypointense lesion on t2w1 images and ring enhancement with hypointense center with gadolinium contrast . with the development of caseation center on treatment with chemotherapy the lesion regresses in size and disappears , to be replaced by an area of gliosis , which probably is seen as an area of hypointensity on mri [ 8 - 10 ] . the most important finding is worsening of the patient s neurologic condition while being treated with adequate anti - tuberculous drugs . an immune reaction has been suggested . in tuberculoma , anti - tuberculous chemotherapy destroys the bacilli and releases tuberculous protein , which mediates lymphocyte proliferation , resulting in expansion of the tuberculoma . surgical excision is recommended in cases of severe cord compression , increasing neurological deficit in the presence of akt . this allows surgical removal without excess trauma to surrounding functional spinal cord tissue . the mycobacterium tb isolated from the tuberculoma will help in deciding the akt in the present scenario of drug resistance to akt akt with four drugs hrze having good cerebrospinal fluid penetration recommended for 12 months post - operative . we suggest a combination of surgical excision of tuberculoma along with akt for imt not responding to akt for a good outcome . tuberculoma should be considered in the differential diagnosis of all intramedullary sol s irrespective of age or presence of extracranial focus of tb in countries endemic to tb . most of these patients respond well to anti - tuberculous drug therapy with good functional recovery . however , timely surgical decompression in selected cases , provide an excellent long - term outcome . this case report highlights role of surgical treatment in management of intramedullary tuberculoma . surgical excision should be performed i patient not responding or deteriorating with medical management .

introduction : intramedullary tuberculoma ( imt)are extremely rare lesions should be distinguished from other space occupying lesions like neoplasms . although it is a treatable condition delaying diagnosis leads to significant morbidity.case report : we report a case of intramedullary thoracic tuberculoma with paraparesis showing deterioration of neurological status during medical treatment . surgical resection had shown the excellent result.conclusion:the possibility of imt should be seriously considered when an intraspinal mass is found with evidence of tuberculosis . anti - tuberculous therapy ( att ) is the mainstay of treatment and had shown good results . surgical removal is to be considered in cases of worsening neurological status while the patient is on att .

1 . he had a history of bilateral brow suspension surgery 10 years ago at another institution . the patient had a 30 cm 20 cm pelvic mass located in the presacral area with boundaries between both parailiac regions displacing the rectum and bladder . it was completely excised with a pathology report of myelolipoma . in physical examination , he had hypertrichosis , finger clubbing , and radiologically cortical thickening of the bones fig . the patient presented with bilateral blepharoptosis with coarse skin folds the patient had cortical thickening of the bones and clubbing his best - corrected visual acuity was 7/10 in the right eye ( re ) and 5/10 in the left eye ( le ) . horizontal length of the upper lids of re and le was 46 mm and 41 mm , respectively . vertical fissure heights were 6 mm and 4 mm with margin - reflex distances of 0.5 mm and 0.5 mm , respectively . vertical eyelid contour of the re showed an inverse v shape for the re and a smooth curve for the le . the eyelids were floppy and easily everted . to correct these clinical findings , a surgery which combines vertical tarsal shortening accompanied with horizontal whole eyelid wedge resection and brown suspension 10 mg/0.25 ml triamcinolone acetate was injected into each supratarsal space to decrease the thickness of the tarsus . horizontal wedge resection which is not a part of routine ptosis surgery was performed in this operation with the aim of normalizing the obvious horizontal length of the lids . pathological evaluation revealed epidermal hyperplasia , severe inflammatory changes in subepidermal level , hyperplastic sebaceous glands , and collagen tissue derangement fig . reconstruction of each eyelid was achieved by vertical tarsectomy , horizontal full thickness lid resection , and supratarsal steroid injection . ptosis was assessed with silicone rod frontalis suspension , and resection of excessive skin was performed as in blepharoplasty skin biopsy showing thickening of the dermis with increased collagen content and lymphocytic infiltration ( h and e , 10 ) the postoperative course was uneventful and patient satisfaction is good within the 1 year of follow - up fig . it is characterized by skin thickening , clubbing , hyperhidrosis , and periosteal reaction in the long bones . primary hoa is predominantly a male disease that at least 90% of patients are men . although symptoms may be seen in childhood , it manifests mostly during the fifth decade of life . described the largest number of primary hoa series in the literature that the authors reported that family history was positive in all studied patients . the pathogenesis of hoa is unclear and some studies explained the role of several growth factors in the evolution of the disease . although clinical findings are similar , secondary hoa differs from primary hoa with absence of family history . secondary hoa is an acquired form that is associated with usually lung disease but also heart , liver , and intestines . the presented patient had a huge pelvic mass which was completely excised with a pathology report of myelolipoma . hence , our diagnosis was secondary hoa due to the paraneoplastic manifestations of pelvic malignancy which causes secretion of many growth factors . blepharoptosis may develop secondary to sebaceous gland hyperplasia , thickening of the dermis with increased collagen content , mucin deposition , and lymphocytic infiltration or due to additionally marked scarring . our patient was a 52-years - old man with negative family history which let us exclude the diagnosis of primary hoa . the differential diagnosis includes several diseases as acromegaly , syphilitic periostitis , and thyroid acropachy . acromegaly is characterized by enlargement of facial bones that are not present in our case . serology , radiology , and clinical findings did not support a diagnosis of syphilis or thyroid disease . hoa may develop secondary to pulmonary or congenital cyanotic cardiac diseases those were not present in our patient . the remaining possible etiological factor for secondary hoa in our case was the huge pelvic malignancy which was excised previously . the surgery for these kinds of pathologies may be planned as single or staged procedures . blepharoplasty with excessive skin excision is usually required both for better cosmesis and to reduce the tissue bulk . no complications were encountered during surgery , but bleeding was more than any other lid surgery . the profound inflammatory reaction in the tissue might be the cause of this excessive bleeding . hoa may not be always primary , particularly in patients with negative family history . in cases of findings with abnormal fibroproliferative and inflammatory changes , detailed systemic examination and investigations both using serologic and imaging modalities the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed . the authors certify that they have obtained all appropriate patient consent forms . in the form the patient(s ) has / have given his / her / their consent for his / her / their images and other clinical information to be reported in the journal . the patients understand that their names and initials will not be published and due efforts will be made to conceal their identity , but anonymity can not be guaranteed .

a 52-year - old male patient presented to our hospital with a history of secondary hypertrophic osteoarthropathy ( hoa ) associated with an abdominal neoplasia and blepharoptosis . he had finger clubbing , hyperhidrosis , and hypertrichosis . he also had a recent history of extensive abdominal surgery with a pathology report of myelolipoma . routine blood work was unremarkable . upper eyelid reconstruction with blepharoplasty , upper eyelid wedge resection , and brow suspension was performed to address his eyelid concerns . by this case report , we would like to attract notice that the eyelid involvement may be a part of hoa and to emphasize the importance of systemic and pathologic evaluation in failed blepharoptosis surgery .

preliminary sequence analysis of the amplicons obtained by the diagnostic hev rt - pcr from the 2010 blsd outbreaks in hungary ( strains identified by hun prefix ) showed a moderate genetic relationship with reference ahev sequences deposited in genbank ( 5 ) . one strain , hun-167732010 , found in a sample of liver tissue , was selected for further analysis . viral rna was extracted by using the sv96 total rna nucleic acid extraction kit ( promega , madison , wi , usa ) . oligonucleotides were designed for our primer walking sequencing strategy on the basis of ahev genome sequence data ( table ) . genomic fragments were amplified by using a onestep rt - pcr kit ( qiagen , hilden , germany ) as described by the manufacturer . , gdll , hungary ) . * the annealing temperature was set at 53c for each primer combination . + , sense ; , antisense . 5 and 3 end primers were designed on the basis of sequence am943646 . the final assembly was 6,543 bp ( genbank accession no . jn997392 ) , with negligible amounts of missing information at the 5 ( 75 nt ) and 3 ends preceding the poly a tail ( 36 nt ) . nucleotide and deduced amino acid sequences of hun-167732010 aligned well with the other 5 prototype ahev strains . the nearly complete coding sequence ( 1,515 aa ) with a missing 17-aa region was determined for orf1 . predicted functional domains ( 12 ) were identified . the sequence similarities ( megalign ; dnastar inc . , madison , wi , usa ) of orf1 in comparison with genotypes 13 ahev strains were 81.0%83.9% at the nucleotide level and 93.0%95.4% at the amino acid level . unique amino acid changes in the orf1 of hun-167732010 were found in 16 positions compared with the european prototype ahev strain ( not shown ) ( 6 ) . similarities to prototype ahev strains were 83.9%87.3% at the nucleotide level and 98.2%99.3% at the amino acid level . we observed 2 unique amino acid substitutions in positions a53s and q / m473 t , the second residue located within antigenic region ii ( 6 ) . this region also shared high ( > 92% ) nt and ( > 90% ) aa similarities with genotype 13 ahevs . when analyzing the nearly complete genome sequences , we found that strain hun-167732010 shared moderate similarity to genotypes 13 ahev strains ( nt , 81.9%84.9% ) . similar ranges of genome sequence similarity ( nt , 81.8%82.7% ) distinguish prototype ahev strains from each other . using simplot analysis ( http://sray.med.som.jhmi.edu/scroftware/simplot ) , we observed this moderate nucleotide similarity of hun-167732010 to the 5 prototype strains along the entire genome ( figure a1 ) , except the near - central region of orf1 , which showed less similarity among ahev strains ; this region encodes the hinge region flanking the papain - like protease and the x domain . similarity in the genome was highest in the 5 end of the genome and the overlapping region of orf2 and orf3 . we found no evidence for recombination between hun-167732010 and genotypes 13 strains ( 13 ) , demonstrating that this strain was not generated through recombination from prototype ahevs . maximum - likelihood and neighbor - joining trees were constructed with the substitution model of tamura and nei , including gamma distribution shape ( g ) parameter ( tn93+g ) as implemented in mega 5 ( 14 ) ; 500 bootstrap replicates were used to test the reliability of the tree topology . phylogenetic analysis of the nearly complete ahev genome revealed 4 major clusters , where genotypes 13 strains formed previously recognized branches ( 10 ) ; the ahev strain , hun-167732010 , appeared to form a fourth cluster ( figure , panel a ) . to determine whether hun-167732010 shared genetic relatedness in the partial helicase gene with recently identified variants of european ahev strains ( 11 ) , we reanalyzed this region by using a larger sample set . our data reaffirmed that hun-167732010 forms an independent branch ( not shown ) . to identify the evolutionary driving forces beyond the heterogeneity of ahev genomes , we individually ran the alignments of all 3 orfs on the datamonkey server by using the random effects likelihood method ( www.datamonkey.org ) . evidence for strong selection pressure was found at the hinge region of orf1 involving 17 sites between amino acid positions 556 and 605 ( figure , panel b ) . however , 905 , 384 , and 3 sites within orf1 , orf2 , and orf3 , respectively , were under purifying selection across ahev genotypes . various genotypes of ahev have been proposed to represent clusters of strains typical for particular geographic locations . however , new evidence has shown that some genotypes could be present on different continents ( 6,10,11 ) . for example , although australia seemingly remained the only continent with genotype 1 ahev , different genotypes could be endemic to the americas and particularly europe , and these areas could show strain diversification . the origin of the novel ahev strain in hungary remains enigmatic ; understanding its recent emergence awaits analysis of further isolates within and outside europe . purifying selection was a key mechanism in the evolution of ahev , suggesting the existence of a strong structural and functional constraint against diversification that would lead to extensive amino acid changes within the 3 orfs in ahev strains . in contrast , positive selection was predicted primarily in the hypervariable hinge region flanking the protease and x domains in orf1 . however , recent findings have demonstrated that this region might be responsible for differences in replication efficiency ( 15 ) . thus , the positive evolutionary selection in this region could directly affect viral fitness and influence the pathogenesis of ahev infections . we provided further evidence for the marked sequence diversity among ahevs by providing the nearly complete genome sequence of a strain identified during a blsd outbreak in hungary . additional ahev genomes could facilitate more penetrating insights into their epidemiologic features and evolutionary mechanisms and also could serve as molecular bases for reliable and robust demarcation criteria in future classification proposals .

to explore the genetic diversity of avian hepatitis e virus strains , we characterized the near - complete genome of a strain detected in 2010 in hungary , uncovering moderate genome sequence similarity with reference strains . public health implications related to consumption of eggs or meat contaminated by avian hepatitis e virus , or to poultry handling , require thorough investigation .

