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a 36-year - old man with repeatedly paroxysmal headache was admitted to our hospital . the patient suffered a severe head trauma in a car accident 21 days prior to the accident . he was admitted to hospital with periorbital soft tissue swelling , ct scanning revealed a skull fracture in sss area . before he was transferred to our hospital , computerized tomographic angiography ( cta ) revealed vascular malformation and venous congestion of the sss , which was considered to be secondary to the trauma . left internal carotid artery angiogram revealed a davf supplied by the anterior falx artery ( figure 1a ) . left lateral ( figure 1b ) , anteroposterior ( figure 1c ) and right lateral ( figure 1d ) view of left external carotid artery ( eca ) angiogram showed the sss davf with bilateral cortical venous reflux . endovascular treatment for a patient with posttraumatic dural arteriovenous fistula ( davf ) of the superior sagittal sinus ( sss ) . a ) left internal carotid artery angiogram before embolization showing the davf supplied by the anterior falx artery ; b , c and d ) lateral ( b&d ) and anteroposterior ( c ) view of left external carotid artery ( eca ) angiogram before embolization showing the davf of sss with bilateral cortical venous reflux ; e ) unsubtracted image showing the scepter balloon catheter navigated close to the fistulous point for onyx injection ( arrow head indicating the balloon markers , arrow indicating the distal tip ) ; f ) unsubtracted image showing the onyx cast extending to the proximal draining vein after embolization ; g ) post - embolization angiogram of the left common carotid artery showing complete disappearance of the fistula . h ) post - embolization angiogram of the right eca showing complete disappearance of the fistula . ( 415 mm , microvention , tustin , california , usa ) was placed at the most distal segment of the left middle meningeal artery ( mma ) and contrast medium was injected into the balloon . unsubtracted image showed the scepter balloon catheter navigated close to the fistulous point for onyx injection ( figure 1e ) . in order to occlude any residual fistula , onyx-18 ( ev3 , irvine , california , usa ) was then injected through the dual lumen balloon catheter positioned in one branch of the mma . unsubtracted image showed the onyx cast extending to the proximal draining vein after embolization ( figure 1f ) . with the balloon inflated , a total of 1.2 ml of onyx-18 was delivered with thorough penetration into the malformation slowly , after which , angiogram of both the left common carotid artery and the right external carotid artery ( eca ) showed complete disappearance of the fistula ( figures 1g - h ) . the balloon was deflated by syringe suction without difficulty . at the end of the procedure , the catheter was removed under constant aspiration without any noticeable adherence to the onyx cast . discussion dural arteriovenous fistula account for 10%15% of intracranial arteriovenous malformations.6 davfs in sss are extremely rare . a small percentage of patients have a history of previous trauma , they have been well reported.7 the common characteristic of davfs happened when a patient suffered skull fraction , or other trauma , not long after a head injury with progressive symptoms such as , exophthalmos , swelling of the eyelids , bruit , and so forth . this scenario most likely accounts for the findings in our patient , and we believe that his davfs probably were secondary to the head injury rather than a congenital anomaly . so far , the first - line treatment for davfs is embolization by using transarterial , transvenous , or occasionally , combined approaches.1 - 8 this approach proved to be effective comparable with davf obliteration in preventing neurologic morbidity with lower levels of procedural risk . the use of onyx has been increasingly reported for the treatment of davfs.2 - 10 using reflux as a plug , operator creates a forward flow of onyx , which is called the plug and push technique . the dual lumen balloon microcatheter served a dual purpose : the onyx-18 injection was allowed through the dual lumen balloon catheter and a mechanical barrier was provided to prevent the onyx reflux at the same time . this technique is an option during extracranial embolic embolization in a few select cases . in this case , a patient with a post - traumatic davf of the sss was effectively treated . it is appropriate to select a dual lumen balloon microcatheter for complex dural arteriovenous fistulas . using a dual lumen balloon microcatheter helps prevent onyx reflux and improves its penetration during onyx embolization of davfs . thus , this is a feasible and effective alternative approach for the management of post - traumatic davfs .

dural arteriovenous fistula ( davfs ) induced by trauma in the superior sagittal sinus ( sss ) are rare and difficult to treat because of their unique midline location , multiplicity of arterial feeders , and critical venous drainage . we report a case of an endovascular treatment using dual lumen balloon microcatheter on a patient with post - traumatic sss davf . by the use of dual lumen scepter balloon microcatheter , proximal onyx reflux was prevented . in this case , complete embolization of the davfs was achieved and the outcome of the patient was fairly good .

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tear fluid drains through the lacrimal punctum into the lacrimal canaliculi ; it then flows through the lacrimal sac and the nasolacrimal duct before being absorbed by the nasal mucosa . when this pathway becomes blocked , the resulting condition is known as nasolacrimal duct obstruction , which may be either congenital or acquired . over the last 15 years , our clinic has experienced 64 cases of nasolacrimal duct obstruction , all of which were successfully treated with lacrimal passage irrigation . in this procedure , 0.4% xylocaine ophthalmic solution is injected using a 1-ml syringe tube and a lacrimal passage irrigation needle , with the tip of the needle being inserted into the inferior lacrimal punctum pointing toward the nose and facing slightly downward . to facilitate irrigation , the patient is asked to snort in order to create negative pressure in the nasal cavity . once the patient has registered the bitter taste of the xylocaine , irrigation should be performed repeatedly with physiological saline . in these 64 cases , we performed at least 10 irrigations for each patient , and for some , we performed 50 or more . of the 64 cases , recurrence only occurred in the 1 case that we present here . in this patient , the blockage resulted from rice grains ingested over a period of many years , forming an obstructive mass . the patient , a 39-year - old male who works as a surgeon and is the lead author of this report , experienced abnormal lacrimation of the left eye while at work . however , the condition recurred 5 times during the following 2 years despite repeated irrigation on each occasion . during the sixth treatment , after approximately 20 irrigations had been completed , an object was suddenly ejected into the rear of the patient 's nasal cavity . on histological examination , this object was found to be an agglomeration of rice grains ( fig . 1 , fig . 2 ) and was believed to have penetrated the nasolacrimal duct over a long period via reflux through hasner 's valve . this mass then became molded into a rectangular , cone - shaped plug , matching the shape of the nasolacrimal duct . since this successful treatment , there has been no further recurrence . approximately 1 month after completing treatment , the author underwent detailed examination of the paranasal sinuses by an otolaryngologist using fiberscopy . although it was not possible to view up to hasner 's valve , no other abnormalities were noted within the range visible by fiberscopy . to the best of our knowledge , there have been no other reported cases of nasolacrimal duct obstruction caused by the retention of foodstuffs . the patient had a habit of talking while eating , and we concluded that a rice grain had at some point penetrated the orifice of the nasolacrimal duct from the nasal cavity side via reflux through hasner 's valve . this led to incomplete valve closure and allowed the subsequent entry of a succession of rice grains , coalescing into a single mass and causing the blockage . in this case , the nasolacrimal duct appears to have been nearly completely obstructed by a lump of rice . during initial treatment , neither anesthetics nor physiological saline solution could pass through the duct , instead pouring out of the inferior punctum . in the latter phase of treatment , as the lump of rice started to slowly drop down into the paranasal sinus , water was gradually able to pass through the site despite meeting some resistance . when the lump of rice emerged from the paranasal sinus , this resistance to passing physiological saline solution disappeared and the patient suddenly felt physiological saline solution flow into the paranasal sinus . backflow caused the rice grain to initially enter the nasolacrimal duct , after which deposits built up over a number of years to cause an obstruction . we then started to treat this with flushing , which could not have caused the backflow considering the chronology , because there is no possibility that backflow occurred as a result of flushing . nasolacrimal duct obstruction is typically attributed to the accumulation of ocular discharge within the lacrimal passages ; however , such cases may be more common , although they may go unrecognized . in the treatment of nasolacrimal duct obstruction , a bougie is normally inserted immediately after an irrigation test . because this blind insertion can be dangerous however , we believe that successful treatment is possible using only repeated irrigation with physiological saline . this approach is safe and simple , allows flushing of the entire lacrimal passage , and is effective in preventing recurrences . to perform this procedure , the physician should wait for the water to completely pass through during the irrigation test . then , instead of immediately inserting a bougie , irrigation should be patiently repeated numerous times . therefore , the irrigation should be considered not only as a test but also as a form of washout therapy . the patient reported here first presented with persistent lacrimation . only after repeated unsuccessful treatments was it determined to be due to obstruction by impacted foodstuffs . we have never observed any other cases in which a lump of foodstuff was retained in the nasolacrimal duct and caused an obstruction , as in the present patient . in other cases , the discharge contained in tears adhered to the lumen of the nasolacrimal duct and caused either narrowing or a state close to obstruction . therefore , such cases could be treated using flushing performed repeatedly to gradually wash out the discharge . this is not a frequent cause of nasolacrimal obstruction , but it should perhaps be considered as a possible factor in similar cases of refractory nasolacrimal disorders .

here we report a case of nasolacrimal obstruction resulting from rice grains ingested over many years that had formed an obstructive mass . this is a hitherto unreported cause of nasolacrimal obstruction . we exclusively treated the obstruction safely with repeated irrigation with physiological saline . although there have been various reports on new techniques such as the blind insertion of a bougie and insertion of a silicon tube under endoscopic guidance , successful treatment of this condition is possible with an old - fashioned approach using the time - honored tools of a syringe and an irrigation needle , if utilized patiently and repeatedly . in japan , we call this learning new lessons from the past. this implies that newer techniques may not necessarily be superior and traditional techniques can offer advantages of safety and simplicity in treating this condition .

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improving quality of life and quality and management of care of clients with a schizophrenic disorder by the use of a comprehensive internet based disease management ( dm ) platform . presenting factors influencing the implementation of an internet based dm platform for clients with schizophrenia . two mental health care institutions in the south of the netherlands participate in the implementation of the digital instrument ( the dm platform ) . the implementation of the dm platform will be followed by a process and an effect evaluation . measurement instruments : in the effect evaluation , quality of life , number of symptoms of schizophrenia , quality of care , social functioning , self management , and insight in schizophrenia and its treatment will be measured . process evaluation : semi - structured interviews will be held with different ( formal primary and secondary , and informal ) caregivers , managers and clients of each institute . these interviews will be conducted before the implementation of the platform and one , two and three years later . in the digital platform the guideline for people with schizophrenia will be implemented and also information for clients and their home caregivers . when the platform is ready clients will be able to communicate digital with their caregivers in the mental health institute and with their home caregivers and gps . the effect evaluation starts in september 2009 , the process evaluation has started in may 2009 . we will have the first results in june 2009 ; they will be used to adjust the implementation and the development of the dm platform . in the meantime we can report about the building of the dm platform and what the expectations from the future users are . what are the factors that influence the process of implementation of a digital tool that provides help and care on a distance ? in what way does the digital tool ( care from a distance ) stimulate people who need chronically care to be more self - supporting ? in what way does the digital tool support professionals to be able to stimulate people with schizophrenia to be more self - supporting ?

purposeimproving quality of life and quality and management of care of clients with a schizophrenic disorder by the use of a comprehensive internet based disease management ( dm ) platform . presenting factors influencing the implementation of an internet based dm platform for clients with schizophrenia.contextthe project started in december 2008 . two mental health care institutions in the south of the netherlands participate in the implementation of the digital instrument ( the dm platform).the implementation of the dm platform will be followed by a process and an effect evaluation.data sourcemeasurement instruments : in the effect evaluation , quality of life , number of symptoms of schizophrenia , quality of care , social functioning , self management , and insight in schizophrenia and its treatment will be measured . process evaluation : semi - structured interviews will be held with different ( formal primary and secondary , and informal ) caregivers , managers and clients of each institute . these interviews will be conducted before the implementation of the platform and one , two and three years later.case descriptionin the digital platform the guideline for people with schizophrenia will be implemented and also information for clients and their home caregivers . when the platform is ready clients will be able to communicate digital with their caregivers in the mental health institute and with their home caregivers and gps . they also get information and feed - back about their illness and psycho education.(preliminary ) conclusionsthe effect evaluation starts in september 2009 , the process evaluation has started in may 2009 . we will have the first results in june 2009 ; they will be used to adjust the implementation and the development of the dm platform . in the meantime we can report about the building of the dm platform and what the expectations from the future users are.discussion what are the factors that influence the process of implementation of a digital tool that provides help and care on a distance? in what way does the digital tool ( care from a distance ) stimulate people who need chronically care to be more self - supporting? in what way does the digital tool support professionals to be able to stimulate people with schizophrenia to be more self - supporting ?