fluorescein angiography ( fag ) is usually performed to evaluate retinal hemorrhage of unknown cause . in contrast , optical coherence tomography ( oct ) enables precise evaluation of the retinal structure in a noninvasive manner . for retinal hemorrhage , oct is also useful for observing a cross - section of the retina affected by hemorrhage . we herein report a case of partial tears of the retinal surface that was mistakenly followed - up as small retinal hemorrhages until oct clearly showed the defects of the retinal surface . a 62-year - old woman was referred to our hospital on suspicion of retinal hemorrhage involving the retinal vessels in her left eye ( figure 1 ) . at her first visit , visual acuity was 20/20 and as she did not have other symptoms she was to be followed - up . as the finding of her fundus had not changed 1 year later , fag and oct were performed to examine the cause of retinal hemorrhage . blocked fluorescence specific to retinal hemorrhage partial defects of the retinal inner layer were however shown by oct ( figure 3 ) . from these results , we diagnosed this case as partial tears of the retinal surface at the vitreoretinal juncture and not hemorrhage . differential diagnosis of retinal hemorrhage and partial tear of the retinal surface may be difficult in some cases , because both lesions have similar retinal colors and configurations . a case like the one we present here is probably overlooked without further examinations in routine work . oct is a useful and noninvasive device for the differential diagnosis of retinal hemorrhage and partial tear of the retinal surface . oct shows an image of partial tear of the retinal surface as a defect of the inner layer of the retina . foos ry emphasized the causative role of vitreous traction with partial tears of the retinal surface being caused by posterior vitreous detachment , and proposed three events that predispose to or cause small surface breaks : developmental thinning of the inner limiting lamina ; subsurface retinal degeneration ; and transmigrating macrophages . when small surface breaks become complicated by vitreous incarceration or by simple epiretinal membrane formation , direct posterior vitreous detachment may peel portions of the superficial retina , and the resulting large surface breaks may in turn provoke complex proliferative lesions of the vitreoretinal juncture.1 in our case , oct clearly showed the defect of the inner layer of the retina and small surface break .

differential diagnosis of retinal hemorrhage and partial tear of the retinal surface may be difficult in some cases . a 62-year - old woman was mistakenly followed - up for small retinal hemorrhages for more than 1 year . blocked fluorescence specific to retinal hemorrhage was not observed by fluorescein angiography ( fag ) . optical coherence tomography ( oct ) clearly showed defects of the retinal surface indicating partial tears of the surface at the vitreo - retinal juncture and not hemorrhage . oct is a useful and noninvasive device for the differential diagnosis of retinal hemorrhage and partial tear of the retinal surface .

sclerosing mucoepidermoid carcinoma with eosinophilia ( smece ) of the thyroid is a recently recognized primary malignant neoplasm of the thyroid gland associated with hashimoto 's thyroiditis / lymphocytic thyroiditis . till now , cases have been reported in the literature that are diagnosed histologically . and fine needle aspiration ( fna ) from smece often show nonspecific features and most often the diagnosis of a poorly differentiated or undifferentiated carcinoma is rendered in most of the cases ; but careful examination of cytological features and the background may sometimes be helpful in diagnosis . here , we describe the cytological features of this rare case along with its differential diagnosis . a 35-year - old female presented with thyroid swelling for 3 years and ipsilateral lymph - node swelling of 6-month duration . her physical examination revealed right - sided thyroid swelling of 4 cm 4 cm size and lymph - node of size 1.5 cm 1.5 cm . her thyroid profile revealed mild hypothyroidism , while routine blood examination and serum calcitonin levels were within normal limits . fna was performed with a 22-gauge needle from both thyroid as well from lymph node and on aspiration from both sites blood mixed material was obtained . the richly cellular smears showed predominantly round to oval cells lying singly as well as in loosely cohesive clusters . these cells had rounded hyperchromatic nuclei with an occasional prominent nucleus and a moderate amount of eosinophilic cytoplasm . a few cell clusters showed squamoid differentiation in the form of pyknotic to densely compact nucleus and deeply orangophilic cytoplasm [ figure 1a ] . at a few paces , the cells formed ill - defined epithelial pearl - like structures and only very occasionally the cells showed intranuclear cytoplasmic inclusion ( inci ) [ figure 1b ] . background population comprised predominantly of lymphocytes and a significant number of eosinophils ( 1 - 2 cells / hpf ) [ figure 1c ] . the important negative findings were the lack of mucus - secreting cells and intermediate cells . so , depending on the abovementioned cytological features , we made the diagnosis of a primary thyroid malignancy with squamous differentiation with a differential of poorly differentiated carcinoma , mucoepidermoid carcinoma with eosinophilia , and a metastasis from a different site . ( a ) smear reveals round to oval cells with hyperchromatic nuclei with a cell cluster showing deep cytoplasmic eosinophilia and dense compact chromatin ( h and e , 40 ) ( b ) tumor cells are forming epithelial pearl - like structure and a cell showing inci ( thin arrow ) ( h and e , 200 ) ( c ) background population consists of lymphocytes and eosinophils ( arrow ) ( h and e , 200 ) the patient underwent radical thyroidectomy . histological diagnosis was further confirmed immunohistochemically as the tumor was positive for thyroid transcription factor-1 ( ttf-1 ) and cytokeratin and negative for thyroglobulin and calcitonin [ figure 2 ] . ( a ) histology showed island of squamoid cells embedded in a sclerotic stroma and dense eosinophil cell infiltrate ( h and e , 100 ) ( b ) normal thyroid parenchyma showed lymphocytic thyroiditis ( h and e , 200 ) ( c ) calcitonin negative ( calcitonin , 200 ) ( d ) ttf-1 positivity ( ttf-1 , 200 ) ( e ) thyroglobulin negative ( thyroglobulin , 200 ) ( f ) cytokeratin positive ( cytokeratin , 200 ) smece is a rare slow - growing neoplasm of thyroid described in adults between 35 years and 70 years of age with a female predominance ( female to male ratio 17:1 ) . though a few studies have described its histological features , only an occasional reports have described the cytological features in which it has been described as a tumor with deceptively bland morphology or as carcinoma with cytoplasmic eosinophilia . cytologically , smece should be differentiated from benign conditions showing squamous metaplasia as well as from primary thyroid neoplasm that can show foci of squamous differentiation such as papillary carcinoma , medullary carcinoma , conventional mucoeidermoid carcinoma , and primary or metastatic squamous cell carcinoma . papillary thyroid carcinoma can be differentiated from smece by its characteristic nuclear features such as nuclear clearing , nuclear grooves , and inci . although , inci is present in very occasional cells but the diagnostic nuclear character was absent in the cells , rather the nucleus was hyperchromatic in the cells . medullary carcinoma have a characteristic nuclear chromatin pattern , i.e. , a salt - and - pepper pattern and usually lacks background population of lymphocytes and is usually associated with raised calcitonin levels . primary squamous cell carcinoma is extremely rare and is classified as anaplastic carcinoma while this neoplasm usually has more nuclear atypia and pleomorphism and is frequently associated with necrosis that lacks a background population of lymphocytes and eosinophils . most of the time straightforward cytological diagnosis of smece is not an easy task and on cytology there is often a dilemma about the diagnosis . likewise , we put forth the differential of a poorly differentiated carcinoma with squamous differentiation and have kept the differential of smece owing to the absence of characteristic nuclear features of papillary and medullary carcinoma , the presence of hyperchromatic nuclei , and the background population of eosinophils and lymphocytes that suggest background thyroiditis . histologically , smece is characterized by small nest and cords of tumor cells separated by fibrocollagenous sclerotic stroma and dense eosinophil cell infiltrate . immunohistochemically , smece is negative for thyroglobulin and calcitonin and positive for ttf-1 and cytokeratin . its constant association with hashimoto 's thyroiditis / lymphocytic thyroiditis has suggested that smece originates from the metaplastic squamous nest found in hashimoto 's thyroiditis / lymphocytic thyroiditis . a few have suggested follicular origin . while others suggested origin from c cell , parathyroid , ectopic salivary gland , and thyroglossal duct . though the definitive cytological diagnosis of this rare neoplasm is very difficult and often has cytologically nonspecific features but whenever a thyroid tumor exhibits squamoid features with the absence of characteristic nuclear features of papillary and medullary thyroid carcinoma one must keep the differential of smece . there are no conflicts of interest .

sclerosing mucoepidermoid carcinoma with eosinophilia ( smece ) of the thyroid is a rare primary thyroid tumor arising in a background of hashimoto's / lymphocytic thyroiditis and has been recently introduced in the world health organization ( who ) classification of thyroid tumors . it is characterized by extensive sclerosis , squamous and glandular differentiation , and inflammatory infiltrate rich in eosinophil . here , we are discussing the cytological features of this rare case in a 35-year - old female presented with thyroid swelling and lymph - node enlargement .

osteoclast - like giant cells ( ogc ) are multinucleated large cells with clear cytoplasm and are encountered in relatively uncommon tumors of the bone tissue and the tendon sheath . although these osseous tumors are generally benign by nature and have a slow growth , they still have a recurrence rate of over 50% and may give pulmonary metastasis . the presence of ogc in different extra - skeletal neoplasms has been cited in various literatures with the involvement of organs including the skin , breast , pancreas , gall - bladder and the kidneys . though the origin of these cells has been proposed to be of the mononuclear - macrophage lineage , their actual role in the histogenesis in these extra - osseous tumors is ill - defined . in this case report we present a detailed case of a patient with gastric adeno - carcinoma with ogc . a special focus will be laid on the immune reaction in the tumor tissue of such neoplasms with a brief discussion on the differential diagnosis of such neoplasms with anaplastic carcinomas which also manifest with atypical giant nucleated cells . a 70-year - old caucasian female was admitted to the surgical department of the st . detailed history revealed a 6-month history of nausea , epigastrial pain and progressive generalized weakness . a fibro - oesophago - gastro - duodenoscopy revealed a stomach with relatively rigid walls . a polypoid protrusion of the gastric mucosa along with multiple erosions with a tendency to bleed in the region of the gastric cardia and the small curvature was noticed . of the three biopsy specimens , the one obtained from the esophagus displayed signs of inflammation whereas the ones from the erosions depicted definite malignant changes corresponding to gastric adeno - carcinoma . following diagnosis , a gastrectomy with d1 lymph node dissection and oesophago - jejunostomy with an inter - intestinal anastomosis was performed . a tumor involving the gastric cardia and small curvature was presented as a polyp like mass with distinct ulcerative changes and blood clots on its surface . the cut surface of the tumor revealed a whitish - pink mass that had an invasive pattern of growth and involved all the layers of the stomach wall but without any encroachment into the surrounding organs . the tumor was diagnosed as being a low differentiated adeno - carcinoma with specific stromal infiltration accompanying multinucleated giant cells and extensive lympho - histiocytic infiltration ( figure 1 and figure 2 ) . the giant cells were predominantly located in the stroma around the malignant glands with seldom peri - glandular localization . the giant cells were characterized by a clear cytoplasm and multiple nuclei ranging between 3 to 7 , arranged either circumferentially or randomly in the cytoplasm . eight of the lymph nodes isolated from the region of the small curvature of the stomach were positive for metastases . the margins of gastrectomy presented with normal gastric tissue , thus confirming the success of the operation . the ptnm was inferred as pt3n2m0 . staining for different immunohistochemical markers , listed in table 1 were carried out on 6rm thick tissue samples placed on polyvinyl slides , with appropriate positive and negative controls . the labeled streptavidin biotin ( lsab - dako ) detection system was used for detection of primary antibodies . staining with pan- cytokeratin ( figure 4 ) was strongly positive for the poorly differentiated malignant gastric glands without any staining of the intervening stromal compartment . in contrast , the stromal compartment was highly stained by the cd68 marker , whereas the staining in the malignant epithelium was almost negligible ( figure 3 ) . interestingly , staining with cd4 and cd8 revealed mostly stromal infiltration by the latter and dual stromal and glandular infiltration by the former . note that the tumor tissue , and the regional lymph nodes , were positively stained for the latent nuclear antigen of ebv . ki67 immunostaining surprisingly showed total absence of any proliferation in the tumor tissue sample , while the anti - apoptotic marker , bcl 2 , was only moderately expressed . these neoplasms are of epithelial origin and were confirmed by positive pancytokeratin staining . given the intense immune reaction in these tumors , and presence of multinuclear giant cells , a crucial question remains as to whether such neoplasms are more aggressive than other adenocarcinomas without such elements . adeno - carcinomas of the stomach with ogc have to be also carefully differentiated from anaplastic carcinomas , which are usually characterized by faster growth , early metastasis and a much poorer prognosis . anaplastic cancer cells may present with several morphological features and may have from a single to multiple nuclei but in all cases the giant cells will be of epithelial origin and hence will stain negative to cd68 or other mesenchymal markers thus validating their epithelial nature . although the etiology of the lymphocytic infiltration of the tumors is not entirely known , ebv infection has been argued to be a probable cause and has been isolated in various cases of lymphoepithelioma - like tumors ( lelt ) . these tumors , although typical for the nasopharynx , have also been encountered in the stomach , bladder and intestinal mucosa , and are identifiable by lympho - histiocytic infiltration . furthermore , such tumors have also been cited to have a better prognosis in comparison to gc cases without ebv infection . the prognostic index in this case was proposed by running immunohistochemical tests on prominent independent markers , ki67 and bcl2 . while traditional view connects the over - expression of bcl2 to low tumor apoptotic ability and hence a poorer prognosis , studies have also linked the expression of bcl2 as an indicator for biological activity of tumor . in our case , we see mild expression of bcl2 and negative expression of ki67 , thus suggesting a low biological activity of the tumor i.e. higher index of apoptosis and lower proliferative index . these results must be , however , considered in the light of the advanced tnm staging , which remains the best predictor in terms of survival . to conclude , ogc represent uncommon tumors of the stomach . the rarity with which such neoplasms present in clinical practice makes it difficult to make universal conclusions about the prognosis . they must , however , be distinguished from anaplastic carcinomas of the stomach , with a more diffuse pattern of growth and a poorer prognosis overall .

out of all the different types of neoplasms affecting the stomach , gastric carcinomas with osteoclast - like giant cells ( ogc ) is one of the most uncommon . although ogc are typically found in osseous neoplasms and tumors of the tendon sheath , few cases of extra - skeletal neoplasms with ogc have been documented . these typically involve organs such as the pancreas , gall - bladder , kidney , and breast . even though the role of ogc in histogenesis of such tumors still remains unclear , their presence in extra - osseous neoplasms may indicate a certain level of immune reaction of the host towards the neoplastic transformation of normal tissue . we report a case of a 70-year - old caucasian female hospitalized for evaluation of epigastric pain . further examinations including endoscopy and biopsy of the stomach revealed gastric adenocarcinoma with ogc . this report also provides a brief insight into the possible immune reaction in such neoplasms

eschar is composed of dead tissue and dried secretions from a skin wound following a burn or an infectious disease on the skin . an eschar normally persists for less than a month before sloughing off or dissolving itself 1 . however , we encountered a patient with prolonged refractory accumulative eschar formation on her fingertips and finger pulps that remained for more than 2 years after a burn injury . the patient had experienced a burn injury on the hand from an alcohol flame 2 years prior . however , there were several areas of the fingertips and finger pulps where the new skin was thin , red , fragile , and sensitive . six months after the injury , the thin skin in these areas was replaced by excretive granulation tissues . initially , the secretions dried and a thin layer of crust formed . however , the crust thickened and led to formation of proliferating eschars . the accumulated thick eschar affected the function of the patient 's hands and interfered with everyday activities . to remove the eschars , the patient soaked her hand in water and removed the crust manually after the eschars had softened . physical examination revealed that the patient 's fingertips and finger pulps were covered with grayish , hard crusts as thick as 1.5 to 2 cm ( fig . the hand was in a functional position and the range of movement of dips and pips was mildly limited . fingers with accumulative eschar on ( left ) and with eschar just been removed ( right ) . to remove the eschars , both hands were soaked in warm saline solution for approximately one hour until all crusts were pale and soft . saline solution was used to irrigate the wound bed until healthy granulation tissue was exposed ( fig . 1 right ) . three hundred units of recombinant bovine basic fibroblast growth factor gel ( rbbfgf ) per square centimeter were applied to the granulation tissue surfaces daily . the wound decreased in size noticeably each day and was completely healed after 2 weeks of treatment . all affected dips could be fully extended , and the range of movement of these joints was normal . the patient had no difficulty performing everyday chores and had even returned to work as an accountant . although the regenerated skin on the affected areas appeared slightly red , the quality , thickness , softness , and elasticity was comparable to surrounding healthy skin . sensory function in the fingers was fully restored . there was no recurrence of any eschar on affected areas after the rbbfgf treatment ( fig . five months after treatment the burn wounds healed completely and there was not eschar recurred . the healing of burn wounds can be delayed if the wound is unattended or improperly managed . however , accumulative eschar formation caused by the proliferation of crust is not common ; the residual wound tends to become an ulcer . in this case , the wound had briefly healed and was covered by a thin layer of epithelium . however , the coverage was not sufficient for the fingertips and finger pulps . the fragile epithelium broke and was replaced by exudative granulation tissue that began the process of eschar formation . recombinant bovine basic fibroblast growth factor has been reported to increase the growth of new capillary vessels and to induce the differentiation of fibroblasts 2 . these changes allow the dermis to regenerate and provide a solid base for scattered epithelial islands to form healthy epidermis . in our case , rbbfgf healed the refractory burn wound quickly and allowed the formation of stable and durable regenerated skin . we believe that the location of the wound might be one of the main factors that contributed to eschar formation in this case . the wounds were on the fingertips and finger pulps , which might have been irritated whenever the patient used her hands . use of topical growth factor gel was the treatment of choice because it accelerated healing of the wound and regenerated highquality dermis .

key clinical messageeschar formation is a potential sequela of burn injuries . definitive management may include escharectomy and eschar debridement . after eschar removal , the wound can be covered with a skin graft or reepithelialization . for prolonged refractory eschar on the fingertips , topical use of rbbfgf after debridement can achieve an optimal outcome .

the patient had received medical treatment due to a mild fever , cough , and yellowish sputum that developed 10 days prior , but the symptoms had not improved . furthermore , his oral intake had become worse 5 days before and hemoptysis started 2 days before admission to our hospital . at first , about a 10 ml of bloody sputum developed , but the amount progressively increased and eventually approximately 100 ml of bloody sputum was produced per hour . the patient was a white - collar worker , who had smoked 1 pack of cigarettes per day for 40 years and had consumed 1 alcoholic drink per day , 4 times a week , for 40 years . he had been diagnosed with diabetes mellitus at a regional hospital 3 years earlier and was still on medication . he was also diagnosed with bronchiectasis at a regional hospital 3 years earlier , and received medical treatment for the bronchiectasis 1 year earlier . at the time of that treatment , he did not undergo a bronchial artery embolization , but continued to receive drug therapy . the three months prior to admission at our hospital , episodic chest tightness developed , which became aggravated especially after intensive exercise . he reported that a reevaluation of his chest tightness and dyspnea at a regional hospital had not revealed any specific findings other than bronchiectasis . when admitted to our hospital , his blood pressure was 117/59 mmhg , heart rate was 87 beats / min , respiratory rate was 19 breaths / min , and body temperature was 37.6. the laboratory test results were within normal limits except the wbc ( 12,400/l ) and crp ( 10.08 ) . the electrocardiogram on admission showed a normal sinus rhythm and a chest radiograph showed a bronchiectatic change in the left lower lung ( fig . 1 ) . contrast - enhanced multidetector chest computed tomography ( mdct ) revealed a severe cylindrical bronchiectatic change in the left lower lung field , and tortuous and anomalous communicating vascular crowding around the left hilum that was communicating with the right coronary artery ( rca ) ( fig . 2 ) . since the transthoracic echocardiography revealed decreased wall motion in the rca territory , a coronary angiography was performed to rule out coronary vascular disease . coronary angiography via the radial artery revealed a coronary - bronchial artery fistula originating from the rca and extending to the bronchial artery . however , we performed surgical treatment in an attempt to treat the underlying bronchiectasis . under general anesthesia , a posterolateral thoracic incision was made . a left lower lung lobectomy was performed and the coronary - bronchial artery fistula was removed . his clinical symptoms including chest tightness and dyspnea improved , and he was discharged from the hospital . at a 24-month follow - up , his postoperative course was found to be uneventful without any recurrence . a coronary - bronchial artery fistula is a rare congenital anomaly that occurs at the coronary artery . it has been reported that this anomaly is detected in 0.18% of patients who undergo coronary angiography . with advances in diagnostic technologies such as mdct , many cases of coronary - bronchial artery fistula have recently been reported . however , its pathogenesis has not yet been elucidated . most cases of coronary - bronchial artery fistula have no clinical implications because they are asymptomatic , whereas some cases are related to the tetralogy of fallot , a supraventricular aortic stenosis , aortitis syndrome . several previous reports have proposed that patients with a coronary - bronchial artery fistula need aggressive treatment , including embolization , in order to prevent lethal complications such as infective endocarditis or an aneurysmal rupture . a comprehensive physical examination and thorough review of a patient 's history are extremely important for determining the diagnosis of this fistula . contrast - enhanced mdct with retrospective electrocardiogram gating is an accurate and non - invasive screening test , which allows for more acceptable diagnostic outcomes with advances in ct resolution . echocardiography is essential to the evaluation of concurrent cardiac anomalies and left - to - right shunts . in our patient , shin et al . have demonstrated that functional tests including the myocardial perfusion scanning are helpful in the diagnosis of coronary - bronchial artery fistula . a great variety of clinical features have been reported in patients who has coronary - bronchial artery fistula . hackett and hallidie - smith reported a case of spontaneous closure of a coronary artery fistula secondary to thrombosis . the severity of the clinical features of a coronary - bronchial artery fistula depends on the degree of the left - to - right shunt . as the degree of the left - to - right shunt increases , complications such as pulmonary hypertension or congestive heart failure . a few cases of rupture or thrombosis of coronary artery fistulas have been reported in association with arterial aneurysms or coronary steal phenomena . our patient presented with only chest pain and hemoptysis , and the diagnosis of a coronary - bronchial fistula was established by mdct , which was performed to assess massive hemoptysis . it seems likely that recurrent life - threatening hemoptysis may be attributed to coronary - bronchial artery fistula associated with bronchiectasis , and that patients with such fistulas can be treated surgically . however , since a standard treatment method for coronary fistulas has not yet been determined , most patients have been managed in light of anecdotal case reports or small series of cases . further studies with a larger number of cases are needed to understand the clinical features and to establish a standard treatment modality . closure of coronary - bronchial artery fistula is mainly indicated for patients with heart failure , myocardial ischemia , or high - flow shunting . shin et al . have proposed prophylactic and therapeutic embolization of coronary - bronchial artery fistula in patients with bronchiectasis . however , patients with such fistulas should be treated based on the presence of concurrent cardiac anomalies as well as the clinical evaluation of other organs . embolization can be safely performed after the anatomical relationships between the fistula and the surrounding structures are completely assessed . kang et al . have indicated that contrast - enhanced mdct coronary angiography is helpful in the identification of the course of a coronary - bronchial artery fistula . we reported the case of a patient with a coronary - bronchial artery fistula who presented with angina symptoms and bronchiectasis and was successfully treated . further studies are needed to understand the precise pathogenesis of the fistula and the relationship between the fistula and bronchiectasis .

a coronary - bronchial artery fistula is a very rare congenital anomaly of the coronary artery whose etiology and pathogenesis have not yet been clarified . most patients with coronary - bronchial fistulas are asymptomatic ; however , some patients present with congestive heart failure , infective endocarditis , myocardial ischemia induced by a coronary steal phenomenon , or rupture of an aneurysmal fistula . furthermore , patients with a coronary - bronchial artery fistula rarely manifest life - threatening hemoptysis due to the associated bronchiectasis . we report herein the case of a patient with a coronary - bronchial artery fistula who had bronchiectasis and a history of massive hemoptysis and myocardial ischemia .

brachial plexus block used for upper limb surgery avoids the use of general anesthetics , provides excellent analgesia , and facilitates ambulatory surgery . phrenic nerve palsy is a common complication after both interscalene and supraclavicular brachial plexus blocks,[13 ] but is more common with the interscalene approach . we report unilateral phrenic nerve paresis along with severe respiratory distress and bilateral bronchospasm following ultrasound - guided ( usg ) supraclavicular brachial plexus block . a 58-kg , 56-year - old woman was scheduled for emergency debridement and tendon repair of right hand . patient was a known hypertensive on therapy with amlodipine 10 mg once a day for 5 years . her preoperative vitals were pulse rate of 84/min and blood pressure 150/90 mmhg in supine position . routine biochemical / hematological investigations and electrocardiogram ( ecg ) were within normal limits . after taking informed consent for supraclavicular block , the patient was shifted to the operation room . an 18 g intravenous ( iv ) cannula was secured and midazolam 2 mg iv was given . under strict aseptic precautions , usg right supraclavicular brachial plexus block was given with a 5-cm nerve block needle , using mixture of local anesthetic 10 ml 0.5% bupivacaine and 10 ml of 2% lignocaine with 1:200,000 adrenaline . about 15 min after the block , the patient became restless , complained of breathing difficulty , and became tachypneic with audible wheeze . patient was propped up and given oxygen supplementation with facemask which improved the oxygen saturation ( spo2 ) from 70% to 85% . two doses of salbutamol nebulization were given , but there was no further improvement in spo2 . patient received salbutamol nebulization and maintained spo2 of 92% with 60% oxygen supplementation delivered by venturi facemask . chest x - ray showed features of right phrenic nerve paresis [ figure 1 ] one hour post block , she became more comfortable , vitals were stable with improved breath sounds bilaterally , and oxygen supplementation was gradually withdrawn . the repeat chest x - ray showed normalization of the elevated right hemidiaphragm and patient was maintaining saturation on room air . incidence of phrenic nerve paresis after interscalene block is greater than supraclavicular,[13 ] probably because the injection point is more cephalad and closer to the cervical plexus in the interscalene approach . despite a decrease in pulmonary function after phrenic nerve palsy , healthy patients may remain asymptomatic as they are able to compensate for the transient decrease in ventilatory function . the close proximity of the phrenic nerve to the site of these injections may lead to spread of local anesthetics to the nerve . close to its origin , the phrenic nerve may give a communicating branch to c5 root of brachial plexus . studies have shown decrease in spirometric volumes after interscalene block , resulting from diaphragmatic paresis . most patients remain asymptomatic though it causes inability to fully inspire to total lung capacity , with resultant decrease in all pulmonary volumes and flows . although the phrenic nerve is blocked in brachial plexus block by both interscalene and supraclavicular approaches , the effects on reduction in forced vital capacity ( fvc ) is different . with interscalene block the incidence of diaphragmatic hemiparesis is 100% and it is accompanied by a 25% reduction in fvc . however , with supraclavicular block , although 50% patients have diaphragmatic paresis , there is no reduction in fvc . one incident of phrenic nerve block leading to dyspnea and reduced air entry after supraclavicular block in an obese patient has been reported . transient hemidiaphragmatic paresis may not be tolerated by obese as their ventilatory reserves are low . bronchospasm after interscalene block in nonasthmatics patients has been reported and can occur in presence of phrenic nerve block and reduction in ventilatory parameters following interscalene block . the local anesthetics when injected in interscalene groove may have spread to opposite side if midline septa were deficient and may have spread upward and downward as far as t4 segments . blockade of t1t4 sympathetic nerve supply may precipitate bronchoconstriction due to parasympathetic overactivity . in our patient , although the drug was injected in supraclavicular plexus , such a spread can not be ruled out . recurrent laryngeal nerve can be blocked if the injection is too deep along the posterior border of sternocleidomastoid and this can lead to bronchospasm . respiratory distress in the case reported was due to unilateral phrenic nerve paresis . in case respiratory distress occurs immediately following a supraclavicular block , phrenic nerve paresis should also be considered and measures to deal with unanticipated respiratory distress must be initiated .