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hyper immunoglobulin e syndrome ( hies ) is a rare primary immunodeficiency disorder characterized by elevated serum ige , dermatitis , and recurrent skin and lung infections . three genetic etiologies of hyper ige have been identified : stat3 , dock8 , and tyk2 . association with tyk2 deficiency has shown some reported cases of hyper ige with disseminated nontuberculous mycobacterial ( ntm ) infection . microbial cultures of recurrent pulmonary infections show staphylococcus aureus , streptococcus pneumonia , and haemophilus influenzae more commonly while nontuberculous mycobacteria are secondary pathogens in pulmonary infections . tuberculosis ( tb ) has been rarely reported in children with hies with sporadic report of miliary tb . a 15-year - old boy who had been previously diagnosed to have hies in view of recurrent skin pustules with dermatitis and elevated serum ige ( ige = 3943 iu / ml [ normal = 10180 iu / ml ] ) along with skin biopsy suggestive of lymphomatoid papulosis presented with recurrent abdominal pain and fever 3 months ago . ultrasound ( usg ) of the abdomen showed multiple mesenteric lymphadenopathy , and mantoux test was positive ( 25 mm ) . he was started on four drugs for antituberculous therapy consisting of isoniazid ( h ) , rifampicin ( r ) , ethambutol ( e ) , and pyrazinamide ( z ) for 2 months and then hr as continuation phase . however , the child stopped antituberculosis treatment ( att ) after taking it only for 3 months . he then again had pain in the abdomen and presented to us for further management . on examination , his weight was 24 kg , height was 134 cm , and he had pallor and papular dermatitis all over the skin . on systemic examination , he had mild tenderness in the periumbilical region . investigations showed hemoglobin of 8.9 mg / dl , white blood cell count of 8100/cumm ( 58% polymorphs , 35% lymphocytes , 5% eosinophils , and 2% monocytes ) , erythrocyte sedimentation rate of 77 mm at the end of 1 h , and platelets of 238,000/cumm . usg of the abdomen showed multiple mesenteric lymph nodes with largest being 1.6 cm 1.4 cm . his urine showed albuminuria with microscopic hematuria . a repeat serum ige was elevated ( 8856 he was started on four drugs for att ( hrze ) , and urine was obtained for mycobacterium tuberculosis culture . natural human immunity to mycobacteria group relies on the functional interleukin-12/23-interferon - gamma integrity of macrophages connecting to t - lymphocytes / natural killer cells . patients with severe forms of primary immunodeficiency diseases have more profound immune defects involving this circuit , as seen in severe combined immunodeficiency , complete digeorge syndrome , x - linked hyper igm syndrome , cd40 deficiency , chronic granulomatous disease , and hies . a study shows that ntm infections are more common in hies patients with structural airway disease . in the absence of predisposing airway changes , ntm infections were not found in hies patients , suggesting that susceptibility to pulmonary ntm in hies may be more related to airway than immune dysfunction . an alternate explanation is the severity of immune dysfunction , through recurring infections , predisposes both to the severity of the structural lung disease and ntm disease . our patient was diagnosed to have abdominal tb based on the presence of mesenteric nodes , a positive mantoux test , and previous response to anti - tb therapy . we do not have bacteriological confirmation of tb as the lymph node biopsy was not undertaken in our patient . our patient did not have previous recurrent chest infections which could predispose to structural lung disease and thus he did not have pulmonary tb . the diagnosis of hies can be made based on a combination of clinical and laboratory findings for both types of hies . clinically , patients usually present with recurrent skin and lung infections , in the form of abscesses , dermatitis , or pneumonia . our patient had clinical features of skin involvement and elevated ige levels with recurrent skin infections suggestive of hies . we have not been able to do mutation analysis in our patient due to nonavailability of test . antibiotic prophylaxis with trimethoprim - sulfamethoxazole is frequently used as prophylaxis against recurrent respiratory infections . treatment of skin conditions such as eczema and skin infections is an important component of hies management . while there is no established guideline for the treatment of tb in primary immunodeficiencies , a case report of disseminated ntm in a patient with hies mentions that treatment with standard combination therapy for tb for a period of 12 months is indicated . another case report of miliary tb in hies was successfully treated with first - line att drugs . the role of interferon - gamma , granulocyte - colony stimulating factor , or other immune modulators in hies is unproven . bone marrow transplantation ( bmt ) is curative for autosomal - recessive hies with dock8 deficiency and it is recommended . autosomal - dominant hies patients do well with intensive therapy and supportive care , and bmt is not recommended for those individuals .

hyper immunoglobulin e syndrome ( hies ) is a rare primary immunodeficiency disorder characterized by elevated serum ige , dermatitis , and immunodeficiency that predisposes to multiple skin and lung infections . the most frequent pathogen responsible for infections in these patients is staphylococcus aureus . tuberculosis ( tb ) in patients with hies is an uncommon finding , and there are only a few reports of mycobacterial infections in known cases of hies . we present a case of abdominal tb that developed in a 15-year - old boy who also had hies .

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an 18-year - old man was referred to our clinic for the evaluation of blurred vision involving the left eye that had manifested four days previous . he had no specific medical , ocular , or trauma history . the subject 's best - corrected visual acuity was 1.0 in the right eye and 0.9 in the left eye . vf ( central 10 - 2 sita - standard strategy ) was measured with a humphrey field analyzer ii ( zeiss - humphrey , san leandro , ca , usa ) under continuous monitoring . the oct demonstrated disruption in the photoreceptor inner and outer segment ( is / os ) junction and undulation of the rpe with backscattering ( fig . 2c ) , but the oct findings did not reveal improvement compared to the initial findings ( fig . the oct demonstrated a recovery of continuity in the photoreceptor is / os junction , as well as decreased rpe irregularity with minimal backscattering ( fig . arpe is an acute , transient , foveal disturbance of unknown cause and which affects young adults . because of scarce case reports and an infrequent prevalence of the disease , the diagnosis can be difficult without suspicion . fluorescein angiography ( fa ) is a critical test for the differential diagnosis of arpe . unfortunately , we could not obtain fa data because t he patient had signs of an anaphylactic reaction , such as dizziness and difficulty breathing , immediately after the fluorescein injection . therefore , we made a diagnosis based on the fundus findings in combination with the oct and other characteristics of the disease . white dot syndromes , especially multiple evanescent white dot syndromes ( mewds ) , should be considered in the differential diagnosis of arpe . white dot syndromes are characterized by multifocal white lesions and are accompanied by mild vitritis . mewds differs from arpe in that the lesions are located outside the macula in the posterior pole , and electroretinogram ( erg ) findings are abnormal . the main lesions responsible for mewds cause damage to the photoreceptor outer segment but not the rpe . in our case , there was a single white dot lesion that was limited to the juxtafoveal region ; also , erg , which was performed on the first visit , showed normal findings . in acute posterior multifocal placoid pigment epitheliopathy ( apmppe ) , multiple , large placoid lesions start in the posterior pole and extend to the post - equatorial fundus , often accompanied by prodromal flu - like symptoms . our patient 's lesions were discrete clusters of a few subtle , small grey spots that resolved without scarring within three months after onset . in contrast , the lesions in patients with apmppe are replaced by rpe changes upon resolution . the oct demonstrated rpe involvement , which was similar to the previously reported oct findings . based on these findings , other diseases were ruled out and the patient was diagnosed with arpe . hsu et al . first described the oct findings of patients with arpe using time domain oct ( oct3 ; carl zeiss ) as a hyper - reflectivity involving the outer nuclear layer , photoreceptor , and rpe . hyper - reflectivity has not yet been evaluated with sd - oct . in our case , we found a definite disruption in the is / os junction , as well as an undulation of the rpe according to sd - oct . because sd - oct can provide better image resolution than oct3 , we believe that the disruption of the is / os junction with sd - oct presented as a hyper - reflectivity of the outer retina with oct3 . as reported by hsu et al . , the thickness of the hyper - reflectivity decreased with increased resolution , and we found a decreased length of the is / os disruption based on the serial observations of the oct findings . the rpe irregularity also decreased as the symptoms improved and as the threshold of the visual field increased . on the basis of these findings , the morphologic changes in inflammation related to arpe do not seem to be permanent .

we investigated the case of a young man with blurred vision in his left eye . his visual acuity was slightly decreased , and ophthalmoscopy disclosed a gray - white lesion in the macula . he had no systemic or ocular history . on the visual field test , the threshold sensitivity was decreased in the corresponding region . spectral domain optical coherence tomography ( oct ) demonstrated a disruption in the photoreceptor inner and outer segment ( is / os ) junction and undulation of the retinal pigment epithelium ( rpe ) with backscattering . we re - examined the patient after two weeks and after three months without any treatment . visual acuity and visual field results were gradually normalized , and oct demonstrated the recovery of continuity in the photoreceptor is / os junction , as well as decreased rpe irregularity with minimal backscattering . we used spectral domain oct instead of time domain oct ( oct3 ) so that we could provide better image resolution of the acute retinal pigment epitheliitis ( arpe ) . finally , we observed recovery of the functional and anatomical changes in the arpe patient with a resolution of the condition within three months following the initial examination , using oct and visual field tests .

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congenital maternal hyperplasia ( cah ) is a group of autosomal recessive disease in which dysfunction of one of the five cortisol coding genes occurs and causes enzymatic defects in cortisol synthesis cycle from cholesterol . the most common form of the disease is 21-hydroxilase deficiency , which accounts for 90 - 95% of the cah cases . in the 1950 s , it was found that there were a small group of patients that developed hypertension and responded to glucocorticoid . clinical manifestations of 11- - hydroxilase deficiency include , hypertension ( roughly occurs in 75% of patients and generally diagnosed during childhood ) and other signs related to overproduction of mineralocorticoids such as hypokalemia or muscle weakness , salt loss , and virilization . signs of androgen excessive secretion include early closure of the epiphysis ( short stature ) . the hypothalamic - pituitary - gonadal axis might cause amenorrhea or spermatogenesis disorders or hirsutism and acne . schmid type is an autosomal dominant skeletal dysplasia in which the commonest form is metaphyseal chondrodysplasia . the schmid metaphyseal chondrodysplasia is characterized by short stature , but bone maturation process is normal . however , bowed legs , coxa vara , and specific metaphyseal changes are seen on radiographs . in this article , we report a case of a 4-year - old boy with cah and schmid metaphyseal chondrodysplasia . our literature survey confirmed that it is the first reported case of coexistence of these two rare diseases . a 4-year - old boy with increased amount of pubic hair was referred to the children s endocrinology clinic . he was the first child of a family with normal growth and development until one year after birth . his parents were not related and there was no positive familial history . an increase in pubic and axillary hair developed four months later , he was treated for cah with 5 mg hydrocortisone , three times a day . another problem with this child was gait disorder and bowed knee ( figure 1 ) . there was a similar history with his cousin , considering that the cousin was affected by skeletal dysplasia . bowed knee in the patient with congenital adrenal hyperplasia . on clinical examination , doc level was 543 ( ng / ml ) and acth , dhea , 17-ohd , testosterone and androstenedione levels were higher than the normal limits . biochemistry tests such as bun , cr , na , p , k , and alkp were normal . laboratory test results radiological examination ( plain plantar view and knee radiography ) estimated the bone age to be about 10 years and 3 months . finally , the patient was treated for precocious puberty due to 11- - hydroxilase deficiency and schmid metaphyseal chondrodysplasia . after 5 months , testosterone level decreased to lower than 10 ng / dl and dhea reached 27 mcg / dl . schmid type metaphyseal chondrodysplasia is an autosomal dominant disorder and can be caused by various mutations in the col10a1 gene . its diagnosis is hard due to the rarity of the disease and its similarity to rickets , particularly vitamin d resistant type . however , rickets and schmid can be differentiated with normal bone density and irregular dense zone in schmid chondrodysplasia . in our case , schmid metaphyseal chondrodysplasia was considered by clinical manifestation and was confirmed with radiology and laboratory tests . bowed knee was the dominant characteristic sign and bone related biochemical tests such as ca , p and alkp were normal . the most important point in schmid disease is that over - treating by vitamin d , which can lead to toxicity , must be avoided . in some cases , congenital adrenal hyperplasias are a group of metabolic disorder diseases that lead to enzymatic defects in the biosynthesis of cortisol from cholesterol . clinical features of 11--hydroxylase deficiency in childhood might be premature pubarche and accelerated bone age , all of which occurred in our patient . the exact diagnosis of 11--hydroxylase deficiency can be performed by the high basal levels of deoxycorticosterone and or 11-deoxycortisol serums or tetra - hydrometabolites , which might be found during a 24-hour urine test . this disorder should be considered in patients with elevated serum levels of acth , about three times higher than the 95 percentile predicted for patient s age . its prevalence is 1 in 100,000 population and can be presented during childhood or adolescence . because of familial marriage in iran , cah incidence is high . according to a study by qaemi et al , rohani showed that precocious puberty could be a manifestation of non - classical form of cah and its early diagnosis and treatment is important . in some cases , . long - term complications of adrenal hyperplasia are short stature , infertility , gender identity disorders , and death . female neonates with ambiguous genitalia and male with precocious puberty are in the highest risk category to develop htn because of the high secretion of deoxycorticosterone ( doc ) . in our patient , mild htn was present . although cah can be associated with other genetic disorders , but this is the first report on the association between cah and schmid metaphyseal chondrodysplasia . 11--hydroxylase deficiency is a rare disorder and must be considered in patients with precocious puberty and secondly presented with hypertension .

congenital adrenal hyperplasia ( cah ) is a group of hereditary diseases , which are autosomal recessive . cah occurs due to defect in one of the cortisol coding genes and often clinically presents itself with signs of androgen overproduction . in this article , we report a case of cah and schmid metaphyseal dysplasia . our literature review indicated that this report is the first attempt on cyp11b1 and schmid dysplasia in a child . the specific diagnosis of 11--hydroxylase deficiency can be determined using high basal levels of deoxycorticosterone and/or 11-deoxycortisol serums .