ultrasound - guided peripheral nerve blocks facilitate ambulatory anesthesia for upper limb surgeries . unilateral phrenic nerve blockade is a common complication after interscalene brachial plexus block , rather than the supraclavicular block . we report a case of severe respiratory distress and bilateral bronchospasm following ultrasound - guided supraclavicular brachial plexus block . patient did not have clinical features of pneumothorax or drug allergy and was managed with oxygen therapy and salbutamol nebulization . chest x - ray revealed elevated right hemidiaphragm confirming unilateral phrenic nerve paresis .

a 67-year - old male with a history of non - obstructive coronary artery disease , chronic obstructive pulmonary disease ( copd ) , and bronchiectasis presented to the emergency department with abdominal pain of 8 h duration . he had no documented history of hypertension , and his medications included atorvastatin 20 mg daily and aspirin 81 mg daily . a ct scan of the abdomen and pelvis with intravenous iodinated contrast was performed , which revealed an infrarenal aaa measuring 6.5 cm in diameter . there was extensive mural thrombus , a patent lumen , and no evidence of frank rupture . there was a sharp , focal extension of the opacified lumen extending into the mural thrombus , and a hyperattenuating crescent in the wall of the thrombus ( fig . adjacent to the aneurysm , there was stranding of the mesenteric fat , as well as a collection of fluid obscuring the anterior margin of the psoas muscle ( fig . ( a ) sagittal ct of the abdomen and pelvis with contrast shows a 6.5 cm aaa with the hyperattenuating crescent sign seen on both the anterior ( white arrowheads ) and posterior ( black arrowheads ) margin of the aorta . ( b ) axial ct of the abdomen with contrast shows an aaa with a focal fissure extending into the mural thrombus ( black arrowhead ) and the hyperattenuating crescent sign seen as a crescent - shaped area of enhancement ( white arrowheads ) in the lateral margin of the thrombus . the anterior margin of the psoas muscle is obscured by a blood collection ( asterisk ) , representing a sentinel leak . within 15 min of the scan , the patient was intubated and placed on a cardiac monitor , and an external jugular venous central line was placed . his heart rhythm deteriorated to ventricular fibrillation , and after unsuccessful resuscitation efforts , including defibrillation , the patient died . aneurysms frequently present with subtle and non - specific signs , yet diagnosis of aneurysms is crucial because of the catastrophic complications that can occur . aaa rupture has a mortality rate of 81% ( 3 ) , and interventions such as endovascular aneurysm repair or open surgery are performed to prevent rupture when the risk of rupture is significant ( 5 ) . the pathogenesis of aaa is a multifactorial process , with underlying genetic , inflammatory , and autoimmune components ( 7 , 8) . after an aneurysm has formed , many factors associated with greater risk of rupture have been identified including maximum diameter of the aneurysm , rate of increase in aneurysm size , hypertension , age , smoking history , copd , bronchiectasis , and family history of aneurysms ( 911 ) . among these factors , the maximum aaa diameter remains the most widespread criterion to predict risk of aaa rupture ( 5 , 11 ) . there is a direct relationship between the size of the aneurysm and risk of rupture , although size alone is not an adequate predictor . other parameters , including aaa expansion rate , intraluminal thrombus thickness , and wall stress , all play a role ( 5 ) . even in asymptomatic unruptured aneurysms , repair of the aneurysm is indicated when the aneurysm exceeds 5.5 cm in size in patients with an acceptable surgical risk ( 1 ) . in recent years , ultrasound has become an increasingly useful modality for the initial detection and size measurement of aaa and has been shown to have a sensitivity of 98.9% , and a specificity of 99% ( 6 , 12 ) . for patients in whom aaa is suspected , ultrasound is the preferred initial imaging test and may be conducted at bedside ( 5 , 6 ) . although ultrasound is useful for identifying aneurysms , it has limited ability to characterize features of impending rupture . in some cases , the usefulness of ultrasound may be limited by the patient 's body habitus , or bowel gas may obscure visualization of the abdominal aorta ( 13 ) . generally , ct with iv contrast is the preferred imaging modality when an aaa has been identified on ultrasound , or when the patient is experiencing severe symptoms , a pulsatile abdominal mass , or has significant risk factors for aaa ( 5 ) . ct may reveal rupture , features of an impending rupture , or an alternative diagnosis for the patient 's symptoms . with the administration of iv contrast , the mural thrombus is believed to be protective from rupture , and a thinner mural thrombus is associated with higher rupture risk ( 14 ) . in cases of impending rupture , the contrast - enhanced blood may be seen penetrating into mural thrombus lining the aneurysm . this focal fissurization represents a tract of blood extruding into the unstable thrombus ( 10 ) . if blood transits beyond the mural thrombus , it may travel along the intimal margin of the aorta and perfuse the periphery of the organized thrombus ( 15 ) . 1 . the hyperattenuating crescent sign has a sensitivity of 77% and specificity of 93% for rupture , pseudoaneurysm , or intramural hematoma found at the time of surgery ( 16 ) . after extending through the mural thrombus often , these collections are seen within the psoas muscle , or obscuring the anterior surface of the psoas ( 17 ) . in cases of impending rupture , prompt intervention is necessary . emergent consultation with a vascular surgeon is required to establish a plan for definitive therapy . blood pressure control is important for stabilization of unruptured aneurysms ( 5 ) . in the event of aneurysm rupture , although counterintuitive , physicians should consider deferring fluid resuscitation if the patient is conscious , and systolic pressure is at least 5070 mmhg ( 19 ) . larger transfusion volume requirements as well as large retroperitoneal hematomas have been associated with increased risk for abdominal compartment syndrome , characterized by intra - abdominal hypertension and multi - organ dysfunction ( 20 ) . a thorough knowledge of the risk factors and tomographic imaging features of impending rupture allow physicians to be prepared to respond decisively . important risk factors for rupture include maximum diameter of the aneurysm , smoking history , and age . on ct scan , focal fissuring of thrombus , the hyperattenuating crescent sign , and small aortic leaks may signify an impending rupture and should prompt rapid surgical consultation . in cases of rupture , volume resuscitation is indicated only when the patient is unresponsive , or if systolic pressure is less than 70 mmhg . all authors have made substantial contributions to the conception and design , analysis , and interpretation of information , have been involved in drafting the manuscript and revising it critically for important intellectual content , and have given final approval of the version to be published . the authors have not received any funding or benefits from industry or elsewhere to conduct this study .

abdominal aortic aneurysm ( aaa ) may present with subtle clinical findings . recognition of the imaging features of an impending rupture is key for timely diagnosis . this report reviews the classic computed tomography findings of impending aaa rupture and presents a recent case which illustrates the key features .

occipital neuralgia ( on ) is a condition characterized by a paroxysmal stabbing pain in the area of the greater or lesser occipital nerves ; it is usually regarded by clinicians as idiopathic . some have suggested that on can be induced by trauma or injury of the occipital nerves or their roots , but tumor has rarely been reported as a cause of on . we report herein a case of foramen magnum meningioma in a 55-year - old woman who presented with on triggered by head motion as the only symptom without any signs of myelopathy . occipital neuralgia ( on ) is a condition characterized by a paroxysmal stabbing pain in the area of the greater or lesser occipital nerves ; clinicians usually regard it as idiopathic.1,2 some have suggested that on is the result of trauma or injury of the occipital nerves or their roots ; tumor has rarely been reported as a cause.2 we report herein a patient with on as the only presenting symptom of meningioma in the craniocervical junction in order to alert clinicians to consider the underlying causes of on . a 55-year - old woman presented with increasing episodes of a paroxysmal headache on the right occipital region , superior and posterior to the ear , over the previous 3 months . she complained of abrupt - onset , sharp , electric - like pain in the right occiput radiating toward the vertex . she perceived a dull ache in the right occipital region while lying down on her back . a neurological examination produced normal results with no hypesthesia in the affected area , no cranial autonomic features , and no occipital skin lesions . magnetic resonance image ( mri ) of the cervical spine showed a gadolinium - enhanced extraaxial mass within the right aspect of the foramen magnum at the craniocervical junction , measuring 2.72.22.3 cm ( fig . the mass extended from the medulla oblongata to the c1-c2 level of the spinal cord , deforming the right aspect of the upper cervical spinal cord and medulla and interrupting the right c2 nerve root . the mass was excised and found to be a meningioma that had originated from the arachnoid near the dentate ligament , pushing against the right vertebral artery and encasing the c1 , c2 , and cranial xi nerves . the findings for this patient indicate that a mass in the craniocervical junction can cause on without other definite neurologic symptoms or signs . it is stated in the literature that most ons are idiopathic , but that they may arise as a result of inflammation , trauma , or compression of the occipital nerves or their roots , or from an isolated upper cervical cord lesion.2 - 6 the diagnostic criteria for on as defined by the international headache society ( ihs ) include paroxysmal stabbing pain with or without persistent aching between paroxysms in the area of distribution of the greater , lesser and/or third occipital nerves , tenderness over the affected nerve , and pain that eases temporarily after local anesthetic nerve block.1 the pain is thought to emanate mainly from the greater and lesser occipital nerves or the nerve roots of c2 or c3 , which are the origins of the greater and lesser occipital nerves.1 - 3 even though our patient fitted the current ihs criteria for on except one criterion of a decrease in pain with anesthetic blockade , prolonged pain for as long as 3 months and pain aggravation with head flexion or when lifting heavy objects suggested the presence of a structural lesion . in our case , the initiation of pain that was apparent following head motion and lifting might be attributable to increased pressure inside the cranium causing pressure on the mass , resulting in excruciation of the c2 nerve root . mass lesions such as osteochondroma , schwannoma , or arthrosis of the c1-c2 joint involving the c2 root have been reported to cause on without other definite neurological signs.4 - 6 common characteristic features of these on cases with mass lesions are prolongation of the period of on up to 1 year and pain exacerbated by neck motion . idiopathic on cases may present with pain aggravation with neck motion , but this is rare . therefore , careful medical attention and consideration of cervical spine mri are needed for on produced by neck motion that has persisted for a relatively prolonged period . meningiomas account for approximately 25% of all spinal tumors ; only 2% of spinal meningiomas occur in the foramen magnum.7 meningioma of the foramen magnum is a potentially lethal condition when unrecognized , resulting in progressive myelopathy with quadriplegia , dysphagia , and sphincter disturbance.7 in our patient , the foramen magnum meningioma deformed the upper cervical cord and medulla ; however , it presented with on as the only symptom without any signs of myelopathy . because of its rarity as a cause of on , prompt diagnosis of the compressive lesion is difficult and hence may be delayed . the early diagnosis of compressive lesions in on is important not only because this offers the possibility of curing the condition , but also because an unrecognized foramen magnum mass is a potentially lethal condition . careful evaluation of the upper cervical spine and posterior fossa is therefore mandatory , particularly in the setting of on aggravated by neck motion . the present case broadens the spectrum of causes of on and suggests that cervical spine mri is useful in on even without clinically definite evidence of myelopathy .

backgroundoccipital neuralgia ( on ) is a condition characterized by a paroxysmal stabbing pain in the area of the greater or lesser occipital nerves ; it is usually regarded by clinicians as idiopathic . some have suggested that on can be induced by trauma or injury of the occipital nerves or their roots , but tumor has rarely been reported as a cause of on.case reportwe report herein a case of foramen magnum meningioma in a 55-year - old woman who presented with on triggered by head motion as the only symptom without any signs of myelopathy.conclusionsthis case indicates that it is important to consider the underlying causes of on . precise neurologic and radiological evaluations such as cervical spine magnetic resonance imaging are needed .

in the course of time , pathologists , anthropologists and radiologists pointed out this situation1,2 ) . more frequently this condition affects females over 60 years of age with no special reference to race or geographical area1 - 4 ) . remodelling of the compensatory spongy bone ( diploe ) , due to erosion on the external table ( the compact bone ) , has resulted in findings of diploe layer waste on histopathologic examination1 ) . therefore , we aim to present a patient with biparietal osteodystrophy , who had parietal bone fracture and epidural hematoma underneath following a minor head trauma . a 78-year - old woman visited to the emergency service with a severe headache after hitting her head to the wardrobe . at first , a neurological examination seemed normal but the patient has undergone computerized tomography ( ct ) scan because of increasing headaches . the ct scan revealed non - depressed linear fracture on the left parietal bone and epidural haematoma underneath ( fig . irrelevantly , biparietal bone thinning with lack of diploe layer and external table was pointed out . patient was treated by the conservative management . no neurological deterioration or a radiological increase of epidural haematoma bilateral thinning of parietal bone is a condition that has been known since the 18th century . in the course of time pathologists , anthropologists and radiologists current definition was changed by the understanding of that it is not an anatomical variety or result of growing old in time . however , it was described as " senile bone atrophy , senile biparietal atrophy and senile cranial atrophy " , firstly by emphasizing atrophy relevant to aging . histological evidence and new cases with younger ages have changed that point of view1,2,4 ) . currently , it is described as " biparietal ostedystrophy " by retrospective radiologic researches and histopathologic examinations . estimated prevalence is 0.4 - 0.5% according to radiological scans , case reports and anthropologic researches1 ) . whereas cederlund et al.2 ) reported its prevalence as 0.25 - 0.8% . however , biparietal osteodistrophy occurs mostly in females over 60 years old , in younger ages , male cases have been reported much more than female cases1 ) . however , diagnosed sporadically , there are some family case reports that considered autosomal transmission1 ) . clinical presentations show differences in pains in the areas thinning to simple local pain due to fractures that may lead to death4 ) . review of literature shows that most diagnoses have been done by retrospective radiologic scans such as simple radiography or ct scan . radiologic investigations generally reveal oval shaped bone loss in the area between parietal prominence and sagittal suture bilaterally4 ) . some authors investigate parietal atrophy under two topics ; simple flattening on both parietal bones and groove - like ( crater - like ) biparietal atrophy1 ) . examinations exposed relevance with osteoporosis and decreased bone formation instead of increased bone destruction4 ) . however , some studies report that external destruction of bone causes loss of external table and therefore triggers remodelling of diploe layer but that remodelling regressed in time1 ) . many theories have been suggested to determine the reason . also , chronic trauma is one of the considered factors of pathology . that idea was brought forward by archaeologists because they thought that pharaohs ' helmet , " pschent " , effected by its pressure . however , even today there are many people who carry load on their heads , the pathology does not exist as before . epidural haematoma underneath parietal fracture due to minor head trauma is generally an unexpected situation . although our patient had no findings before , incidental minor head trauma on pathologic bone segment led the clinical situation . the absence of external table and diploe layer , old age and female sex are compatible with biparietal osteodystrophy .

thinning of parietal bone bilaterally is extremely rare but well known phenomenon . approximate prevalence is 0.4 - 0.5% according to radiological scans , case reports and anthropologic researches . even though biparietal osteodystrophy occurs mostly in over 60-year - old women , it shows no special association with race or geographical area tendency . current definition was changed by understanding that is a pathological situation , not an anatomical variety or result of growing old in time . biparietal osteodystrophy may have an unusual presentation and treatment still remains unclear . we aim to present a patient with biparietal osteodystrophy associated with minor head trauma that caused parietal fracture and epidural hematoma underneath .

in june 2006 , a 74-year - old woman residing in a house in rural new orleans was bothered by a considerable number ( > 50 ) of insect bites . the woman observed many bugs in the house and showed them to a fumigator , who identified them as triatomines . an internet search showed the potential for transmission of chagas disease , and the woman sought help from a local health sciences center . serum samples from both residents of the house were tested for antibodies to t. cruzi at the centers for disease control and prevention ( cdc ) by an indirect fluorescent antibody ( ifa ) test . samples were also tested at loyola university ( new orleans , la , usa ) and then at cdc . by using an experimental dipstick assay ( trypanosoma detect ; inbios international inc . , the woman resident was positive for antibodies to t. cruzi by ifa at dilutions of 1:128 ( 4 weeks after being bitten ) and 1:64 ( 10 weeks after being bitten ) and by dipstick assay . she was positive for trypanosomes by hemoculture testing with 10 ml blood and coculture in macrophages ( 13 ) 4 months after being bitten . trypanosomes consistent with t. cruzi were observed in culture beginning on day 46 of culture , and amplification of a t. cruzi specific 24s rrna gene target confirmed that the isolate was t. cruzi . the index resident had a history of 5 trips to areas endemic for chagas disease : zacatecas , mexico ( 1970 ) ; cozumel , mexico ( 1990 ) ; belize ( 1991 ) ; guatemala ( 19988 ) ; and costa rica ( 1998 ) , each of < 2 weeks duration , with stays in improved tourist hotels ( less likely to harbor triatomines ) except for the belize trip , which included an 1-week stay in a palm thatch - roofed cabin . she had not used intravenous drugs or had a blood transfusion or organ transplant , and she is not the daughter of latin american immigrants . except for fatigue , cardiac evaluation that included an electrocardiogram showed normal results , and she decided not to take medication . her residence of 29 years was located on 7.66 acres of bottomland hardwood forest , with many gaps that provided ready access for insects . a house inspection showed fecal streaks characteristic of triatomines on walls , which were identical to what the patient reported seeing on her nightgown . twenty dead adult triatomines were collected in the house ( after fumigation ) and in another building on the property that contained a bed . no nymphs or eggs were found , which suggests that the house had not been colonized . one live second - stage nymph was collected in a nearby armadillo burrow 50 m from the house . all triatomines collected were identified as t. sanguisuga according to the key of lent and wygodzinsky ( 6 ) ( figure ) . because all triatomines except the nymph were dead , pcr was used to determine t. cruzi infection status ( 14 ) . the last 2 segments of the abdomen were removed from each insect , placed in 200 l 1 pcr buffer ( applied biosystems , foster city , ca , usa ) , boiled for 15 min , and centrifuged . a total of 5 l of supernatant was amplified in a 50-l reaction ( 3.5 mmol / l mgcl2 and 2 u taq dna polymerase ) . the primers used anneal to the t. cruzi minicircle dna and were tc3 : 5-ttgaacgcccctcccaaaac-3 and tc4 : 5-gattggggttggtgtaatata-3. the cycling parameters were an initial denaturation step at 94c for 3 min ; 35 cycles at 94c , 55c , and 72c , each for 1 min ; and a 10-min extension at 72c ( programmable thermal controller ; mj research , watertown , ma , usa ) . twenty percent of the pcr product was subjected to electrophoresis on a 1.8% agarose gel and visualized by uv transillumination after staining with ethidium bromide . a positive control of 5 l of t. cruzi parasites boiled in 1 pcr buffer and a negative control without the dna template were included with every pcr . samples that failed to amplify were spiked with 5 l of t. cruzi parasites boiled in 1 pcr buffer and reamplified to ensure that the lack of product was not caused by inhibition of the pcr . more than half of the triatomines were positive for t. cruzi ( 56% , 10/18 ; 3 failed to amplify ) , with more positive females ( 73% , 8/11 ) than males ( 50% , 3/6 ) . plasma from the resident dog and 7 other dogs living 1 mile away all tested negative by ifa at cdc . the assertion that the patient contacted t. cruzi in louisiana is strongly supported by limited travel history to disease - endemic areas and stays mostly in improved housing ( risk for chagas disease transmission is associated with longer residence in disease - endemic areas ) , lack of other risk factors , and large numbers of infected t. sanguisuga in the house . however , the streaks on her nightgown consistent with triatomine feces indicate exposure , and the parasite could have been introduced into any of her numerous bite wounds . however , hurricane katrina had hit the area 9 months earlier and increases in domestic infestation with triatomines have been previously reported after a hurricane ( 15 ) . anecdotally , the armadillo population increased substantially in the months after hurricane katrina , and one can speculate that these hosts supported a larger bug population , who later sought other bloodmeal sources as the armadillo population returned to prestorm levels .

autochthonous transmission of the chagas disease parasite , trypanosoma cruzi , was detected in a patient in rural new orleans , louisiana . the patient had positive test results from 2 serologic tests and hemoculture . fifty - six percent of 18 triatoma sanguisuga collected from the house of the patient were positive for t. cruzi by pcr .

plants were grown under greenhouse conditions ( 16-h day ) at the university of arizona during january june 2011 , and self - pollinated to obtain 6-dap kernels . using a protocol for laser - capture microdissection ( lcm ) of plant tissues , and further modified using methods that were subsequently published , we isolated the central portion of the starchy endosperm ( fig . 1 ) with a leica lmd 6500 instrument ( leica microsystems , inc . ) . the captured region included the presumptive central starchy endosperm and a portion of the conducting zone cells , , . 13 , june 2011 ) were obtained from nine separate kernels ( three each ) on a single ear while sections for the fourth replicate ( rep . 4 ) were obtained from a separate ear ( on a separate plant , march 2011 ) . rna was extracted from microdissected sections using an arcturus picopure rna isolation kit ( applied biosystems / life technologies , cat . kit0204 ) , and its size and integrity were evaluated using an rna 6000 pico kit ( agilent technologies , cat . 5067 - 1513 ) on an agilent 2100 bioanalyzer ( agilent technologies ) before and after dnase treatment ( fig . 1 about 5060 ng of dnaase - treated rna was used as template for cdna synthesis in order to amplify the captured rna ( two rounds ) using a t7 polymerase - based linear amplification system ( arcturus riboamp hs plus rna amplification kit , applied biosystems / life technologies , cat . kit0525 ) to produce a peak size of ~ 200500-nt rna fragments ( fig . 1 ) . standard barcoded rna - seq libraries were generated to facilitate sequencing of four samples using a single illumina flowcell lane . cluster generation and sequencing was carried out on the illumina cluster station and genome analyzer iix ( gaiix ) instrument using single read cluster generation kit ( cat . 15003925 ) , respectively . 82 cycles of imaging were carried out using a modification of the ga2_76cycle_sr_v7.xml sequencing program . methods for rna - seq data filtering and processing were essentially as those described previously . nearly 24.30 million reads were generated from the four replicates ranging from 4.33 million in rep . 2 . using tophat , ~ 88.590.2% of the reads were mapped to the reference b73 genome ( release 5b.60 ) ( table 1 ) . using bedtools , 31,130 of the 110,028 genes in the working gene set ( wgs ) were detected in at least 2 replicates with at least one raw read count . inter - sample differences in library size were eliminated by total count ( tc ) normalization method . read counts for all the genes in wgs were divided by the total counts of mapped reads ( or library size ) associated with their sample and multiplied by the mean total count across all the samples of the dataset to calculate the normalized read counts and deposited in geo . similarity of expression profiles between the replicates was determined by a pearson correlation coefficient ( pcc ) analysis . the four replicates showed high correlation with each other , with pcc scores ranging from 0.95 to 0.96 ( fig . 2 ) . together , the evidence suggests that our lcm - generated rna - seq data is highly reproducible . plants were grown under greenhouse conditions ( 16-h day ) at the university of arizona during january june 2011 , and self - pollinated to obtain 6-dap kernels . using a protocol for laser - capture microdissection ( lcm ) of plant tissues , and further modified using methods that were subsequently published , we isolated the central portion of the starchy endosperm ( fig . 1 ) with a leica lmd 6500 instrument ( leica microsystems , inc . ) . the captured region included the presumptive central starchy endosperm and a portion of the conducting zone cells , , . 13 , june 2011 ) were obtained from nine separate kernels ( three each ) on a single ear while sections for the fourth replicate ( rep . 4 ) were obtained from a separate ear ( on a separate plant , march 2011 ) . rna was extracted from microdissected sections using an arcturus picopure rna isolation kit ( applied biosystems / life technologies , cat . kit0204 ) , and its size and integrity were evaluated using an rna 6000 pico kit ( agilent technologies , cat . 5067 - 1513 ) on an agilent 2100 bioanalyzer ( agilent technologies ) before and after dnase treatment ( fig . 1 about 5060 ng of dnaase - treated rna was used as template for cdna synthesis in order to amplify the captured rna ( two rounds ) using a t7 polymerase - based linear amplification system ( arcturus riboamp hs plus rna amplification kit , applied biosystems / life technologies , cat . kit0525 ) to produce a peak size of ~ 200500-nt rna fragments ( fig . 1 ) . standard barcoded rna - seq libraries were generated to facilitate sequencing of four samples using a single illumina flowcell lane . cluster generation and sequencing was carried out on the illumina cluster station and genome analyzer iix ( gaiix ) instrument using single read cluster generation kit ( cat . 82 cycles of imaging were carried out using a modification of the ga2_76cycle_sr_v7.xml sequencing program . methods for rna - seq data filtering and processing were essentially as those described previously . nearly 24.30 million reads were generated from the four replicates ranging from 4.33 million in rep . 2 . using tophat , ~ 88.590.2% of the reads were mapped to the reference b73 genome ( release 5b.60 ) ( table 1 ) . using bedtools , 31,130 of the 110,028 genes in the working gene set ( wgs ) were detected in at least 2 replicates with at least one raw read count . inter - sample differences in library size were eliminated by total count ( tc ) normalization method . read counts for all the genes in wgs were divided by the total counts of mapped reads ( or library size ) associated with their sample and multiplied by the mean total count across all the samples of the dataset to calculate the normalized read counts and deposited in geo . similarity of expression profiles between the replicates was determined by a pearson correlation coefficient ( pcc ) analysis . the four replicates showed high correlation with each other , with pcc scores ranging from 0.95 to 0.96 ( fig . 2 ) . together , the evidence suggests that our lcm - generated rna - seq data is highly reproducible .