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biotinidase ( ec 3.5.1.12 ) is the enzyme responsible for cleaving biocytin and recycling biotin from dietary protein - bound sources , . profound biotinidase deficiency ( less than 10% of mean normal serum activity ) ( omim # 253260 ) is an autosomal recessively inherited metabolic disorder . untreated individuals with profound biotinidase deficiency usually exhibit neurological and cutaneous symptoms with metabolic acidosis and organic aciduria , . symptoms of the disorder can be markedly improved or prevented with pharmacological doses of oral biotin . however , if treatment is delayed , once vision or hearing problems or developmental delays occur , they are usually irreversible . all states in the united states and many countries screen their newborns for the disorder . the gene encoding biotinidase ( btd ) has been isolated and characterized , and over 150 mutations causing biotinidase deficiency have been identified . we now report the first microdeletion of btd that involves three of the four exons of the gene . this deletion further exemplifies the importance of performing microarray analysis or other methodologies for a deletion of btd when the enzymatic activity indicates lower activity than can be attributed to the mutations identified by dna sequencing . microarray analysis was performed by whole genome chromosome prenatal reveal snp microarray ( integrated genetics , labcorp specialty testing group ) . dna sequencing of the biotinidase ( btd ) gene was performed by pcr amplification using primers and conditions described previously . all exonic and intron - exon boundaries of the btd gene were sequenced by prevention genetics ( marshfield , wi ) . a non - consanguineous couple had prenatal diagnosis by microarray analysis for advanced maternal age . the results of the microarray analysis revealed a 26 kb interstitial microdeletion of chromosome 3p25.1 p25.1 ( arr { hg19 } 3p25.1 ( 15,674,11915,700,291 ) 1 . this heterozygous deletion is involves a major portion of the btd gene , including exons 24 ( fig . 1 ) . based on this information , it was important to determine if the baby had a mutation on the other allele causing biotinidase deficiency . to determine the likelihood that the baby had such a mutation , the parents had their serum biotinidase activities determined . the father 's activity was 5.5 nmol / min / dl ( range of normal activity is 5.7 to 8.7 nmol / min / dl ) and the mother 's activity was 2.9 nmol / min / dl . these results indicated that the father had normal activity and did not have a mutation of btd and the mother had activity in the heterozygous range . in fact , microarray analysis of the parents revealed that the mother did have the microdeletion . the fetal dna was sequenced and did not reveal any other mutations or variants . at birth , the infant did not have biotinidase deficiency on newborn screening ; however , the infant did not have serum enzymatic testing to confirm that her biotinidase activity was in the heterozygous range . we have previously reported a child with a contiguous gene deletion that involved three genes , including the btd gene . the child reported here is heterozygous for a microdeletion that only involves the btd gene . this deletion involves three of the four exons of the btd gene and is predicted to result in complete loss of biotinidase activity . this was confirmed by finding activity in the heterozygous range in the mother who also has the microdeletion . the reference laboratory that performed the microarray analysis indicates that they report deletions as small as 50 kb and they may report susceptibility genes when they are associated with clinical presentations that have a clear phenotype . however , many commercial laboratories that perform microarray analyses do not report microdeletions or duplications of less than 200 to 400 kb , unless the alteration involves a gene known to cause a dominant pathogenic disorder . in the instance reported here , the deletion is only 26 kb and an alteration of the involved gene , btd , which is only pathogenic as an autosomal recessive disorder . therefore , it is possible that some or most laboratories would have not reported this deletion , unless they were specifically performing testing for biotinidase deficiency . if , for example , an individual has biotinidase activity in the profoundly biotinidase deficient range and has a missense mutation on one allele and a deletion on the other , sequencing would have only identified the missense mutation . if this scenario occurs in an asymptomatic , profoundly enzyme deficient infant identified by newborn screening , it is imperative to reconcile the low enzymatic activity with finding only a single mutation . there are occasions when enzymatic activity is lower than expected from mutation analysis . in these cases , it is precisely for this reason we have recommended that confirmatory enzymatic activities be performed on the proband , parents and an unrelated control , . if there is confidence that the reduced enzymatic is not due to poor sample storage , then it is important to consider the possibility that a microdeletion is present on the second allele to explain the lower enzyme activity . it is important to consider microarray analysis for a possible deletion in children identified as having enzymatic activity on newborn screening consistent with profound biotinidase deficiency , but only are found to have a single mutation by btd sequencing . the possibility of a deletion involving part or all of the btd genes must be considered in those children having enzymatic deficiency that is inconsistent with the results of their mutation analysis .

we report the first microdeletion ( 26 kb ) of the biotinidase gene ( btd ) that involves three of the four exons of the gene . this deletion further exemplifies the importance of performing microarray analysis or other methodologies for a deletion of the btd gene when the enzymatic activity indicates lower activity than can be attributed to the mutations identified by dna sequencing .

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in a recent issue of the new england journal of medicine , greet van den berghe and co - workers published a confirmation of the life - saving effects of tight glycaemia control by intensive insulin therapy ( iit ) in medical intensive care unit ( icu ) patients . this second study intended to answer some of the questions and criticisms raised by the landmark leuven study of 2001 , which reported a 4% decrease in mortality in a surgical icu population [ 2 - 7 ] . hopefully , these questions will be answered by the multi - centre assessments of the effects of iit , the nice - sugar and glucontrol trials , currently underway in australia and europe , respectively . before the completion of these two indispensable studies , greet van den berghe and colleagues wanted to confirm the life - saving effects of iit in medical icu patients in order to answer specific criticisms related to the type of patients , mostly surgical with two - thirds being post - cardiac surgery patients , in the first leuven study . indeed , patients with myocardial ischaemia could particularly benefit from a combination of a high amount of glucose and insulin , as reviewed recently . interestingly , krinsley reported a similar reduction in mortality in a mixed population of medical and surgical icu patients . greet van den berghe and colleagues succeeded but , as in most major contributions , their study raised more questions , which further argue for the importance of multi - centre trials . , this study can not be considered to be positive , as the long - stayers in whom a survival benefit was found were actually not randomised . the sample of long - stayer patients ( n = 767 ) was smaller than the calculated sample size of 1,200 patients that was required to test the working hypothesis ( a 7% reduction of the absolute risk of death ) with an alpha level of less than 0.05 and a beta level of 0.2 . in the entire set of patients , the intent - to - treat analysis indicated that there was actually no benefit related to iit . moreover , mortality in the patients in whom the stay was shorter than anticipated ( less than 3 days ) was higher in the iit group ( 26.8% ; 56/209 ) than in the conventional treatment group ( 18.8% ; 42/224 ) . this increase was found to be significant when analysed by the chi square test , but not by uncorrected proportional - hazards analysis , even after correcting for the difference in baseline risk factors . compared to the first leuven study , some of the recorded secondary end points that could be considered as surrogate markers of severity ( icu and 28-day mortality , requirement for dialysis , incidence of bacteraemia , requirement for prolonged antibiotic therapy , incidence of hyperbilirubinaemia and ' hyper - inflammation ' ) were not influenced as hospital mortality was . other local factors could also have influenced the results , thereby questioning the applicability of the findings to patients managed in other icus . the mean amount of parenteral glucose infused ( a mean of more than 220 g / day in long - stayers ) was probably higher than in most other icus , and parenteral steroids were used in more than half of the patients . unequivocally , these two factors reduce the risk of hypoglycaemia and the duration of episodes of hypoglycaemia , a major side effect of iit . nonetheless , the risk of hypoglycaemia was substantial , with 25.1% of the patients in the iit group experiencing a blood glucose level below 2.2 mmol / l ( 40 mg / dl ) at least once , compared to only 3.9% in the conventional group . the safety monitoring board of the german multi - centre study visep considered a similar increase in the incidence of hypoglycaemia important enough to stop this trial . clearly , the safety of iit needs to be assessed in patients with significant risk of hypoglycaemia . the target ranges of glycaemia were 4.4 to 6.1 mmol / l ( 80 to 110 mg / dl ) and 10 to 11 mmol / l ( 180 to 200 mg / dl ) , implying that there was no assessment of an intermediary blood glucose value , which is often used . greet van den berghe and colleagues already answered this question by analysing the data of the first study and concluded that there was a dose - response effect , with the largest improvement found in patients with the lowest blood glucose level . this hypothesis was found retrospectively , however , and clearly requires confirmation from prospective trials . the use of iit in leuven is probably easier than in other institutions with a lower nurse - to - patient ratio . for an iit approach , lastly , insulin exerts many effects other than a decrease in blood glucose , which could possibly be beneficial or deleterious in different subsets of patients ; these effects can not easily be assessed and monitored in the presently available trials . in summary , questions about the efficiency and safety of iit in different icus around the world are far from answered and much work has still to be done to answer the new questions raised by the second leuven study .

the second study on tight glycaemia control by intensive insulin therapy ( iit ) confirmed in medical intensive care unit patients the decrease in hospital mortality reported by the same team in the first iit trial in surgical patients . however , methodological concerns , the high rate of hypoglycaemia in spite of the infusion of large doses of parenteral glucose and the frequent use of steroids presently preclude considering these results as recommendations in other intensive care units , but rather argue for the need for large - scale assessment of the iit approach by multi - centre studies to confirm the efficacy and safety of this therapeutic modality .

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for decades , urinary sodium ( nau ) was used to define the presence of structural damage to the kidneys in the setting of oliguria or azotemia . the preserved capacity of the tubules to retain sodium was the physiological basis to interpret low levels of nau as a functional response to a low renal perfusion state : socalled ' pre - renal ' azotemia . the loss of this capacity by the kidneys was considered a marker of ' acute tubular necrosis ' ( atn ) . in the past , levels of nau below 20 meq / l were considered markers of pre - renal impairment and above 40 meq / l as markers of intrinsic renal disease . recently , ' pre - renal ' and ' atn ' paradigms have been frequently criticized : first , because many cases classified as atn lack this finding in histopathological studies ; and , second , because increasing knowledge of acute kidney injury ( aki ) revealed a dissociation between renal hemodynamics and nau , especially in sepsis . therefore , these old paradigms gave place to a new paradigm : that nau is useless as a tool in aki management . the aim of this commentary is to question if this new paradigm should be sustained . in 2006 , a systematic review revealed that nau values were widely variable within and between studies with no consistent values to distinguish normal kidney function , pre - renal azotemia and atn . a contemporaneous experimental study inducing hyperdynamic sepsis revealed progressively lower levels of nau . it was hypothesized that the sodium retentive state was due to loss of glomerular filtration pressure . the authors concluded that nau was not a reliable marker of renal perfusion ( breaking the old ' pre - renal ' paradigm ) . since then , a new paradigm has emerged : nau must not be used as a diagnostic tool in aki . this phenomenon is the possible explanation for apparently paradoxical increases in the sublingual tissue partial pressure of carbon dioxide ( pslco2 ; a marker of microcirculatory stagnation ) in parallel with increasing cardiac output during sepsis . hence , a similar phenomenon could explain the paradox between an increased renal blood flow and low nau levels . glomerular perfusion pressure , not total renal blood flow , is the main determinant for nau levels . in inflammatory states , low glomerular perfusion pressure may occur in the presence of increased renal blood flow , with activation of sodium - retaining mechanisms . although tubular injury is an early event in aki , most studies still found low fractional excretion of sodium levels in this context . we may conclude that too much injury is needed to impair the global tubular capacity to retain sodium . recent unpublished results by our group also suggest that sodium retention is progressively more intense with increases in aki severity except in very advanced stages ( aki network stage 3 ) ; we hypothesize that extensive tubular injury jeopardized sodium reabsorption . in our findings , such progressive decreases in nau began earlier than increases in creatinine , as described in a case report . first , nau is measured only once instead of sequentially ; as previously demonstrated , nau responds fast to acute hemodynamic alterations so that relative alterations in it may be more relevant than an isolated nau value . it is important to remember that nau has a very large physiological range that depends on numerous variables . of these , the most likely responsible for an abrupt decrease in nau value is a decrease in glomerular filtration rate . second , nau is still treated as a categorical variable ; the dynamism of nau is lost if nau is viewed as ' < 20 meq / l ' or ' > 40 meq / l ' . third , nau is usually assessed only in the presence of oliguria or azotemia . in a recent article , we suggested that urinary electrolyte measurement may alert for the presence of aki development before increases in creatinine or oliguria . in that study , patients who developed aki in the first 4 days after admittance to the icu had significantly lower nau values at admission . low nau values in aki can be a sign of microcirculatory impairment in the kidneys . we have observed many critically ill patients with very low nau levels on the day that renal replacement therapy was initiated . this is not surprising in the context of multiple organ failure , which may be caused by systemic microcirculatory failure . from this perspective , the lower the nau , the greater the microcirculatory stress . on the other hand , including 10 healthy volunteers , the mean nau was 104 48 meq / l . meq / l at icu admission in patients who did not develop aki during the study period . however , high nau values can be found in patients with aki receiving diuretics or in advanced aki stages . as for many other monitoring parameters in critical care medicine , the first step in defining nau utility in daily practice is to understand properly what it is saying to us and in which contexts . aki : acute kidney injury ; atn : acute tubular necrosis ; nau : urinary sodium .

urinary sodium ( nau ) is one of the oldest parameters used in the evaluation of azotemia and oliguria . over the past years , however , it has progressively been considered as obsolete and useless , especially in sepsis . it is common sense that nau frequently does not correlate well with global renal blood flow . if intrarenal microcirculatory changes are more important in acute kidney injury ( aki ) than changes in global renal blood flow , we speculate that decreases in nau may be viewed as a possible marker of microcirculatory impairment in the kidneys . recent findings by our group ( some not yet published ) in which sodium retentive capacity is preserved until advanced stages of aki and the observation of decreases in nau preceding increases in creatinine bring us to conclude that the new paradigm of abolishing nau consideration from daily approaches to managing patients at risk for aki must be reevaluated .