endosperm is a product of double fertilization , and provides nutrients and signals to the embryo during seed development in flowering plants . early stages of endosperm development are critical for the development of its storage capacity through synthesis and accumulation of starch and storage proteins . here we report on the isolation and sequencing of mrnas from the central portion of the starchy endosperm of zea mays ( maize ) b73 at 6 days after pollination . we detected a high level of correlation among the four biological replicates of rnas isolated using laser - capture microdissection of the cell type . because the assayed developmental stage precedes the synthesis and accumulation of the major storage proteins and starch in the endosperm , our dataset likely include mrnas for genes that are involved in control and establishment of these storage programs . the mrna - seq data has been deposited in gene expression omnibus ( accession number gse58504 ) .

a 28-year - old male patient visited our dental office for exodontia of the third molars . during the inferior left alveolar nerve block , an anesthetic needle fracture occurred due to patient movement or a possible manufacturing error . the left mandibular third molar was removed and radiographic monitoring was carried out ( fig . , the authors opted to take a panoramic radiograph because it provides an image of both arches , including the areas of the jaw and soft tissues of the maxilla and mandible . panoramic radiographs obtained at intervals of 15 days demonstrated that the metallic object had changed its location . 2 ) . to accurately localize the needle , the following radiographs were obtained : 1 ) panoramic , 2 ) lateral cephalography , and 3 ) multislice ct with 64 detectors ( figs . 3 and 4 ) . it was found that the fractured needle was partially located in the medial pterygoid muscle and partially in the parapharyngeal space . loss of the anatomical fat structure and the formation of granulomatous tissue was also observed ( figs . 5 , 6 , 7 ) . the patient was referred to a head and neck surgery specialist for evaluation of the necessity of removing the object , but because of its location , monthly radiographic monitoring was chosen . three months after the incident , the fractured fragment stabilized , obviating the need for surgical removal ( fig . quarterly radiographic monitoring was advised , and 2 years later , the patient did not present with any symptoms ( fig . however , their occurrence is frequently associated with the inferior alveolar nerve block , and the fragment usually settles in the region near the pterygomandibular raphe,14567810 as observed in this case . therefore , complementary exams are necessary , as panoramic radiographs and specific radiographic techniques , including ct , are required to assess its location accurately.24567910 some authors have reported the use of metal detectors , magnetic resonance imaging , and ultrasound for the accurate localization of the object.36710 in this case , panoramic radiographs , teleradiography , and the waters view were performed to verify the location of the fractured fragment , as well as ct to evaluate its relationship with adjacent structures . bedrock et al . in 19992 described controversies regarding the removal of the fractured needle fragment , and argued that due to the absence of symptoms , the removal of the object is not generally necessary , as removal appears to be favorable only for the patient 's psychological state with the goal of calming the patient . the possibility of migration of the fractured fragment is insignificant , although its removal might be recommended because of the possibility that it could cause injuries in adjacent tissues , especially when it is located near important structures.13610 in association with a needle fracture , the patient can present with an infection , inflammation , pain , trismus , difficulty in swallowing , and other symptoms indicating that surgical removal may be necessary.2389 in this case , fragment removal was initially recommended because of its migration in the first 3 months and the patient 's uneasiness . after it stabilized , its location was verified to be in the parapharyngeal space ( fig . 5 ) , the patient was reassured , and with the consensus of the team of professionals involved , it was decided to carry out radiographic monitoring of the fragment . dentists should develop the practice of explaining anesthetic block and possible complications during the procedure to their patients , requesting their cooperation in addition to a detailed inspection of the needle before the procedure . if defective needles are noticed , they should be discarded.123 it is very important that professionals know the proper technique for the nerve block of each nerve and use specific and appropriate needles , since the same type of needle should not be used for each nerve.234 this can prevent difficulties during dental local anesthesia . it is necessary for dental surgeons to adopt procedures to prevent needle fractures and to perform a detailed inspection before anesthetic procedures . if a fracture occurs , it is ideal to inform the patient while trying to sedate him / her and to obtain the patient 's cooperation in carrying out the necessary procedures for his / her well - being , such as radiographic monitoring or in some cases , surgery to remove the fragment .

some complications can arise with the usage of local anesthesia for dental procedures , including the fracture of needles in the patient . this is a rare incident , usually caused by the patient 's sudden movements during anesthetic block . its complications are not common , but can include pain , trismus , inflammation in the region , difficulty in swallowing , and migration of the object , which is the least common but has the ability to cause more serious damage to the patient . this report describes a case in which , after the fracture of the anesthetic needle used during alveolar nerve block for exodontia of the left mandibular third molar , the fragment moved significantly in the first 2 months , before stabilizing after the third month of radiographic monitoring .

spontaneous intracranial hypotension ( sih ) is an uncommon cause of sudden and persistent headache and patients typically present with postural or exertional headaches that can be temporarily relieved by lying in a supine or recumbent position . associated symptoms are common , among which there are cranial nerve palsies , frequently resulting in ophthalmoplegia [ 38 ] , especially of the abducens nerve . clinical symptoms are usually accompanied by magnetic resonance imaging ( mri ) findings related to cerebrospinal fluid ( csf ) depletion including subdural fluid collections , enhancement of the pachymeninges , engorgement of venous structures , pituitary hyperemia and sagging of the brain . the present case , reporting the association with an isolated third nerve palsy , shows that a deep knowledge of sih clinical presentation may avoid misdiagnoses . we report a case of a 21-year - old man , admitted to our hospital , for a sudden onset of a severe holocranial pain and diplopia . the headache was continuous , and changing position , particularly orthostatism , caused worsening of symptoms which conversely were relieved by lying flat . the patient did not complain about phonophobia , vertigo or tinnitus and vomiting , but reported photophobia and nausea . neurological examination was normal other than for cranial nerves evaluation that showed diplopia due to mild restricted adduction of the left eye toward right and ipsilateral partial ptosis . pupils were equal and reactive to light ( both directly and consensually ) and accommodation . diplopia disappeared within 2 h. saccadic eye movements and ocular examination were normal with visual fields full to confrontation . the patient did not experience head or neck trauma and fever or rashes were not present . to exclude a possible subarachnoid hemorrhage ( sah ) , the patient underwent a brain ct scan which was negative for hemorrhage . to rule out a diffuse inflammatory disease or neoplastic processes , routine blood , rheumatic and autoimmune tests , paraneoplastic markers , serum levels of angiotensin - converting enzyme , thoracic radiography and abdominal ultrasound , brain and spine magnetic resonance imaging ( mri ) without contrast revealed a mild descent of the brainstem with mild cerebellar tonsillar herniation and flattening of pontine surface , dilated sagittal sinus and enlargement of the pituitary gland ( fig . 1a ) , associated with a subdural hematoma , surrounding bilaterally the fronto - parietal and temporo - polar regions . another subdural fluid collection was present in the anterior section of the spinal cord ( at c5d1 level ) with a consequent posterior dislocation . in addition , symmetrically dilated vascular structures , with abnormal epidural venous engorgement , were again seen anterior to the cervical cord without cord compression , from c4 level up to atlanto - occipital junction ( fig . after gadolinium , mri showed diffuse pachymeningeal enhancement in supratentorial and infratentorial regions ( fig . 1a sagittal t1-weighted brain mri , b sagittal t2-weighted spinal mri , c coronal t1-weighted brain mri post - gadolinium contrast administration , d mr myelogram of upper and lower spinal subdural space a sagittal t1-weighted brain mri , b sagittal t2-weighted spinal mri , c coronal t1-weighted brain mri post - gadolinium contrast administration , d mr myelogram of upper and lower spinal subdural space mr myelography showed no signs of spreading cerebrospinal fluid ( csf ) at the level of the cervical and dorsal root pockets ( fig . according to the international headache society ( ihs ) ichd - ii criteria , the mri findings and the clinical history were consistent with a diagnosis of headache attributed to low csf pressure . although ichd - ii comments suggest that dural puncture should be avoided in patients with positive mri signs , such as meningeal enhancement with contrast we performed csf analysis to corroborate our diagnostic hypothesis . csf opening pressure was of 5 cmh2o and a lymphocytic pleocytosis ( 20 cells / ml ) was detected . initially , the case was managed by bed rest , hydration and steroidal therapy , but after 3 weeks of conservative treatment with limited benefit on the orthostatic headache , the sih syndrome was successfully managed with the application of lumbar epidural blood patch ( ebp ) using approximately 28 ml of autologous blood , resulting in immediate relief of clinical symptoms . after 6 months , in contrast , mri findings were substantially unmodified , showing only a mild reduction of brain sagging , with persistent subdural fluid collection in the same regions , epidural venous engorgement and diffuse meningeal enhancement in supratentorial and infratentorial regions ( images not shown ) . to our knowledge , this is the first report of a transient and isolated third nerve palsy as the presenting sign of sih . csf exerts a necessary buoyant force on the cranial contents , suspending the brain and cranial nerves and protecting them from downward traction . when the csf volume decreases , the burden on the vascular and/or dural pain sensitive structures , subjected to traction and distortion , may cause the headache . a relatively low csf volume may result by either post - traumatic or spontaneous dural laceration . . the second one can be frequently due to micro ruptures of the dura occurring at weak points along the spinal root sleeves ( especially in the presence of connective tissue disorder ) resulting in a syndrome known as sih . orthostatic , diffuse or dull headache that worsens within 15 min after sitting or standing and that can be temporarily relieved by lying in a supine or recumbent position , is the main manifestation of sih . other common signs and symptoms include tinnitus , hypoacusia , vertigo , photophobia , nausea and neck stiffness . a variety of cranial nerve palsies is frequently associated with sih ( 3035% ) resulting in ophthalmoplegia and visual disturbances . abducens nerve palsy is the most common occurring in about 80% of reported patients with sih - related ophthalmoplegia . although less common , both unilateral and bilateral paresis of the third and fourth cranial nerves might occur with sih . ferrante et al . reported a woman with a paresis of the right third combined to sixth cranial nerves . moreover , warner described an isolated , partial third cranial nerve palsy due to sih , although eighth cranial nerve involvement was present in the form of intermittent whistle in the ear . pathophysiology of cranial nerve paresis in sih is not completely understood and potential causes include traction on cranial nerves due to downward displacement of cerebral structures and cranial nerve compression or might be due to secondary brainstem compression and transitory ischemia in the acute phase of sih . we believe that a deep knowledge of clinical presentation of sih may avoid misdiagnoses , especially with sah . indeed , the presence of third cranial nerve palsy ( alone or in association with other cranial nerves involvement ) with sudden onset orthostatic headache should prompt a possible diagnosis of sih which must be considered only after sah has been carefully ruled out . usually after ebp procedure , clinical symptoms recede together with the disappearance of mri features , however , in line with the present case , a clinical radiological dissociation in which clinical syndrome improved , while mri features did not , has been already reported .

spontaneous intracranial hypotension is an uncommon cause of sudden and persistent headache : associated symptoms are common , among which there are cranial nerve palsies , especially of the abducens nerve . we report a case of a 21-year - old man with a transient and isolated third nerve palsy due to spontaneous intracranial hypotension . to our knowledge , there are only few reports in the literature of such association .

allergic reactions to antibiotics are associated with increased rates of morbidity and mortality , as well as increased medical costs ( 1 - 3 ) . although many cases of anaphylaxis to penicillin and cephalosporin have been reported , anaphylaxis to tetracycline ( 4 ) is less common . tetracycline is a broad - spectrum polyketide antibiotic produced by the streptomyces genus of actinobacteria , which has been indicated in the treatment of many types of bacterial infections . although the use of tetracycline has decreased , minocycline , a semisynthetic , second - generation tetracycline derivative , is still used worldwide in the treatment of acne . although minocycline is relatively safe , it has been reported to have adverse effects in 13.6% of treated patients ( 5 ) . common adverse effects of minocycline include gastrointestinal disturbances ( nausea and vomiting ) , vestibular disturbance ( dizziness ) , and cutaneous symptoms ( hyperpigmentation of skin and mucous membranes , pruritis , urticaria , and photosensitive rash ) . serious but rare adverse effects of minocycline include pneumonitis , hepatitis , pancreatitis , nephritis , polyarthritis , serum sickness - like reaction , drug - induced lupus erythematosus - like eruption , and hypersensitivity syndrome ( 6 , 7 ) . to our knowledge , only one case of anaphylaxis to oral minocycline has been reported to date ( 8) . here we report a case of anaphylaxis to oral minocycline , confirmed by an oral challenge test . a 56-yr - old woman visited our hospital after experiencing three episodes of recurrent anaphylaxis during the previous year . in every episode , she presented with urticaria , angioedema , dyspnea , and hypotension and visited the emergency department . in the first episode , she had generalized skin rash with an itching sensation , periorbital and perioral swelling , dyspnea , and dizziness . her blood pressure decreased to 73/46 mmhg and she was diagnosed with and treated for anaphylaxis . at that time , the precipitating cause was not evaluated . during the second and third episodes , however , she noticed that her symptoms developed within a few minutes after taking bepotastine besilate , methylprednisolone , and minocycline for acne . her initial laboratory tests at the first visit to our hospital revealed no apparent abnormalities except for mild leukocytosis ( 16,700/l ) . we performed an oral minocycline challenge test to confirm the causative drug of her recurrent anaphylaxis . the patient took a half capsule of minocin [ 50 mg] ( sk chemical life science , seoul , korea ) . within 4 min , she began to feel an itching and burning sensation in her face and forearms , followed by the development of generalized wheal . , we heard a wheezing sound in her whole lung field and an abrupt fall in blood pressure , to 70/50 mmhg . at that time , her pulse rate was 55 beats / min , her respiratory rate was 24/min , and her spo2 was 93% . she was administered two 0.3 cc doses of epinephrine intramuscularly 5-min apart , along with intravenous chlorpheniramine ( h1-antagonist ) 4 mg , famotidine ( h2-antagonist ) 20 mg and hydrocortisone 250 mg . thirty minutes later , however , she again developed hypotension ( 80/40 mmhg ) , for which she was treated with additional intramuscular epinephrine . 2 ) , for which she was managed with the same regimen . on the following day , the patient had fully recovered without apparent complications and discharged . she was educated about avoidance of tetracycline and given a ' drug - alert card ' . since stopping minocycline , it is a systemic reaction mediated by vasoactive amines released from mast cells and basophils sensitized by immunoglobulin e ( ige ) . immediate hypersensitivity reactions commonly involve at least two of the following major organ systems : cutaneous ( generalized hives , pruritus , swollen lips - tongue - uvula ) , cardiovascular ( hypotension ) , respiratory ( dyspnea due to laryngeal edema , bronchospasm , stridor ) , and gastrointestinal ( vomiting , diarrhea , abdominal cramps ) ( 9 ) . individuals differ in the time of appearance of symptoms and signs , but the hallmark of an anaphylactic reaction is the onset of some manifestations within seconds to minutes after introduction of the antigen . several reports have described anaphylactoid reactions to tetracycline ( 4 ) , doxycycline ( 10 ) , and oral minocycline ( 8) . in the latter report , a 27-yr - old woman diagnosed with salpingitis and treated with 100-mg oral minocycline developed generalized wheal and erythema , dyspnea , and hypotension within 30 min after taking minocycline . the symptoms cleared in about 4 hr , and a scratch test performed on her forearm with minocycline resulted in a positive reaction . in that case , however , an oral challenge test was not performed . these types of hypersensitivity reactions are thought to be caused by a reactive metabolite of a drug , which can bind to tissue macromolecules , causing cell damage , or can act as a hapten , eliciting an immune response . although it is not known whether minocycline produces a reactive metabolite , it may generate an iminoquinone derivative . neither tetracycline nor doxycycline contains the amino acid side chain that has the potential to form a reactive intermediate , and therefore the hypersensitivity associated with minocycline may be specific to this antibiotic . further evaluation of the pathogenesis associated with minocycline is required ( 11 ) . to our knowledge , this is the first case report to describe anaphylaxis to oral minocycline in korea . the immediate and typical anaphylactic symptoms and signs , the dramatic triphasic reactions after oral challenge , and the previous history of anaphylaxis after exposure to minocycline , indicate that anaphylaxis in this patient was mediated by type i hypersensitivity reactions . to our knowledge , this report is also the first to utilize an oral challenge test , the standard method for the diagnosis of drug allergies , to confirm that minocycline was the culprit drug . since minocycline remains a useful drug for the treatment of acne vulgaris , physicians should keep in mind that minocycline can induce anaphylaxis . in addition , this drug should be considered a causative agent in patients developing allergic reactions .

minocycline is a semisynthetic tetracycline derivative that is often used in the treatment of acne vulgaris . to date , there has been only one case report of anaphylaxis to minocycline . we report here a case of anaphylaxis to oral minocycline . a 56-yr - old woman visited our hospital after three episodes of recurrent anaphylaxis . we performed an oral challenge test , the standard method for diagnosing drug allergies , with minocycline , one of the drugs she had taken previously . she developed urticaria , angioedema , nausea , vomiting , hypotension , and dyspnea within 4 min and was treated with intramuscular epinephrine , intravenous antihistamine and systemic corticosteroid . however , she presented similar symptoms at 50 min and at 110 min . in prescribing oral minocycline , physicians should consider the possibility of serious adverse reactions , such as anaphylaxis .

progressive nodular histiocytosis ( pnh ) is a rare normolipemic macrophage disorder first described by taunton et al . , in 1978 . it belongs to a subgroup of non - langerhans cell histiocytosis ( lchs ) , the juvenile xanthogranuloma ( jxg ) family , which includes xanthoma disseminatum ( xd ) , benign cephalic histiocytosis ( bch ) , multiple adult xanthogranuloma ( maxg ) and generalized eruptive histiocytosis ( geh ) . according to weitzman and jaffee , they present positive reaction to factor xiiia , cd68 , cd14 and negative reactions to cd1a and s-100 proteins . these disorders are difficult to categorize , primarily because of the diverse pathological findings and due to overlapping clinical and microscopic features . a 30-year - old gentleman presented to us with complaints of asymptomatic nodular skin lesions all over the body of 10 years duration . at the age of 20 years , he started developing few small skin - colored lesions on the bilateral peri - orbital areas . these lesions continued to increase in size over time and few lesions became pedunculated especially over the calves . along with the appearance of the cutaneous lesions , he also noticed a gradually progressive , bilateral painless loss of vision . he did not complain of itching , watering , photophobia or discharge from the eyes . he was diagnosed to have eale 's disease and underwent laser photocoagulation for the same . he developed premature cataracts of the eyes and was treated with phacoemulsification and posterior capsular intraocular lens implantation in both eyes . prior to presentation to us , he underwent intra - lesional steroid therapy using triamcinolone acetonide in the peri - orbital lesions with good response seen in smaller lesions but no effect on the larger lesions . the lesions at other sites were removed using various modalities like cryotherapy , radiosurgery and surgical excision of the larger , troublesome lesions . cutaneous examination revealed multiple , approximately 30 to 40 , yellowish to skin - colored , firm , non - tender papules and nodules ranging in diameter from 5 to 40 mm , over the face [ figure 1 ] , trunk , extremities , elbows , hands , palms and soles [ figure 2 ] in decreasing order of frequency . these lesions were located almost symmetrically over the peri - orbital area , mostly over the outer canthi , but also over the lower eyelids , leading to ectropion . the smaller lesions on the face were mounted on the skin , while the larger lesions on the calf area were pedunculated . papular and nodular lesions on the face and eyelid large nodules seen over the elbows , hand and sole visual acuity at the time of presentation was 6/18(r ) and 6/60(l ) . the clinical differential diagnoses considered at the time of presentation to us were multiple eruptive dermatofibromas , xanthoma disseminatum and pnh . blood examination revealed hemoglobin of 10.5 gm / dl with a microcytic hypochromic blood picture , total leukocyte count of 7200/mm and a normal differential leukocyte count . fasting lipid profile , renal , total serum proteins and albumin were normal with no reversal of the a : g ratio . a cellular tumor was seen centered in the dermis , composed of sheets of oval to spindle - shaped cells arranged haphazardly , in short fascicles and in a storiform pattern at places [ figures 3a - c ] . the spindle cells had oval to elongated benign looking nuclei with vesicular chromatin and small nucleoli . on immunohistochemistry the cells were positive for cd68 [ figure 4 ] but negative for s-100 , cd34 , cd21 , cd35 and hmb45 supporting a diagnosis of spindle cell histiocytic tumor . dab chromogen 100 based on the history , clinical examination and histopathological correlation , his dermatological disorder was diagnosed as pnh . he was started on tab imatinib 400 mg od and some of the lesions developed encrustation but it was stopped after 5 months as he continued to develop new lesions while on this drug . second - line treatment with pazopanib ( 800 mg od ) was started after stopping tab imatinib , in lines of soft tissue sarcoma . based at the age of the onset of the lesions , areas of the body involved , progression of disease , lab abnormality ( serum lipids ) , it is possible to differentiate pnh from other forms in the jxg family . benign cephalic histiocytosis and jxg usually occur in early childhood , but jxg has also been reported in adults and the lesions have a tendency to resolve spontaneously over a period of time . multiple adult xanthogranuloma is another important differential diagnosis of pnh which has similar features to jxg but occurs in adults . xanthoma disseminatum was ruled out based on the absence of mucosal and systemic involvement and with normal lipid profile as it usually presents with hyperchylomicronemia and hypertriglyceridemia and diabetes insipidus . since the accurate diagnosis of a specific disorder within the jxg subgroup is quite difficult , we made the diagnosis of pnh based on the history , presence of the typical skin lesions , progressive course and histopathological demonstration of spindle cells that were positive on immunohistochemistry for histiocyte marker , cd68 , but negative for s-100 . non - langerhan 's cell histiocytosis disorders are difficult to diagnose and our case presents another clinical expression of this variable and confusing disorder . to our knowledge this is the first case report of progressive nodular histiocytosis associated with eale 's disease .

progressive nodular histiocytosis ( pnh ) is a rare normolipemic macrophage disorder and belongs to a subgroup of non - langerhans cell histiocytosis ( lchs ) which is characterized by a progressive course with no sign of spontaneous resolution but without systemic involvement . we report a 30-year - old gentleman who presented with skin lesions all over the body associated with gradual bilateral painless loss of vision . on examination , approximately 30 to 40 , skin - colored , firm , non - tender papules and nodules were noted over the body especially on the face and trunk . a skin biopsy revealed a cellular tumor in the dermis composed of oval to spindle - shaped cells , positive for cd68 but negative for s-100 , cd34 , cd21 , cd35 and hmb45 , supporting a diagnosis of spindle cell histiocytic tumor . ophthalmic examination revealed a generalized arteriolar attenuation in both eyes . he received tab imatinib 400 mg od for 5 months followed by tab pazopanib 800 mg od for 4 months and both the drugs were stopped due to lack of any response in the skin lesions . we report this case due to its rarity , characteristic clinical presentation , and its association with eale 's disease . primary treatment remains surgical excision of bothersome lesions and optimal systemic treatment is still unknown .