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seroprevalence of hydatidosis is between 1.2 and 13.8% in the different region of iran ( 1 ) . brain involvement occurs in 12% of patients with hydatidosis and comprises 2% of all brain masses in the endemic region , especially among the children ( 2 ) . symptoms of the brain hydatid cyst vary by location and size of the cyst . in a retrospective study of 117 children in tunis , common symptoms were a headache , vomiting , hemiparesis , seizure , mood alteration , and skull deformity ( 3 ) . more than 50% of patients with the brain hydatid cyst have multiple cysts in the brain , and about 18% have visceral involvement , which has 9% mortality rate ( 4 ) . surgical treatment and excision of the cyst ruptures of the cyst and anaphylactic reaction are the most frequent complication of the surgery with the prevalence of 10% . here we present an adult male with a large brain hydatidosis and atypical symptoms , who was treated successfully by surgery . a 19 yr - old male livings in ardabil , northwest of iran , was referred to neurosurgery clinic in khanevadeh university hospital , tehran , iran for a headache and progressive left hemiparesis since a week ago . he was treating for depression and incuriosity for two months by a psychologist . during two weeks , he developed headache , vomiting , and in the last week , left - side paraparesis occurred . peripheral neurological examination showed left hemiparesis with the force of three to fifth in all muscle groups and normal deep tendon reflexes . brain contrast - enhancing magnetic resonance imaging ( mri ) showed a large round mass about eight centimeters in the right frontoparietal lobe and mildly shifting the cerebral hemisphere to the left , without any ring - enhancement or surrounding edema ( fig . 1 ) . contrast - enhanced t1-weighted brain mri shows a large brain hydatic cyst in the right frontoparietal lobe with mildly shifting of the cerebral hemisphere laboratory results showed mild anemia ( hemoglobin=13.1 milligrams per deciliter ) . further laboratory and imaging findings were normal , except for antibody titer against echinococcosis , which raised fourfold above - normal range in igg . oral treatment with albendazole at a dose of 400 mg twice daily was initiated , and the patient underwent surgical excision of the cyst . right craniotomy through parietal bone with left side , head turning position was done under general anesthesia . a largely fluctuated mass with adhesion to surrounding tissue appeared after dural opening , and 3% warm saline infused around the mass during 30 min , which led to slowing delivery of the mass in the next 40 min ( fig . 2 ) . surgical excision of the brain hydatic cyst via right craniotomy through parietal bone finally , free layers were sutured anatomically consequence , and the bone fixed by bone - adhesive glue . oral treatment continued about three months . after one - yr follow - up , left hemiparesis improved , and the left side limbs forces raised to four - fifths . at this time brain mri cystic hydatid disease ( chd ) is an infectious parasitic disease , often caused by e. granulosus . this parasite distributes worldwide , and the main routes of transmission are ingestion of foods or water , which is contaminated by scolex or egg . 12% of the patients have cerebral involvement , especially in the parietal lobe ( 2 ) . brain hydatid cysts may grow up 110 cm a year and symptoms are often related to elevated intracranial pressure ( icp ) and focal neurological deficit ( 5 ) . in our patient , symptoms of rising icp appeared in the advanced course of the disease and the main complaints , which resulted in seeking medication , were depressive mood alteration , personality instability , and apathy . dowling technique is often used for surgical excision , which consists of craniotomy through a big opening hole , cortical dissection and hydrostatically delivery of the cyst using an injection of hypertonic saline ( 3 ) , such as our patient . ruptures of the cyst and anaphylactic reactions are the most common complication of the surgery , but other complications such as subdural effusion , hematoma , pneumocephalus or herniation must be considered ( 6 ) . emergent brain surgery was performed in a 13 yr - old male for cerebral herniation because of a massive brain hydatid cyst ( 5 ) . despite the significant size of the cyst and adhesion to surrounding tissue in our patient , he did not experience any complication . it seems that other related factors such as adhesion to surrounding tissue , the severity of intracranial pressure , access to the operative field , and expertness of the surgeon may influence the outcome . we believe that the best surgical technique for excision of the cyst is adequate craniotomy , gentle decortication and enough time spending during the surgery .

brain hydatid cyst constitutes one of the leading causes of cranial mass in the endemic region , especially among the children . symptoms of the disease are associated with rising of intracranial pressure and focal neurological deficit . surgical excision of the cyst is essential to treatment . here we present a 19 yr - old male livings in ardabil , northwest of iran , referred to neurosurgery clinic in khanevadeh university hospital , tehran , iran with a large brain hydatid cyst and atypical symptoms . he was treated by surgical excision of the cyst without any complication . there were not any clinical or radiological signs of recurrence after one - year follow - up .

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the problem of finger in the patient with diabetes mellitus is important consideration in diabetology . generally , peripheral neuropathy that manifests with finger paresthesia , numbness , and blanching is common . nerve conduction studies , vibration and temperature threshold measurements , and neurovascular function tests are useful for assessment of these cases . however , some recent reports mention the concern on trigger digits in diabetic patients . in this brief article , the authors focus review and discussion on this specific topic . indeed , trigger digits can be seen in any population . however , some recent publications report on the importance of this disease in diabetes mellitus . the incidence of trigger digits was about four times higher than in the general population . the trigger digits can be seen in 1:20 ( cases with trigger digits : all diabetic patients ) . however , some other reports such as that by aydeniz et al . show no significant increase in the incidence of this condition in diabetic patients . it is concluded that the trigger digits are an important problem for the diabetic patients . screening for diabetes may be warranted in patients with involvement of more than three digits . the summary of the important publications reporting on the prevalence of the problem can be seen in table 1.[711 ] although there is a difference in the rate of reported prevalence and whether the prevalence among diabetic patients is higher than that of normal population or not is still questionable , it can not be refused for the important of the problem on trigger digits among diabetic patients . summary on some important publications reporting the prevalence of trigger digits among diabetic patients however , there is no clear evidence that diabetes mellitus increases the risk for development of trigger digits . although it is not questionable that overall musculoskeletal problems increases in diabetic patients , there is no conclusion on the specific trigger digits problem . focusing on the existed evidences , trigger digits are common in old diabetic patients but not relating to sex , age , and type of diabetes.[71113 ] it is of interest that there is an observation that limited joint mobility is related to multiple digits involvement in diabetic patients with trigger digits , but there is no relationship with age , sex , type of diabetes . although the trigger digits can be seen and similarly diagnosed in both normal and diabetic patients , natural history of the condition in diabetic patients and the outcome of treatment may not be the same . it is reported that the insulin - dependant cases usually have more sever symptoms and multiple digits involvement and require surgical release for relief of symptoms . the steroid injection might be used although it does not provide a good success rate ( about 30%).[1719 ] in addition to use of steroid injection , the use of nonsteroidal anti - inflammatory drugs can provide a little relief from the symptoms.[1719 ] the use of steroid injection ( either methylprednisolone acetate or triamcinolone acetonide ) is proved to be safe ; however , there is also a report showing that the use of steroid injection can result in hyperglycemia . in addition , the recurrent rate is very high in the diabetes type 1 cases . focusing in detail , for the surgical treatment , it is used in the severe cases.[172426 ] the recommended surgical technique is surgical release of the first annular ( a1 ) pulley . the surgery might be a definitive treatment ( success rate up to 99% ) but the complications can be seen and the postsurgical physiotherapy is still required for a long time . of interest , a recent report indicated that diabetes was not a risk factor for trigger digits and postoperative complications of trigger digits surgery . however , closed observation and special care are still recommended for the cases with diabetes mellitus due to the risk for existence of microangiopathy . trigger digits is an important problem in diabetic patients , especially for the old ones . however , the cause result relationship between diabetes mellitus and trigger digits is still the topic for further study . in management , glucose control is important and the standard managements for the general population can be effectively used for the diabetic cases .

the problem of finger in the patient with diabetes mellitus is important consideration in diabetology . generally , peripheral neuropathy that manifests with finger paresthesia is common . however , some recent reports mention the concern on trigger digits in diabetic patients . in this brief article , the authors focus review and discussion on this specific topic . the searching of standard database , pubmed , on available publication on this area selecting by keywords trigger digits and diabetes mellitus was done and all derived papers were extracted and further synthesized in this review article . epidemiology and management of the condition are the mainly focused reviewed areas and presented in this article .

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clear - cell sarcoma ( ccs ) is a rare malignant connective tissue tumor that was first described by enzinger in 1965 . ccs occurs most commonly in adolescents and young adults and the patients aged > 60 years are rare . it has a high rate of local recurrence , regional lymph node metastasis , and distant metastasis [ 1 , 2 ] . ccs occurs most commonly in the deep soft tissues of the extremities , with rare involvement of the head , neck , and trunk . we experienced a very rare case of primary ccs in the mediastinum with successful surgical resection in an elderly patient . in october 2011 , a 63-year - old man presented with a mass in the right upper mediastinum on chest radiograph . computed tomography showed a large oval mass ( 55 55 85 mm ) with clear margins and heterogeneous enhancement in the right upper posterior mediastinum between the phrenic nerve and vertebrae ( fig . 1b ) , and 18f - fluorodeoxyglucose positron emission tomography showed abnormal uptake with a maximum standardized uptake value of 15.1 . the tumor was excised by video - assisted thoracoscopic surgery , with negative surgical margins . the tumor was soft , well circumscribed , encapsulated , and did not invade the pleura . histological examination of the resected tumor with hematoxylin and eosin ( he ) staining showed morphological features compatible with conventional soft tissue ccs . the tumor cells were distributed in nests separated by fibrous connective tissue , showing an alveolar pattern ( fig . the tumor cells had eosinophilic and clear cytoplasm and oval vesicular nuclei of varying sizes with characteristic prominent eosinophilic nucleoli . 2c ) , s-100 protein , calretinin , cd34 , cd56 , cd68 , and cd117 ; and negative for melan - a , cytokeratins ae1ae3 , cytokeratin cam5.2 , chromogranin a , desmin , caldesmon , myogenin , epithelial membrane antigens , cd57 , cd99 , and d2 - 40 . ccs is a very rare soft tissue neoplasm , which occurs in the extremities in 9095% of cases . our case was diagnosed with a primary ccs in the mediastinum . a search of the english literature revealed only one previously reported case of primary ccs in the mediastinum . recent cytogenetic studies showed that ccs is associated with the translocation t(12;22 ) ( q13;q12 ) ; this translocation is observed in up to 93% of patients with ccs , but never observed in malignant melanoma . in our case , we were unable to determine whether this translocation was present , because of the lack of availability of such analysis in japan . the rarity of ccs in the mediastinum makes it difficult to draw conclusions regarding prognostic factors . reported that complete excision of the primary tumor with wide surgical margins appears to be the optimal approach to treatment , with or without adjuvant radiation therapy . ccs is an aggressive malignant tumor , and unlike most soft tissue sarcomas , it often metastasizes to regional lymph nodes . in our case , we did not have a definitive diagnosis at the time of operation , and therefore we did not perform excision of the tumor with wide margins or dissect the regional lymph nodes . in most cases , ccs has a relentlessly progressive course and results in death because of widespread dissemination . the reported overall survival rates without local recurrence are 30% at 5 years and 16% at 10 years . some patients experience rapidly fatal progression , and late metastasis after many years of freedom from disease is also relatively common . we believe that regular long - term follow - up with computed tomography , magnetic resonance imaging , and 18f - fluorodeoxyglucose positron emission tomography examination is important to identify local recurrence or distant metastasis as early as possible .

we report a case of primary clear - cell sarcoma ( ccs ) in the mediastinum . in october 2011 , a 63-year - old man was admitted to our hospital for surgical resection . the tumor was completely excised by video - assisted thoracoscopic surgery . the tumor was well encapsulated and did not invade the pleura . histological examination led to a final diagnosis of primary ccs in the mediastinum . the patient remains alive without evidence of recurrence at 15 months after surgery .

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a 78-year - old person , retired as school teacher with no history of any substance abuse or medical history . he had been suffering from bilateral knee pain diagnosed as osteoarthritis for the last 20 years and was not on any steroids . he presented with 1-month duration of altered behavior characterized by excessive psychomotor activity , emotional lability , increased self - confidence , excessive talkativeness , decreased need for sleep , and increased sense of energy level . there were a few occasions when he misrecognised his daughter as his wife who had passed away 6 months ago . on examination , he was an elderly obese person weighing 92 kg with bmi > 27 . mental status examination revealed increased irritability , increased psychomotor activity , and increased self - confidence and authoritativeness . he had similar behavior 3 years ago , diagnosed and treated as mania in the same hospital . there was positive family history in the form of his mother having had symptoms suggestive of postpartum psychosis . investigations including hiv , vdrl , serum vitamin b12 level , tft , eeg , routine hemogram , serum electrolytes , blood urea , and serum creatinine were normal . ecg revealed no abnormalities , except for a prolonged corrected qt interval ( qtc ) of 486 msec . his ct brain [ figure 1 ] showed cortical atrophy , especially pronounced in the temporal and frontal areas . the periventricular regions in the frontal lobe showed mild hypodensity suggestive of small vessel ischemic disease . he didnt fulfill international consensus criteria for behavioral variant of frontotemporal dementia ( ftd ) . ct brain shows frontal cortical atrophy ( top left ) , with frontal subcortical white matter hypodensities ( top right ) . widened sylvian fissures suggestive of temporal pole atrophy and basal ganglia calcifications are also seen ( bottom left ) . widening of temporal horns of lateral ventricles ( bottom right ) is further evidence of temporal lobe atrophy he was initiated with aripiprazole 5 mg / day because of his qtc being 486 msec . given his age , we chose aripiprazole as it is less sedating and has lower chances of producing postural hypotension . he also required intravenous lorazepam 2 mg sos for his agitated behavior for the first 2 days . aripiprazole was gradually increased to 15 mg , and by the end of 3 weeks he showed complete remission of his manic symptoms . at the time of admission in psychiatry ward , he scored 27 on mania severity on young 's mania rating scale ( ymrs ) and it came down to 4 at the end of 3 weeks . occurrence of first episode of mania in the elderly ( after 60 years age ) warrants a high index of suspicion for organic causes . , we did not find any obvious etiology for his mental illness , except for frontal and temporal atrophy and possible frontal sub - cortical white matter involvement . one possibility considered was that of ftd , but this was ruled out as he did not fulfill criteria for dementia and behavioral component of ftd . it is possible that the manic episodes were the initial manifestations of ftd in this patient and this will be tested only in the follow - up . there was family history of postpartum psychosis in his mother , which is associated with a family history of bipolar disorder and the index case had been diagnosed as mania 3 years ago with complete remission in the interval period . these two points and absence of any abnormalities in laboratory and clinical examination favored diagnosis of mania . aripiprazole has relatively high affinity for both 5-ht2a and d2 receptors similar to other atypical antipsychotics but differs from them due to its partial agonist activity at d2 and 5-ht1a receptors . it has a more favorable side - effect profile compared to typical and even atypical antipsychotics . aripiprazole has lesser propensity to cause extra - pyramidal symptoms , an important factor in the elderly . other desirable properties are lesser chance for causing side - effects like dyslipidemia , glucose intolerance , hyperprolactinemia , postural hypotension , sedation , qt prolongation , and weight gain . its efficacy has been proven as monotherapy in treating acute mania and in prophylaxis of bipolar disorder . hence , we chose aripiprazole for our patient in view of his already prolonged qtc interval and age of 78 years . this case is unusual in that age of onset of mania is very late ( 75 years ) and during the current second episode we could not find any organic etiology . hence , aripiprazole may be a safe and effective antipsychotic medication for the control of mania in the elderly .

new - onset bipolar disorder is rare in the elderly . symptom profile is similar to that in young adults but the elderly are more likely to have neurological co - morbidities . there are no case reports of elderly mania being treated with aripiprazole , an atypical antipsychotic . a 78-year - old gentleman presented to us with symptoms suggestive of mania of 1 month 's duration . he had similar history 3 years ago and a family history of postpartum psychosis in his mother . there were no neurological signs on examination and work - up for an organic etiology was negative except for age - related cerebral atrophy . he improved with aripiprazole and tolerated the medications well . the use of psychotropic medications in the elderly is associated with side - effects of sedation , increased cardiovascular risk , and greater risk of extra - pyramidal side - effects . the use of partial dopaminergic antagonists like aripiprazole may be useful in the balancing of effects and side - effects .

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( baill . ) is a source plant of traditional chinese medicine . the s. chinensis fruit ( sf ) , which has five - flavored fruits ( salty , pungent , bitter , sweet and sour ) , is called wuweizi in china . it naturally distribute in china , russian , korea and japan and was used in treatment of diseases including liver injury , tumor inhibition , urinary tract disorders , insomnia and palpitation , even inhibit both hiv-1 rt - associated dna polymerase activity and virus replication . in addition to its application as a chinese herbal medicine . it is also used for making beverages , tea , wine , health food industries and cosmetics . therefore , recent research has been focused on extracting active ingredients and identifying function . despite of the importance of s. chinensis , the next generation sequencing technology has dramatically improved the efficiency and speed of gene discovery . two cultivars yanhong ( red skin ) and jinwuwei ( white skin ) were grown in orchard located in zuojia town , jilin city , jilin province , china ( 440647 n 1260718 e ) that belong to the plant resources nursery of institute of special wild economic animal and plant science , chinese academy of agricultural sciences . fours samples , including fruits and skins , were harvested and immediately frozen in liquid nitrogen for further experiments . total rna was extracted using modified ctab method , library construction and high - throughput sequencing for each sample was performed at a contract sequencing company ( novogene , china ) . the cdna library was sequenced using an illumina hiseq2500 platform . with the purpose of determining the red fruit , white fruit , red skin of red fruit and white skin of white fruit of s. chinensis . we obtained a total of 72.5 million , 55.1 million , 68 million and 69.3 million raw data reads from four sequencing libraries prepared , respectively ( table 1 ) , > 94.9% bases has a q value 20 ( an error probability of 0.035% ) . after cleaning and quality checks , the de novo assembly of all sequencing data using the trinity method . it generated 92,415 all - transcripts with an average length of 496 bp and an n50 of 1466 bp ; and 71,443 all - unigenes were achieved . of these , 46,461 ( 65.0% ) were 200500 bp , 11,322 were 5001000 bp , 8612 were 12 kb and the remaining 5078 were > 2 kb ( table 2 ) . the results provides us useful information for further explorer the gene synthesis pathways of active ingredients . two cultivars yanhong ( red skin ) and jinwuwei ( white skin ) were grown in orchard located in zuojia town , jilin city , jilin province , china ( 440647 n 1260718 e ) that belong to the plant resources nursery of institute of special wild economic animal and plant science , chinese academy of agricultural sciences . fours samples , including fruits and skins , were harvested and immediately frozen in liquid nitrogen for further experiments . total rna was extracted using modified ctab method , library construction and high - throughput sequencing for each sample was performed at a contract sequencing company ( novogene , china ) . with the purpose of determining the red fruit , white fruit , red skin of red fruit and white skin of white fruit of s. chinensis . we obtained a total of 72.5 million , 55.1 million , 68 million and 69.3 million raw data reads from four sequencing libraries prepared , respectively ( table 1 ) , > 94.9% bases has a q value 20 ( an error probability of 0.035% ) . after cleaning and quality checks , the de novo assembly of all sequencing data using the trinity method . it generated 92,415 all - transcripts with an average length of 496 bp and an n50 of 1466 bp ; and 71,443 all - unigenes were achieved . of these , 46,461 ( 65.0% ) were 200500 bp , 11,322 were 5001000 bp , 8612 were 12 kb and the remaining 5078 were > 2 kb ( table 2 ) . the results provides us useful information for further explorer the gene synthesis pathways of active ingredients .

the fruit of schisandra chinensis turcz . ( baill . ) , namely wuweizi in china , is a well - known herbal medicine and health food . at present , research focused on the extraction of effective chemical component and function identification . little known about the secondary metabolism gene pathway of chemical composition . its fruit color usually red , however , the white fruit color variation has been found . it made us interested in exploring which gene change lead to this result . in order to understand the genetic background of s. chinensis , we performed a transcriptome analysis of s. chinensis , including red fruit and skin of yanhong cultivar and white fruit and skin of jinwuwei. we obtained 26.4 gb raw data ( ncbi accession number : ssr4449123 ) . de novo transcriptome assembly using trinity revealed 92,415 transcripts and generated 71,443 unigenes . all unigenes were annotated in database.this study provides transcriptome of s. chinensis , which might be useful for comparative transcriptome analyses and understand gene expression of secondary metabolites .

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neuroendocrine ( ne ) carcinoma of the breast was first described by cubilla and woodruff in 1977 . primary ne carcinomas of the breast are rare and represent about 2 - 3% of breast carcinomas . they are much rarer in the male breast occurring in less than 1% of the cases . ne breast cancers encompass a heterogeneous group of tumors exhibiting morphological features similar to those of ne neoplasms of the gut and lung . they invariably express one or more ne markers ( neuron specific enolase , chromogranin , synaptophysin ) in at least 50% of the tumor cells . we report one such rare case in male breast diagnosed by fine needle aspiration cytology ( fnac ) . an 80-year - old male presented with a mass in the right breast of 10 years duration with rapid progression in growth since the previous 3 months . mammography showed a well - defined mass in the right breast with ipsilateral axillary lymphadenopathy . fine - needle aspiration cytology showed cellular smears with dispersed single cells and loose sheets . many nuclei showed moderate anisocytosis , irregular nuclear contours , prominent nucleoli and sprinkled chromatin [ figure 1a and 1b ] . considering these features , a cytological diagnosis of ne carcinoma of the breast ( a ) highly cellular smear , dispersed cells and loose sheets ( pap , 100 ) . ( b ) tumor cells with anisocytosis , irregular nuclear borders , prominent nucleoli and sprinkled chromatin ( pap , 400 ) the patient underwent right radical mastectomy with axillary lymph node dissection . gross examination revealed a tumor measuring 10 cm 8 cm 8 cm , with a solid , grey - white appearance and a focal cystic area [ figure 2a ] . histopathological sections studied showed a malignant tumor with cells in solid nests , trabecular and organoid patterns , separated by delicate fibro - vascular connective tissue stroma . a focal area in the tumor also showed features of invasive papillary carcinoma [ figure 2c ] . four out of the six resected lymph nodes showed metastatic deposits from the primary breast tumor . ( a ) gross specimen showing solid , grey - white tumor with a cystic area . ( c ) focal area in the tumor with features of papillary carcinoma ( h and e , 400 ) . ( d ) tumor cells with cytoplasmic chromogranin positivity ( ihc , 400 ) immunohistochemical staining revealed cytoplasmic positivity for chromogranin which confirmed the ne nature of the tumor [ figure 2d ] . to exclude a non - mammary primary site , the chest , abdomen and pelvis were thoroughly examined for abnormalities , but none was detected . final diagnosis of primary pure solid ne carcinoma of the breast with a minor component of papillary carcinoma and metastasis to ipsilateral axillary lymph node was arrived at , following which the patient received adjuvant chemotherapy . features of primary pure neuroendocrine breast carcinomas ( nebc ) were considered to be special features within conventional breast carcinomas until recently . in 2003 , the world health organization classification of breast tumors definitely established that , the immunohistochemical expression of ne markers in more than 50% of the tumor cell population is the unique requisite for nebc diagnosis . but , invasive papillary carcinoma associated with ne carcinoma in the male breast as seen in the present case is an unusual occurrence . endocrine hormone related syndromes are rare in primary nebc despite the presence of widespread disease . the tumor cells in ne carcinomas of the breast are argyrophilic in contrast to the gut and lung carcinomas which may be of either argyrophil or argentaffin types and are hence called argyrophilic carcinomas . argyrophilic carcinomas of the male breast are ne tumors containing predominantly chromogranin b. most argyrophilic tumors show uniform cellularity and expansive growth as in the present case . despite being a distinct morphological and immunohistochemical entity , no statistically significant differences are seen between argyrophilic and the other commoner types of male breast carcinomas . stage on an average is higher than for the other breast carcinomas , as in the present case , since more than 50% of the patients show lymph node metastasis at the time of presentation . however , in the present case , the predominant component was ne carcinoma which was associated with a minor component of papillary carcinoma . about 50% of well - differentiated and moderately differentiated ne carcinomas express chromogranin a and b. only 16% express synaptophysin . mucin as seen in the present case , as also observed by stita et al . , in 26% of cases and was considered to be a favorable prognostic factor by them . ne breast carcinomas are observed not to exhibit aggressive behavior despite the presence of adverse prognostic factors . good prognosis in these tumors relates to their intrinsic nature and high rate of treatment responses . ne carcinoma in male breast is a rare entity but does occur and can be diagnosed on fnac . however , it is important to distinguish the primary nebc from metastatic carcinoma because the management and prognosis differ . primary ne carcinoma of the breast may pose a cyto - histologic diagnostic challenge due to the rarity of its occurrence and paucity of literature . nevertheless , it is an entity which no pathologist can afford to miss since it is less aggressive and shows a high rate of treatment response .

primary neuroendocrine ( ne ) carcinoma of the male breast is very rare . ne breast cancers encompass a heterogeneous group of tumors exhibiting morphological features similar to those of ne tumors of the gut and lung . they express one or more ne markers in at least 50% of the tumor cells . they are rare lesions representing about 2 - 3% of all breast cancers and more frequently affecting elderly patients . the present case report deals with a ne breast carcinoma in an 80-year - old male who presented with a right breast mass and axillary lymphadenopathy . the cytological features were suggestive of ne carcinoma . a thorough clinical evaluation helped confirm the primary nature of the tumor . the histopathology and immunohistochemistry helped confirm the cytological diagnosis . the histogenesis and prognostic implications of this rare breast tumor with predominant ne differentiation , unusually occurring in a male , is discussed here .

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how does the care for older people maintain of good quality , according to their needs , while still fundable ? looking at the most vulnerable group of older people with dementia , for which family and friends represent past , present and future , adjustments need to be made . family , nurses and organization all acknowledge that nursing home care will benefit when these three parties unite their forces as partners in care . thuis voelen is a guide filled with practical suggestions to accomplish a valuable partnership between family , nurses and organization . loosely structured conversations between these three parties aim to establish reciprocity and trust and finally to end up with a number of incentives , which concern family as well as employees and range from the moment of intake until the phase of saying goodbye . advises in the area of architecture , furnishing , public space , human resources and ict are provided as well . a nursing home creates its own worthy care agenda, to accomplish small , practicable changes . resulting in : a changing ( physical ) environment ; the family enjoys visiting the nursing home and stays longer , nurses enjoy their work environment more and feel relieved. more mutual complicity and responsibility in wanting to improve the quality of life of elderly together . a changing ( physical ) environment ; the family enjoys visiting the nursing home and stays longer , nurses enjoy their work environment more and feel relieved . more mutual complicity and responsibility in wanting to improve the quality of life of elderly together . thuis voelen is an example of how small scale innovations , new ideas and simple things that matter can establish an improvement of the situation in nursing homes . within an open - minded organization , any moment an inhabitant and/or his relatives are satisfied and happy , will have a positive effect on the ( culture in ) organization ; time and money will be spared .

introductionhow does the care for older people maintain of good quality , according to their needs , while still fundable ? looking at the most vulnerable group of older people with dementia , for which family and friends represent past , present and future , adjustments need to be made . family , nurses and organization all acknowledge that nursing home care will benefit when these three parties unite their forces as partners in care.aimsthuis voelen is a guide filled with practical suggestions to accomplish a valuable partnership between family , nurses and organization . loosely structured conversations between these three parties aim to establish reciprocity and trust and finally to end up with a number of incentives , which concern family as well as employees and range from the moment of intake until the phase of saying goodbye . advises in the area of architecture , furnishing , public space , human resources and ict are provided as well.resultsa nursing home creates its own worthy care agenda, to accomplish small , practicable changes . resulting in : a changing ( physical ) environment ; the family enjoys visiting the nursing home and stays longer , nurses enjoy their work environment more and feel relieved. more mutual complicity and responsibility in wanting to improve the quality of life of elderly together . conclusionsthuis voelen is an example of how small scale innovations , new ideas and simple things that matter can establish an improvement of the situation in nursing homes . within an open - minded organization , family is thought of as valuable . any moment an inhabitant and/or his relatives are satisfied and happy , will have a positive effect on the ( culture in ) organization ; time and money will be spared .