rapidly progressive renal failure ( rprf ) warrants rapid diagnosis so that treatment options can be readily instituted . most often , in association with other clinical and laboratory findings , the diagnosis will be a rapidly progressive glomerulonephritis . a 41-year - old female was referred to our centre for rapidly progressive renal failure in january 2009 . her symptoms began in september 2008 with dysuria , diagnosed as a urinary - tract infection treated with a short course of ciprofloxacin . after 1 month , she had recurrence of her dysuria and further investigations done at that time revealed a creatinine of 205 mol / l ; haemoglobin 90 g / l ; urine blood 1 + , protein 1 + and a urine culture positive for klebsiella pneumonia . mol / l in may 2007 which increased to 393 mol / l by december 2008 . there was no history of arthralgias , skin rash , oral ulcers , hypersensitivity , sicca , raynaud 's phenomenon , jaundice , use of herbal medicines or non - steroidal anti - inflammatory medications . her blood pressure was 140/80 mmhg and the remainder of the physical examination was non - contributory . urine analysis showed 4 + blood ; 2 + protein ; haemgranular and rbc casts ; 24-h urine protein 1 g / day ; haemoglobin / l ( 0.160.38 ) ; ch50 1.9 ( 60145 ) ; positive p - anca at 1:80 ; positive ra factor 46 iu / ml ( normal 019 ) , ana > 1:640 . the remainder of the serologic work up including , anti - dsdna , anti - smith , le cells , anti - sm / rnp , anti - jo 1 , anti - scl 70 , anti - ssb ( la ) , anti - ssa ( ro ) , hbsag , anti - hcv , anti - hiv and serum protein electrophoresis , was negative . a renal ultrasound demonstrated normal sized kidneys with increased echogenicity and no hydronephrosis . on renal biopsy , one of six glomeruli was sclerosed and few demonstrated collapsed capillary loops with increased mesangial matrix , but neither necrosis , nor spikes on silver stain were revealed . within the interstitium , there were a large number of inflammatory cells predominantly composed of plasma cells , lymphocytes , macrophages and occasional eosinophils ( figure 1 ) . immunofluorescence ( if ) staining showed tubular basement membrane ( tbm ) deposits of igg , igm and c3 , with deposition of immunoglobulin around the bowman 's capsule ( figure 2 online supplementary material ) . electron microscopy ( em ) revealed abundant electron - dense deposits in tbm ( figure 3 ) . the interstitium contains a large number of inflammatory cells : plasma cells , lymphocytes , macrophages and occasional eosinophils ( haematoxylin and eosin stain ) . immunostaining shows a heavy deposition of igg along the tubular basement membrane ( immunofluorescence , ftic - labelled anti - human igg ) . the tubular basement membrane contains abundant electron - dense deposits ( arrows ) , consistent with immune complexes . the patient was treated with 1 g iv methylprednisone for three consecutive days followed by oral prednisone 1 mg / kg ( 60 mg ) . she was discharged and followed up in the ambulatory clinic . at 10 weeks post - therapy , her last creatinine was 156 mol / l and the prednisone was tapered to 15 mg daily . this patient presented with rprf with associated features of active urine sediment , strongly positive ana and low complements . the differential diagnosis in this setting included lupus nephritis , membranoproliferative glomerulonephritis ( mpgn ) , cryoglobulinaemia and post - infectious glomerulonephritis . the positive p - anca adds consideration to a diagnosis of vasculitis . all of the above diagnoses have significant glomerular pathology with minimal or no tubulointerstitial nephritis . we diagnosed this case as hypocomplementaemic immune - complex tubulointerstitial nephritis , a very rare cause of tubulointerstitial nephritis . reported the largest series , with eight cases of tubulointerstitial nephritis with tubulointerstitial immune deposits in adults with hypocomplementaemia with no evidence of sle or sjogren 's disease . hypocomplementemia , tbm immune complexes and glomerular sparing were consistent findings in all these cases [ 14 ] . prednisone was used in 10/11 cases either alone ( 7/11 ) or in combination with other immunosuppressive agents [ 25 ] . the duration of treatment and follow - up ranged from 3 to 12 and 3 to 36 months , respectively . renal function as reflected by serum creatinine improved in 7/11 and deteriorated in 2/11 cases . potential response mori et al . reported such a case and reviewed the literature of only 10 reported cases . all of them fulfilled american rheumatology association ( ara ) criteria for the diagnosis of sle . sle as the underlying disease was excluded in our case presented here , as it did not meet ara criteria . negative serology for anti - ro and anti - la , low complements and paucity of clinical symptoms ruled out ss in this case . tinu ( tubulointerstitial nephritis and uveitis ) was ruled out due to the absence of ocular symptoms , low complements and the presence of immune - complex tbm deposits . anca - associated vasculitis presenting as isolated tubulointerstitial nephritis has been reported in the literature . however , the presence of low complements and positive if excluded this possibility . a similar argument discounted any possibility of drug ( ciprofloxacin)-induced acute interstitial nephritis ( ain ) . in summary the weight of evidence suggests that it is immunologically mediated predominant tubulointerstitial nephritis with sparing of glomeruli . it is associated with multiple autoantibodies including ana , anca and ra . however , at present , this appears to be a heterogeneous disorder of unknown specific aetiology .

we report a rare cause of rapidly progressive renal failure associated with low complement , positive ana but negative anti ds - dna . a renal biopsy demonstrated tubulointerstitial nephritis with positive immunoglobulin staining involving the interstitium and tubular basement membrane but glomerular sparing . a review of the literature and differential diagnosis are discussed .

in recent years , drug - eluting stents ( dess ) have been shown to dramatically reduce the rate of restenosis and the need for repeat revascularization.1 - 3 ) despite these promising results , late stent thrombosis seems to occur more frequently with dess and seems to be closely associated with the discontinuation of dual aspirin / thienopyridine derivative ( usually clopidogrel ) antiplatelet therapy.1 - 4 ) we report a case of late des thrombosis after discontinuation of dual antiplatelet therapy . a 49-year - old man was transferred to the emergency department complaining of squeezing chest pain that had increased in severity over the past four days . five years prior to presentation , he had undergone percutaneous coronary intervention ( pci ) for acute myocardial infarction ( mi ) , along with implantation of a bare metal stent ( bms ) ( 3.528 mm arthos inert stent , amg , korea ) in the left anterior descending artery ( lad ) ( fig . 1 ) . six months later , he developed angina and underwent balloon angioplasty for in - stent restenosis ( fig . two years after that , he had a paclitaxel - eluting stent ( 2.7532 mm taxus stent , boston scientific , usa ) placed in the right coronary artery ( rca ) after suffering another acute mi ( fig . the patient was treated with dual antiplatelet therapy : aspirin ( 100 mg daily ) and clopidogrel ( 75 mg daily ) . sixteen days after discontinuing medication , he developed chest pain and presented to the emergency department . an electrocardiogram ( ecg ) performed at that time showed new st - segment depression and t - wave inversion in leads ii , iii , and avf ( fig . cardiac enzymes were also elevated ( creatine kinase - mb 9.9 u / l , troponin i 7.73 ng / ml , troponin t 2.64 ng / ml ) . the patient underwent emergency coronary angiography , which revealed total occlusion of the des in the proximal rca due to very late stent thrombosis with grade ii collateral flow ( fig . 5c ) . after receiving an intravenous glycoprotein iib / iiia receptor blocker ( abciximab ) , the patient underwent repeat balloon angioplasty and thrombus aspiration secondary to recurrent , immediate thrombus formation and coronary occlusion ( fig . 6a ) . we report this case to draw attention to the phenomenon of very late stent thrombosis after des implantation , which might be associated with serious clinical implications after the discontinuation of dual antiplatelet therapy . it is possible that dess are associated with substantially higher rates of thrombosis when compared with bmss . it may occur in the acute ( < 1 day ) , subacute ( < 30 days ) , late ( < 1 year ) , or very late ( > 1 year ) periods and may result in serious complications such as mi and death.5 ) very late stent thrombosis ( vlst ) is defined as a stent thrombosis event that occurs beyond one year . the risk of vlst was analyzed using trial level data in a meta - analysis of 14 randomized trials in which 6,675 patients had been randomly assigned to pci with des or bms.6 ) the incidences of vlst were 5.0 events per 1,000 patients in the des group and 0 events per 1,000 patients in the bms group ( relative risk=5.0 , 95% confidence interval 1.3 - 19.5 ) . in another report , dess were reported to have an approximately 0.3 to 0.5 percent greater risk of vlst compared with bmss.7 ) several factors were shown to be associated with st , including older age , black race , diabetes mellitus , bifurcation lesion , instent restenosis lesion , post - procedure acute renal failure , and lack of clopidogrel therapy.8 ) discontinuation of antiplatelet therapy , as an independent predictor of stent thrombosis , even several years after des implantation , increases the risk of late stent thrombosis . this finding suggests that clopidogrel compliance may minimize the incidence of vlst after des implantation . current recommendations suggest extending dual antiplatelet therapy beyond one year in patients with low bleeding risk . antiplatelet treatment should be continued even if there is an increased risk of minor bleeding complications so that life - threatening complications , such as acute mi , are avoided . patients with previously implanted dess who are currently taking dual antiplatelet therapy are at high risk for developing stent thrombosis when a situation arises that requires cessation or interruption of dual platelet inhibition.9)10 ) the optimal duration of antiplatelet therapy in patients with coronary artery stents still remains to be determined . further large - scale studies are needed to determine the optimal combination and duration of antiplatelet therapy that should be used to prevent these serious thrombotic events .

drug - eluting stents ( dess ) are the treatment of choice for obstructive coronary artery disease when percutaneous intervention is feasible . however , late stent thrombosis seems to occur more frequently with dess and is closely associated with the discontinuation of dual antiplatelet therapy . we report a case of very late stent thrombosis after discontinuation of dual antiplatelet therapy . the patient suffered from acute myocardial infarction ( mi ) and underwent bare metal stent ( bms ) implantation in the left anterior descending artery ( lad ) five years prior to presentation . three years after bms implantation , he presented again with acute mi and had a des implanted in the right coronary artery ( rca ) . he ran out of his medication , but failed to refill his prescription . sixteen days after discontinuing medication , he experienced an episode of chest pain and was taken to the cardiac catheterization laboratory , where he was found to have thrombosis in the des , but no thrombosis in the bms . it is possible that dess are more vulnerable to late thrombosis than are bmss , supporting the use of prolonged dual antiplatelet therapy in patients treated with dess . the patient was successfully treated with balloon angioplasty and thrombus aspiration without complications .

mycobacterium kansasii is a non - tuberculosis mycobacterium that is more prevalent in southeastern and southern coastal states and the central plains . chronic diseases were common among hiv - negative persons ; however , 40.3% had no predisposing medical condition . mycobacterium kansasii usually presents as lung disease in which the most common radiologic presentation is cavitation of the lung parenchyma [ 35 ] . involvement of the musculoskeletal system occurs in approximately 5 - 10% of patients with atypical mycobacterial infection . the patient was a 14-year - old girl who presented with thoraco - lumbar pain over a one - year period . the thoraco - lumbar mri revealed a collapsed t8 vertebral body and discitis of t8 - 9 . a hemogram revealed hemoglobin of 11 g / dl , total leukocyte count of 11.3 10 ( 80% neutrophils ) , and normal platelet count . blood chemistry and urine analysis were normal . levels of immunoglobulin were in the normal range and the hiv test was negative . a ct scan of the chest showed a cavitary lesion in the left lung upper lobe ( fig . para - vertebral fluid collection was sampled and a biopsy was obtained from t8 ( fig . collapse of t8 ( upper vertical arrow ) and extending abscess ( horizontal arrow ) and osteopenia in t9 ( lower vertical arrow ) infiltration of inflammatory cells in vertebrae pcr of purulent material collection in the paravertebral region for mycobacterium tuberculosis was negative . one month later , pcr of culture of fluid collection of this region was positive for m. kansasii . her thoracolumbar pain resolved after about two months , esr decreased and the pulmonary lesion disappeared . we found only two reports of m. kansasii spondylitis in an immunocompetent patient [ 7 , 8 ] . in patients with mycobacterial spondylitis , local tenderness , pain , and limitation of spinal mobility are the presenting symptoms , whereas constitutional symptoms such as fever , malaise , and weight loss may also occur . the radiologic manifestations of spine osteomyelitis are involvement of one or several contiguous vertebral bodies ; destruction of the intervening disks ; absence of reactive sclerosis ; and formation of soft - tissue abscesses . the diagnostic criteria to support non - tuberculous mycobacterium ( ntm ) clinical infection as opposed to colonization of secretions include one of the following : positive culture results from at least two separate expectorated sputum samples ( regardless of afb smear result ) or positive culture result from at least one bronchial wash or lavage ( regardless of afb smear result ) or transbronchial or other lung biopsy with mycobacterial histopathological features ( granulomatous inflammation or afb ) and positive culture for ntm or biopsy showing mycobacterial histopathological features ( granulomatous inflammation or afb ) and one or more sputum or bronchial washings that are culture positive for ntm or a positive culture from pleural fluid or any other normally sterile extrapulmonary site . the american thoracic society recommendation for treatment of this infection consists of a three - drug combination consisting of isoniazid ( 300 mg daily ) , rifampin ( 600 mg daily ) , and ethambutol ( 15 mg / kg per day ) . our case was the third case of m. kansasii spondylitis in an immunocompetent patient in the reported cases that we could find in the literature . because of the rarity of the disease in immunocompetent patients her diagnosis was delayed , so we suggest including m. kansasii infection in the differential diagnosis of immunocompetent patients with clinical findings similar to tuberculosis .

we report the second case of mycobacterium kansasii spondylitis in a 14-year - old caucasian girl who presented with pain in the thoracolumbar region . there was collapsed t8 and discitis , and a cavitary lesion in new chest ct . biopsy taken from thoracic lesion t8 with ct guidance which had evidence of osteomyelitis and its smear were negative for acid fast bacilli . bronchoalveolar lavage ( bal ) was performed pcr was negative for acid fast bacilli . empirical treatment of tuberculosis started and her symptoms resolved . pcr of thoracic sampling revealed m. kansasii after one month . two months after treatment of m. kansasii , her thoracolumbar pain resolved and the cavitary lesion of the lung disappeared . in patients who are immunocompromised in the association of cavitary pulmonary lesion and infectious spondylitis , atypical mycobacteria may be on the list of common clinical diagnoses but not in immunocompetent patients such as our patient .