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first - line procedural investigations for non - specific epigastric pain include endoscopy and endosonography , combined with biopsy of suspicious areas in the upper gastrointestinal tract . despite high sensitivity and specifity , obtaining a definite histopathologic differentiation is sometimes unsuccessful , particularly if biopsy is not representative , e.g. because of tumor bleeding into the affected tissue . in such a case , the single remaining diagnostic option is a curative laparoscopic endoscopically - assisted wedge - resection of the stomach . we present an interdisciplinary case report in which a tumor of the stomach , suspicious for gist , was removed surgically and was found to be a benign glomus tumor by definitive histology . a 44-year old patient presented with relapsing epigastric pain of variable intensity and tarry stool , beginning three days earlier . there were no clinical or laboratory findings to suggest an acute posthemorrhagic anemia ( hemoglobin 14.7 g / dl , hematocrit 43.2% , pt 118% , aptt 27 s ) . upper endoscopy and endosonography were performed , revealing a spherical , submucosal , solid tumor approximately 50 mm in diameter located in the pyloric antrum ( fig . overlying ulceration was considered the likely source of bleeding , and fine needle biopsy of the lesion ( 19 g ) was performed , followed by preventive clip - application . an endosonographic picture with a poorly reflective , non - homogeneous pattern and echo - free areas was compatible with a 35 30 mm gist ( fig . investigations thus far were inadequate to exclude a malignant process , and surgery ( endoscopically - assisted laparoscopic wedge - resection of the stomach ) was indicated . histological section of the surgical specimen revealed a rare benign glomus tumor ( positive reaction on markers specific for glomus tumor ( vimentin / actin ) , fig . glomus tumor is a ( quite ) rare neoplasm and despite local invasion of vessels is mostly benign . it is often found in the skin ( particularly in the dermis / subcutaneous tissues of the limbs ) , but can also be found in the gastrointestinal tract ( usually intramurally in the mucosa / submucosa and serosa ) as well as other solid organs . the present assumption that the first glomus tumor of the stomach was identified in 1948 and described with another two cases in 1951 smol'iannikov wrote that the first glomus tumor of the stomach was clearly described in a 64-year old man in 1928 by talijeva . furthermore , the first malignant glomus tumor of the stomach was diagnosed in 1939 by kirschbaum et al . in a 40-year old man . there are case reports of malignant glomus tumors of the stomach in girls aged 1214 years , published by yannopoulos et al . . in addition , there have been a number of reports pertaining to glomus tumors of solid abdominal / retroperitoneal organs and of the colon [ 6 , 7 , 8 ] . there is no gender bias in the incidence of glomus tumors but their peak incidence occurs between the fourth and sixth decade of life . immunohistochemically , most glomus tumors show a positive expression of vimentin / actin without expressing chromogranin a , neuronspecific enolase ( nse ) , carcinoembryonic antigen ( cea ) or epithelial membrane antigen ( ema ) . the rarity of glomus tumor , its variable organ involvement , its non - specific symptoms at presentation and the often equivocal results of standard first line investigations all contribute to diagnostic difficulty . endoscopic and endosonographic images in glomus tumors of the stomach show a solid , submucosal tumor with or without ulceration and do not differentiate it from other important diagnoses , e.g. gist , neuro - endocrine neoplasia ( carcinoid ) , angiomyoma or lymphoma . hence , presurgical diagnostic confirmation is often impossible . only immunohistochemical analysis of representative biopsies ( gist : positive reaction with cd-117 antibodies and missing expression of glomus tumor typical actin / vimentin ) can confirm the diagnosis , and hence aid the clinician in determining appropriate therapy and prognosis [ 9 , 10 , 11 ] . compounding pre - surgical diagnostic difficulties , there have also been reports of malignant transformation of glomus tumor . therefore , surgery or en bloc endoscopic enucleation is in most cases the remaining diagnostic and therapeutic option [ 12 , 13 ] . a definite immunohistochemical confirmation of the diagnosis is essential , because the prognosis of a potentially malignant lesion is otherwise unpredictable . in the case presented , a patient with recurring epigastric pain and melaena , there was an endoscopic and endosonographic finding of a submucosal tumor of the gastric antrum . given the non - diagnostic biopsy result and the ongoing risk of gastrointestinal bleeding , endoscopically assisted laparoscopic wedge - resection of the stomach was performed as a combined diagnostic and therapeutic procedure . histological and immunohistochemical analysis of the resected tissue showed an entirely removed ( ro ) glomus tumor . unlike gist , if complete removal of a benign glomus tumor ( ro ) is verified histologically , there is no indication for further specific therapy [ 14 , 15 ] . sonographic ( for solid organs ) and endoscopic follow - up for early detection of recurrence or metastasis constitutes the most reasonable postoperative follow - up .

glomus tumor is an infrequent and in most cases benign mesenchymal neoplasia which affects subcutaneous / submucosal tissue and occurs in the gastrointestinal tract , solid organs ( e.g. liver , kidney ) and the extremities . visceral glomus tumor of the stomach generally presents with non - specific epigastric pain , loss of appetite and gi bleeding ( melaena ) , often without haemodynamic instability . macroscopic appearances on upper gi endoscopy are non - diagnostic . endosonographic appearances are generally heterogenous and poorly - reflective , hence fail to differentiate glomus tumor from other potential diagnoses . histological confirmation of the diagnosis is only possible when a fine needle biopsy is inclusive of abnormal tissue . these difficulties in diagnosis mean that in many cases , only immunohistochemical analysis of surgically resected tissue can distinguish glomus tumor from several possible differentials . therefore , endoscopically - assisted laparoscopic curative wedge - resection of a lesion suspicious for glomus tumor of the upper gastrointestinal tract must be considered first - line in terms of a combined investigative and curative approach .

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a 37-year - old man of english descent presented with long - standing photophobia , reduced vision , and retinal pigmentary changes . he subsequently developed lymphoma in the left kidney , treated by nephrectomy and rituximab , and two bowel lymphomas treated with chemotherapy and rituximab . at age 29 , he developed peripheral myopathy when dd was diagnosed on muscle biopsy . he took cyclosporin 100 mg bd , mycophenolate 500 mg bd , ramipril 10 mg daily , amlodipine 10 mg daily , pravastatin 20 mg daily , ezetimibe 10 mg daily , prednisolone 5 mg daily , and aspirin 100 mg daily . on examination , he was highly myopic ; best - corrected snellen visual acuity ( va ) was 20/30 in the right eye and 20/200 in the left eye . fundoscopy revealed midperipheral pigmentary changes , with near - complete loss of rpe in the remaining retina . cirrus spectral - domain optical coherence tomography ( sd - oct ) ( zeiss , germany ) scanning demonstrated asymmetric macular cysts in the inner retinal layers and patchy photoreceptor loss in both maculae [ fig . 1 , table 1 ] . full - field erg testing was performed using a gold foil electrode according to the international society for clinical electrophysiology of vision standards ( lkc technologies , inc . ) . this showed non - specific abnormalities : scotopic responses were of reduced amplitude and normal implicit time ; photopic responses were of normal amplitude but increased implicit time . fundus photographs right eye ( a ) , left eye ( b ) , infrared images right eye ( c ) , left eye ( d ) , and horizontal spectral - domain macular oct scans at baseline right eye ( e ) , left eye ( f ) , and after treatment right eye ( g ) , left eye ( h ) with oral acetazolamide of a patient with histologically confirmed danon disease . these demonstrate the peppered pigmentary mottling and cystic - appearing lesions within the macula with photoreceptor loss and minimal response to treatment retinal thickness and visual function in response to treatment treatment with the carbonic anhydrase inhibitor ( cai ) dorzolamide 2% topically bd to both eyes was initiated . following 30 weeks of treatment , the patient reported no change in central vision or glare symptoms . there was no significant change in vision , central subfield thickness , or macular cube volume . he was then treated with acetazolamide 500 mg daily while cyclosporine levels and renal function were monitored . following 18 weeks of treatment , he reported no change in central vision or glare symptoms . best - corrected va was 20/40 in the right eye and cf in the left eye . central subfield thickness , macular cube volume , and repeat erg testing ( roland , germany ) showed no significant changes . danon retinopathy is a rare x - linked disorder related to mutations in the lamp2 gene . the patient showed characteristic signs of danon retinopathy and his erg results were typical of the disease . the patient was myopic , but the macular changes are not typical of pathologic myopic foveoschisis , typically a thickened retina at the posterior pole , with hyporeflective splitting between the outer and inner retina , or of progressive myopic maculopathy , both of which are usually detected in patients with a posterior staphyloma . patients with retinal dystrophies have been reported to have both cystoid macular edema ( cme ) and non - cme macular cysts . the patient refused fluorescein angiography , which is the diagnostic test to differentiate between cme and non - cme cysts , but the asymmetry of cystic changes seen on oct is typical of cme associated with late hyperfluorescence on intravenous fluoresce in angiography and strongly suggests that the cysts are cme rather than non - cme cysts . the cme is probably due to leakage of fluid through the rpe consequent to failure of the rpe pump . the reason for abnormal lamp2 protein resulting in presumed rpe pump failure is not known . to our knowledge , this is the first reported case of cme in histologically confirmed dd . in our case , topical dorzolamide 2% eye drops and oral acetazomide were not effective in the treatment of cme associated with dd . the reasons for lack of effect may have included low dose of acetazolamide , limited by renal disease ; pre - existing photoreceptor atrophy and long - standing visual loss , particularly in the left eye ; and pre - existing rpe atrophy , given the mechanism of action of cai via rpe acidification which helps to enhance chloride transport , and consequently water transport . also , any non - cme macular cysts may not respond to cais . lack of effect of cai is in contrast to previous studies which have demonstrated a beneficial effect of cais in patients with retinal dystrophy and cme such as retinitis pigmentosa , and patients with macular cysts , including x - linked retinoschisis and enhanced s - cone syndrome , although some patients fail this treatment . despite lack of response in our patient , we recommend consideration be given to carbonic anhydrase inhibition for patients with cme associated with danon retinopathy , given the response seen in other retinal conditions with associated cme and macular cysts with this treatment . a further treatment option to consider for similar patients is intravitreal bevacizumab , which this patient refused . in conclusion , to our knowledge our patient is the first reported case of danon retinopathy with cme .

to report a patient with danon retinopathy with cystoid macular edema treated with topical dorzolamide 2% eye drops and oral acetazolamide . a 37-year - old caucasian man with danon disease treated with topical and oral carbonic anhydrase inhibitors participated in the study . examinations performed before and during treatment included visual acuity ( va ) , spectral - domain optical coherence tomography , and electroretinography . following total 48 weeks of treatment , va decreased from 20/30 od , 20/200 os , to 20/40 od , cf os . the mean central retinal thickness was unchanged from baseline 263 m od , 226 m os , after treatment 283 m od and 202 m os . in our case , carbonic anydrase inhibitors were not effective . however , a general recommendation can not be given based on a single case .

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individuals with a hematologic malignancy may have an immunosuppressive condition and develop cutaneous or invasive bacterial infections . the infections may be a first sign of a malignancy or a sign of a relapse . acute promyelocytic leukemia ( apl ; acute myeloid leukemia m3 ) is caused by balanced reciprocal chromosomal translocation t(15;17 ) , which produces an oncogenic protein pml - rara by fusion of the promyelocytic leukemia gene ( pml ) and the retinoic acid receptor a gene ( rara ) . apl is created by a blockage of differentiation , resulting in overproduction of immature myeloid cells of promyelocytes in the bone marrow . we herein report a 71-year - old man with cellulitis as a first sign of apl . a 71-year - old man was referred to us on october 2011 with a painful eruption on the right thigh that had appeared 12 days earlier . a physical examination revealed a swollen and painful erythematous lesion with an elevated temperature ( 38.6c ) on the right thigh ( fig . laboratory blood examination results were as follows : white blood cell count 1,300/l with neutrophils 18.4% ( band neutrophils 10.0% and segmented neutrophils 8.4% ) , lymphocytes 56.0% , monocytes 0.3% , eosinophils 0% , basophils 0% , metamyelocytes 1.3% and leukemic cells 24.0% ; red blood cells 3.29 10/l ; hemoglobin 11.5 g / dl ; platelets 11.9 10/l ; c - reactive protein 18.526 mg / dl ; soluble interleukin-2 1,249 u / ml ; ferritin 607 ng / ml ; c3 170 mg / dl ; c4 36 mg / dl ; ch50 73.8 u / ml ; fibrinogen 632 mg / dl ; fibrin degradation product 24.1 g / ml ; antithrombin iii 109% ; thrombin - antithrombin iii complex 6.3 ng / ml ; plasmin - a2 plasmin inhibitor complex 3.1 g / ml ; d - dimer 15.8/g / ml , and endotoxin less than 5.0 pg / ml . blood cultures showed no bacterial growth . g - band testing of twenty bone marrow cells showed 46,xy , t(15;17)(q22;q12 ) in 19 cells and 46,xy in 1 cell . all - trans retinoic acid ( atra ) , idarubicin hydrochloride , and cytarabine were administered after the bacterial infection had been eliminated . one month after the administration of atra , a sample of bone marrow cells contained the following : myeloblasts 2.1% ; promyelocytes 3.5% ; myelocytes 21.8% ; metamyelocytes 20.2% ; band neutrophils 12.0% , and segmented neutrophils 5.6% . apl cells can be forced to differentiate in the presence of atra . in this case , a bone marrow examination indicated that the apl cells differentiated into maturing cells after the administration of atra . patients with leukemia are predisposed to pancytopenia , and may show infectious disorders resulting from leukocytopenia , purpura resulting from thrombocytopenia , and shortness of breath resulting from erythrocytopenia . cellulitis may develop secondary to a bacterial infection in patients with leukemia , including apl . cellulitis is an inflammation of loose connective tissue of dermal and subcutaneous tissue , and is caused by bacterial infection . however , cellulitis without leukocytosis may be present in patients with malignant hematologic disorders as shown here , or in persons with cold cellulitis by cutaneous leishmaniasis or leprosy . girmenia et al . reported that their analysis of septicemias in apl patients receiving atra and idarubicin showed a significantly lower rate of bloodstream infections , even though patients receiving atra occasionally suffered life - threatening and lethal infections . the authors noted that 6 of 89 patients with apl withdrew from antileukemic treatment due to infections . infection control before and during the treatment is indispensable for individuals with apl , even though septicemias are less frequent in apl than in other leukemias . dermatologists and hematologists should keep in mind that patients with a hematologic malignancy , such as apl , can develop cellulitis with leukocytopenia .

patients with hematologic malignancies are immunosuppressive and may develop cutaneous or invasive infections as a primary sign of immune suppression . acute promyelocytic leukemia ( acute myeloid leukemia m3 ) is caused by translocation of reciprocal chromosomal rearrangement t(15;17 ) , which produces an oncogenic protein . we herein describe a 71-year - old man having cellulitis with leukocytopenia as a first sign of acute promyelocytic leukemia . dermatologists and hematologists should keep in mind that patients with a hematologic malignancy , such as acute promyelocytic leukemia , can develop cellulitis with leukocytopenia .

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spare parts surgery means use of parts from non - salvageable digits in replantation or cross replantation ( amputated non - replantable digits used to reconstruct thumb ) . it is a well - established procedure in microsurgery . here , we present a case of harvesting double free flaps from an amputated non - replantable lower limb for the contralateral limb . this is based on the gillies principle , which states -do not throw away any tissue . this opportunity does not come very often in the clinical practice and one must be ready to grab it when it does ! to the best of our knowledge , there are a very few reported cases , where double free flaps from the amputated limb or digit has been used for spare parts surgery . a 45 year old male patient who was run over by a train resulting in a right leg amputation at the level of the knee and a crush injury of the left foot . the right lower limb had a severe comminution and bone loss at the knee joint , with the loss of skin and soft- tissue and crushing of muscle above and below the knee [ figures 1 and 2 ] . the left forefoot was completely degloved and all the toes were crushed and degloved as well [ figures 3 and 4 ] . amputated right lower limb right lower limb amputation stump crushed left foot - dorsal aspect crushed left foot - plantar aspect the right lower limb was deemed not replantable as the knee joint was severely damaged and not salvageable , in addition , debridement of crushed and devitalized tissues would result in a 15 - 20 cm shortening and a limb that was at least 15 cm short with fused knee joint would not be functionally useful and primary insertion of prosthetic knee joint was not considered to be feasible by the attending orthopaedic surgeon . focus was then shifted to the crushed left foot with a view to perform immediate debridement and early soft - tissue cover , to salvage as much of the foot as possible and get it fully healed and weight bearing at the earliest . stable and sensate skin cover were vital , as this would be the only surviving foot . a large defect such as this would require a large distant flap or even two flaps . best replacement for skin on the foot was anatomically identical skin from the opposite foot , which in this case was provided by the well - preserved amputated limb . the general condition of the patient was stable and he had no other life - threatening injuries . immediate double free tissue transfer from the amputated limb was done . a plantar flap based on the posterior tibial vessels [ figure 5 ] and a dorsal flap based on the dorsalis pedis vessels [ figure 6 ] , with the communication between the dorsal and plantar systems left intact through the deep branch of the 1 dorsal metatarsal artery . satisfactory inset of dorsal flap [ figure 7 ] , and of plantar flap [ figure 8 ] was achieved . post - operatively the patient developed a hematoma below the dorsal flap . since flaps were harvested from the amputated limb , small blood vessels that could not be seen were probably left unligated . re - exploration and evacuation of hematoma was performed . harvest of flap from plantar aspect harvest of flap from dorsum inset of plantar flap long - term follow - up - dorsal flap long - term follow - up - plantar flap for years surgeons have been amputating limbs at a higher level in limb crush injuries , to ensure good skin and soft - tissue cover for the stump and to make the stump more appropriate for prosthesis fitting . covering the stump with vascularised tissue using the microvascular techniques from another part of the body has now become common . in crush injuries of the foot , very often , early free tissue cover to replace the lost skin and soft - tissue can avoid an amputation . this will however require surgery on another part of the body , with additional trauma and scarring . spare parts surgery , transferring tissue from the amputated limb using microvascular techniques , is one step ahead because it uses tissue that would otherwise have been discarded , without any additional scarring and other such sequelae . since such opportunities are rare and have to be grasped immediately , we need to constantly be aware of this possibility and ready to use it to maximise patient benefit . if the patient is not haemodynamically stable , the harvested flap can be stored by wrapping in a saline soaked gauze in a sterile container and placing the container in a box of ice or cold water as per the same guidelines as for an amputated limb for replantation . recommended ischaemia times for reliable success with spare part surgery are parallel to that recommended in replantation i.e. , 12 h of warm and 24 h of cold ischaemia for harvesting flaps from amputated digits and 6 h of warm and 12 h of cold ischaemia for harvesting flaps from major amputations . use of skin preservation solutions to preserve the skin flaps has been tried in an animal model . furthermore occurrence of haematoma below the flap in spare part surgery is a realistic complication resulting from inability to achieve haemostasis during the harvest of the flap from an amputated achieving haemostasis is then a must after perfusion of the flap lest the flap bleeds after perfusion resulting in a haematoma post - operatively .

bilateral limb trauma poses many possibilities for management . in a situation of bilateral amputation , if the amputated limb is not salvageable or replantation is not advisable , the amputated limb can be used to harvest tissue for free tissue transfer to cover the amputation stump . we describe a case here in which we have used these principles .

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little is known about the relationship between good self - reported health and disability in connection with life- expectancy . to show empirical evidence of the significant statistical association between self - reported health status and disability using spanish survey data and to discuss implications for measures of life expectancy . we model jointly absence of disability and self - reported good - health status correcting for socio - demographic characteristics such as age , gender and years of education . more than 50% of the correlation existing between self - reported health status and disability can not be explained . the proportion of years lived in disability or in good health with respect to the remaining life expectancy increases with age , but longevity is more related to disability than to self - reported bad - health . women report to have good health less frequently than men , while men report disability more frequently . joint factors other than basic socio - demographic indicators induce a significant association between a disability - free status and good self - reported health . life - expectancy in good self - reported health is longer than life - expectancy in a disability - free condition .

introductionlittle is known about the relationship between good self - reported health and disability in connection with life- expectancy.aims and methodsto show empirical evidence of the significant statistical association between self - reported health status and disability using spanish survey data and to discuss implications for measures of life expectancy.resultswe model jointly absence of disability and self - reported good - health status correcting for socio - demographic characteristics such as age , gender and years of education . more than 50% of the correlation existing between self - reported health status and disability can not be explained . the proportion of years lived in disability or in good health with respect to the remaining life expectancy increases with age , but longevity is more related to disability than to self - reported bad - health . women report to have good health less frequently than men , while men report disability more frequently . the influence of the number of years of education is similar for both concepts.conclusionsjoint factors other than basic socio - demographic indicators induce a significant association between a disability - free status and good self - reported health . life - expectancy in good self - reported health is longer than life - expectancy in a disability - free condition .

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hyperpigmentation of the gingiva has always been a concern to certain patients , especially young females . although melanin pigmentation of the gingiva is completely benign and does not present a medical problem , its unaesthetic appearance as black gums is more pronounced in patients having a high smile line ( gummy smile ) . gummy smile can be due to incomplete passive eruption , maxillary protrusion , hyperactive muscle of lips , short lip , gingival enlargement , etc . gingival depigmentation can also be performed by surgical blade , coarse diamond bur , electrosurgery , cryosurgery or lasers . considering the advantages of lasers over other modalities of treatment , this article will focus on the management of such a case using gaalas diode lasers of 810 nm . a 22-year - old female patient was referred with a complaint of gummy smile and hyperpigmented gingiva [ figure 1 ] . on examination , pseudopockets ( approximately 23 mm ) were detected in the maxillary and mandibular anteriors along with generalized excessive amount of melanin pigmentation on the gingiva [ figure 2 ] . the patient was explained about the treatment options and informed that , after depigmentation , melanin pigmentation tends to recur and , as gingivectomy is also performed simultaneously , the overall appearance would be worth doing it . profile view of gummy smile with hyper melanin pigmentation of the gingiva intraoral view of gummy smile with hyper melanin pigmentation of the gingiva gingivectomy and depigmentation were planned in the 1525 region and the 3444 region using a 810 nm diode laser . although procedures like this can be performed under topical anesthesia , infiltration was planned as the patient was anxious and sensitive . under infiltration anesthesia with 2% lignocaine ( with 1:100,000 adrenaline ) , an 810 nm gaalas diode laser ( picasso)*(picasso , by amd lasers ) was used in contact mode with the gingival tissues . e gel was prescribed for local application -three to four times a day for 2 days . because a large area of the raw wound was kept exposed , antibiotic cap . immediate postoperative view of gingivectomy and depigmentation using an 810 nm diode laser the patient reported no adverse effects or discomfort during the postoperative healing phase . she was happy and satisfied with 6-month postoperative results [ figures 4 and 5 ] . gingivectomy and depigmentation were planned in the 1525 region and the 3444 region using a 810 nm diode laser . although procedures like this can be performed under topical anesthesia , infiltration was planned as the patient was anxious and sensitive . under infiltration anesthesia with 2% lignocaine ( with 1:100,000 adrenaline ) , an 810 nm gaalas diode laser ( picasso)*(picasso , by amd lasers ) was used in contact mode with the gingival tissues . e gel was prescribed for local application -three to four times a day for 2 days . because a large area of the raw wound was kept exposed , antibiotic cap . immediate postoperative view of gingivectomy and depigmentation using an 810 nm diode laser the patient reported no adverse effects or discomfort during the postoperative healing phase . she was happy and satisfied with 6-month postoperative results [ figures 4 and 5 ] . diode laser is a versatile laser and has been successfully used at three wavelengths : 980 nm , 810 nm and , more recently , 940 nm . it has numerous soft tissue applications , viz frenectomy , crown lengthening , gingival depigmentation , troughing , etc . in a diode laser , the substance stimulated to produce a coherent light beam is a semiconductor . this technology has produced a laser that is compact and economic . a diode laser has become an important tool in the dental armamentarium due to its exceptional ease of use and affordability . the diode laser is well absorbed by melanin , hemoglobin and other chromophores that are present in periodontal disease . this provides the diode laser with broad clinical utility : it cuts precisely , coagulates , ablates and vaporizes the target tissue with much less trauma , improved postoperative healing and faster recovery time . melanin pigmentation in gingiva is present only when melanin granules synthesized by melanocytes are transferred to the keratinocytes . therefore , recurrence of the pigmentation is inevitable and has been reported from 24 h to 8 years following surgery . conventional procedures for corrections of pigmented gingiva consist of surgical stripping with scalpel , use of electrosurgery , cryosurgery or a coarse diamond bur . however , the laser is the least - invasive and predictable method of performing this procedure . it has a high level of precision , requires minimal anesthesia and causes very low collateral tissue damage . cases where only gingivectomy is required can be performed without anesthesia or under topical anesthesia , but because both gingivectomy and depigmentation were planned together in this patient and a significantly larger area needs to be operated upon , infiltration anesthesia was given . given the incredible ease of use and its versatility in treating soft tissue , the diode laser becomes the patients with complaints of hyperpigmented gingiva should also be assessed for the presence of gummy smile . once the amount of gingiva displayed is reduced by gingivectomy , the patient 's concern for hyperpigmented gingiva will also reduce . as a result , in most of the cases , a repeat surgery for depigmentation

hyperpigmentation of gingiva becomes more pronounced if it is associated with gummy smile . correction of gummy smile and depigmentation together are key to complete patient satisfaction . an 810 nm ( 1.5 w , pulsed ) gaalas diode laser was used to achieve the desired results in a 22-year - old female patient . the 6-month follow - up results showed excellent color and contour of the gingiva . mere depigmentation without correcting gummy smile may look cosmetically good but esthetically unacceptable . diode laser was used as it is known to be an excellent tool as compared with other conventional surgical procedures in terms of patient and operator comfort .

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an hiv - negative male , in his sixties , originally presented with a 6-month history of an isolated large nodule on the right malleolar region which was confirmed as kaposi 's sarcoma ( ks ) on excision . subsequently he continued to develop more papules and plaques on the feet and over the following 7 years underwent excision of around 20 skin lesions under local anaesthesia . more recently he developed , simultaneously , a crop of 5 nodules and a large plaque of ks 5.5 cm in diameter on the dorsum of the right foot with extension to the interdigital spaces and toes ( fig . 1a , fig . chemotherapy with intravenous liposome - encapsulated doxorubicin was not considered because of the significant comorbidities including mild to moderate heart failure . therefore , in the following order , we tried two courses of photodynamic therapy , imiquimod 5% cream applied once daily for 8 weeks and a cycle of anti - herpes therapy based on valganciclovir . it was decided to administer intralesional vinblastine 1 mg / ml diluted 1:5 with saline solution . however , the patient developed side effects such as general malaise , swelling and erythema of the perioral and periocular areas and tingly tongue after the first injection , in keeping with a possible allergic reaction . at that point , at a bit of a loss for options , we switched to doxorubicin . following a serial dilution test from 1:20 to undiluted , this medication was used intradermally at the dose of 2 mg / ml diluted 1:1 with saline solution . the large ks plaque on the dorsum of the right foot was treated by spacing the intradermal injections every 5 mm , using a similar technique to that used in the treatment of axillary hyperhidrosis with botulinum a toxin . the treatment led to complete resolution of the ks lesions after 4 treatments spaced 6 weeks apart . we observed skin necrosis in some well - delimited areas following each injection ( fig . 1b ) , associated with mild to moderate pain which was well controlled with paracetamol 1,000 mg twice daily . the patient has not developed any new lesions for almost 18 months and continues to remain clear ( fig . ks is an angioproliferative disease associated with human herpesvirus 8 . the classic variant of ks is characterised by a low mortality and significant morbidity , and treatment needs to be finely balanced between these two aspects . to date , a curative treatment has yet to be found and therapeutic management includes surgery , radiotherapy , chemotherapy or a combination of all of the above . although in recent years numerous agents such as thalidomide , imatinib , sirolimus as well as matrix metalloproteinase inhibitors ( col-3 ) , anti - angiogenic factors ( bevacizumab ) and nf-b inhibitors ( bortezomib ) have been tried , the outcome of these treatments remains unsatisfactory . liposomal anthracyclines ( pegylated or liposomal daunorubicin or doxorubicin ) and taxanes such as paclitaxel therefore remain the cornerstones of systemic therapy against ks . doxorubicin acts through intercalation of double - stranded dna bases and by inhibiting dna topoisomerase ii whose activity is markedly increased in proliferating cells . it represents a particularly effective treatment for widespread ks as it accumulates in highly vascularised lesions . common agents for intralesional use in ks include interferon alpha , and more recently vincristine and vinblastine . although intralesional doxorubicin is a standard treatment for ks in some centres , there are no publications showing the effectiveness of this treatment in ks . intralesional doxorubicin was very successful in this case and the persistent remission induced would suggest that doxorubicin might be equally or even more effective than other standard intralesional treatments for ks cutaneous lesions in selected cases . further studies with a large cohort of patients could be helpful to clarify the impact of intralesional doxorubicin in ks .

classic kaposi 's sarcoma ( ks ) is a disease with low mortality but high morbidity . the optimum treatment of ks depends upon several factors , including location of lesions , disease progression , severity of symptoms and patient preference . we report the long - term response to the use of intralesional doxorubicin to successfully treat a large cutaneous lesion of ks on a patient refractory to traditional treatments .

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laparoscopic sleeve gastrectomy ( lsg ) is currently one of the most commonly performed bariatric surgeries in the world . known perioperative complications include haemorrhage , leak , small bowel obstruction and infections ( mostly wound and intra - abdominal abscess ) . intra - abdominal abscess presents in < 1% of all procedures , and , to the best of our knowledge , only six cases of splenic abscess as a complication of lsg exist in the literature [ 26 ] . a 45-year - old woman with a past medical history significant for essential hypertension , asthma and morbid obesity status post lsg 20 days before admission , presented with abdominal pain of 10 days duration . the pain was localized to the left upper quadrant and was associated with fever , chills , nausea and infrequent vomiting . at the time of the lsg , her post - operative course was uneventful and she was discharged home 3 days after admission . however , she had not attempted any solid food due to nausea . on admission , physical exam was significant for mild pain on palpation of the left upper abdominal quadrant . a computed tomography ( ct ) of the abdomen was obtained and showed a well - circumscribed 7.1 by 5.4 cm air - fluid collection in the medial aspect of the spleen . of note , an upper gastrointestinal series with gastrografin showed no evidence of leak from her gastrectomy staple line . the patient was started on empiric piperacillin / tazobactam and a ct - guided drainage of the abscess was performed . the patient was treated with meropenem and linezolid for 4 days , and then switched to intravenous ceftriaxone once the microbiology results became available . she was discharged home to complete 2 weeks of intravenous antibiotics . however , after 3 weeks the patient returned to the hospital with new left upper abdominal pain that referred to the left shoulder , poor oral intake , fever and shortness of breath . a repeat ct of the abdomen showed an air - fluid collection in the medial spleen comparable in size to the one present at post drainage . a ct - guided drainage of the splenic abscess was performed and the patient was discharged home to complete 2 weeks of intravenous ceftriaxone and oral metronidazol . splenic abscess is a rare occurrence , with the vast majority of the available literature consisting of case reports and series from tertiary care centres . splenic abscess as a complication of lsg appears to be an extremely rare entity with only six cases in the literature [ 26 ] . these patients were ages 1946 , none had a reported immunosuppressive condition , presented in the late post - operative period , and only one had an intraoperative complication ( table 1 ) . previous authors have pointed out that splenic abscess in the bariatric population could have a different aetiology from the classically described in the general population . among the proposed factors are extension from a gastric staple - line leak , splenic injury during surgery and inadvertent splenic ischaemia [ 3 , 6 ] . regarding the latter , in a recently published series of 565 lsg , 0.53% presented with splenic abscess in post - operative days 914 . interestingly , the authors of this paper reviewed the video recordings of all cases and reported a rate of spleen infarcts of 7.79% . all splenic abscesses in this series had evidence of infarct during the original procedure , leading them to suggest that they are both related . a similar study with closer follow - up suggested this as well . in our case , there was no evidence of intraoperative injury to the spleen , staple - line leak or splenic ischaemia . the causative agent was s. anginosus , usually found as a commensal of the oral cavity and gastrointestinal tract , but capable of suppurative infections that are often associated with antecedent disruption of the gastrointestinal mucosa . in the present patient , without any obvious perforation or anastomotic leak , haematogenous spread caused by transient bacteraemia during the surgical procedure is certainly a possibility . she responded well to the combination of intravenous antibiotics and percutaneous drainage , and there was no need for splenectomy . table 1characteristics of previous case reports of splenic abscess as a complication of lsg.case ( reference)age / seximmunosuppressionimmediate complicationspost - operative day of presentationtreatmentevidence of leakagecultured organismrojas et al . 46/fnohaemoperitoneum , splenic hilum and hepatic injury14iv antibiotics , percutaneous drainageyess . no75iv antibiotics , percutaneous and laparoscopic drainagenostaphylococcusspp.,enterobacter cloacae , streptococcus mitis and oralisavulov et al . 44/mnono70iv antibiotics , percutaneous drainage , splenectomynoklebsiella pneumoniae , streptococcus pneumoniae , acinetobacterspp.current study , 201645/fnono20iv antibiotics , percutaneous drainagenos . anginosus characteristics of previous case reports of splenic abscess as a complication of lsg . in conclusion , splenic abscess is an uncommon complication of lsg , albeit one that should be entertained in the patient with abdominal pain and fever given its high mortality . a ct of the abdomen with contrast is the preferred diagnostic tool ; patients should be treated with a combination of intravenous antibiotics and either percutaneous drainage or splenectomy . further studies with a larger sample of lsg patients should be conducted in order to elucidate the role of splenic ischaemia in abscess formation .

abstractsplenic abscess as a complication of laparoscopic sleeve gastrectomy ( lsg ) is rare . there have only been six cases in the literature . in most of these cases , the classic predisposing factors for developing splenic abscess were absent , leading to the hypothesis that transient bacteraemia caused by mucosal disruption during the surgical procedure and splenic ischaemia may play a role . these patients usually present in the late post - operative period with abdominal pain , fever and leucocytosis . the preferred treatment is intravenous antibiotics and percutaneous drainage or splenectomy . we report a case of splenic abscess caused by streptococcus anginosus that occurred 20 days after lsg in a 45-year - old woman without immunosuppressive conditions . the patient was successfully treated with antibiotic therapy and percutaneous drainage .

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besides its classical role in calcium and bone homeostasis , vitamin d is considered a potent immunomodulator that can affect the pathogenesis of several autoimmune diseases . our aim is to evaluate the effect of vitamin d correction to a patient with new onset graves disease ( gd ) with an underlying vitamin d deficiency . we describe the effect of vitamin d3 on untreated graves disease with vitamin d deficiency . a healthy saudi woman in her 40s sought consultation with a three - month history of palpitation . she denied any history of heat intolerance , weight loss , menstrual irregularity or sweating . . physical examination revealed a mild diffusely enlarged and non - tender thyroid gland with no bruit . the initial thyroid function test , which was done in an outside hospital , revealed a tsh , 0.01 miu / l ; ft4 , 22.5 vitamin d 25-oh level was done in our hospital and showed a result of 26.0 nmol / l with a tsh , 0.013 miu / l ; ft4 , 16.7 tc-99 m thyroid scintigraphy demonstrated an enlarged thyroid gland with increased radiotracer trapping and heterogeneous distribution . the patient was given only oral cholecalciferol 4000 iu per day since november 2012 ( prescribed by an outside hospital ) then from may 2013 onwards she was given 50,000 iu per month . follow - up laboratory exams revealed improved vitamin d levels as well as tsh and ft4 . vitamin d deficiency may exacerbate the onset and/or development of gd and correction of the deficiency may be able to reverse it . however , further prospective clinical studies will be needed to define the role of vitamin d treatment in gd . more recently , vitamin d has been shown to be a modulator in both innate and adaptive immunity.1 there is a well - established link between vitamin d deficiency and various autoimmune diseases , including type 1 diabetes mellitus ( t1 dm ) , systemic lupus erythematosus ( sle ) , rheumatoid arthritis ( ra ) , inflammatory bowel disease ( ibd ) , and multiple sclerosis ( ms ) . furthermore , it has been found that the supplementation of vitamin d can prevent the onset and/or development of different kinds of autoimmune disorders in human beings and animal models.2 in addition , it has been shown that the prevalence of vitamin d deficiency is common in patients with graves disease ( gd),3 and is associated with higher thyroid volume.4 in our case report , we evaluated the effect of vitamin d correction to a patient with new onset gd with an underlying vitamin d deficiency . a healthy saudi woman in her 40s sought consultation with a 3 months history of palpitation . she denied any history of heat intolerance , weight loss , menstrual irregularity , diarrhea , or sweating . there was no personal or family history of thyroid disease and no specific medication history . physical examination revealed a mild diffusely enlarged and non - tender thyroid gland with no bruit . the initial thyroid function test , which was done in an outside hospital , revealed a tsh , 0.01 miu / l ; ft4 , 22.5 vitamin d 25-oh level was done in our hospital and showed a result of 26.0 nmol / l with a tsh , 0.013 miu / l ; ft4 , 16.7 anti - thyroid antibodies showed a tg , 17.1 iu / ml ; tpo , 0.19 iu / ml with a positive tsh receptor antibody . tc-99 m thyroid scintigraphy demonstrated an enlarged thyroid gland with increased radiotracer trapping and heterogeneous distribution ( fig . the patient was given only oral cholecalciferol 4000 iu per day since november 2012 ( took it from an outside hospital ) then from may 2013 onwards she was given 50,000 iu per month . the serial thyroid function tests , vitamin d levels , and titer autoantibodies are summarized in table 1 . follow - up laboratory exams revealed improved vitamin d levels as well as tsh and ft4 . written informed consent was obtained from the patient for the publication of this case and accompanying images . it has become apparent that multiple factors contribute to the etiology of gd , including genetic and environmental factors . these activated t cells in turn increase the secretion of thyroid - specific autoantibodies from b cells . the prevalence of vitamin d deficiency was reported to be common in patients with gd.3 whether vitamin d deficiency has a causal relationship with gd remains a controversial issue . misharin et al.5 observed that vitamin d deficiency was found to modulate graves hyperthyroidism induced in balb / c mice by thyrotropin receptor immunization . in this study , balb / c mice on a vitamin d deficient diet were more likely to develop persistent hyperthyroidism than other mice receiving adequate vitamin d supply . in another study , combination treatment with methimazole and vitamin d3 ( 1,25 ( oh)2d ) in patients with gd has more rapid euthyroidism achievement compared with patients receiving methimazole alone.6 in addition , vitamin d supplementation has been shown to inhibit inflammatory responses in human thyroid and t cells.7 interestingly , vitamin d deficiency is found to be associated with higher thyroid volume in patients with newly onset gd.4 it has been recently discovered that vitamin d - receptor gene and vitamin d - binding protein gene polymorphisms are associated with gd.8,9 our present case supports the current literature and strongly suggests that vitamin d deficiency may exacerbate the onset and/or development of gd and correction of which may be able to reverse it . however , further prospective clinical studies will be needed to define the role of vitamin d treatment in gd .

objectivebesides its classical role in calcium and bone homeostasis , vitamin d is considered a potent immunomodulator that can affect the pathogenesis of several autoimmune diseases . our aim is to evaluate the effect of vitamin d correction to a patient with new onset graves disease ( gd ) with an underlying vitamin d deficiency.methodwe describe the effect of vitamin d3 on untreated graves disease with vitamin d deficiency.resultsa healthy saudi woman in her 40s sought consultation with a three - month history of palpitation . she denied any history of heat intolerance , weight loss , menstrual irregularity or sweating . she has a history of chronic muscle aches and pains . physical examination revealed a mild diffusely enlarged and non - tender thyroid gland with no bruit . she had no signs of graves ophthalmopathy . in laboratory examinations , the initial thyroid function test , which was done in an outside hospital , revealed a tsh , 0.01 miu / l ; ft4 , 22.5 pmol / l and ft3 , 6.5 pmol / l . vitamin d 25-oh level was done in our hospital and showed a result of 26.0 nmol / l with a tsh , 0.013 miu / l ; ft4 , 16.7 pmol / l ; and ft3 , 3.8 pmol / l . tsh receptor antibody was positive . tc-99 m thyroid scintigraphy demonstrated an enlarged thyroid gland with increased radiotracer trapping and heterogeneous distribution . the patient was given only oral cholecalciferol 4000 iu per day since november 2012 ( prescribed by an outside hospital ) then from may 2013 onwards she was given 50,000 iu per month . follow - up laboratory exams revealed improved vitamin d levels as well as tsh and ft4 . she eventually improved both clinically and biochemically with a satisfactory outcome.conclusionvitamin d deficiency may exacerbate the onset and/or development of gd and correction of the deficiency may be able to reverse it . however , further prospective clinical studies will be needed to define the role of vitamin d treatment in gd .

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melanoma is the malignant tumor of melanocytes which may present in a wide morphological spectrum including highly pigmented to amelanotic appearance . clinically there are four main subtypes of cutaneous melanoma : superficial spreading melanoma , lentiginous melanoma , nodular melanoma and acral lentiginous melanoma . the diagnosis of amelanotic variants of melanoma may especially be challenging both clinically and histologically . in addition to clinical variants of melanoma , in the literature various atypical histological variants of melanoma have been reported , such as fibroblastic , desmoplastic , chondroid , osteoid , and myxoid melanoma , which were classified according to stromal changes [ 13 ] . myxoid melanoma is an unusual variant of malignant melanoma , which is characterized by atypical spindle cells and dense mucin deposition in dermis . the prevalence of this melanoma variant is not well known and may develop on cutaneous or extracutaneous sites , including the sino - nasal passages . this tumor may be confused with other mucin - containing neoplasms , histologically benign or malignant , and clinically is usually reported in elderly people with a similar progress to other variants of melanoma . a 28-year - old male presented to our outpatient clinic with a history of an asymptomatic pink nodule which had been growing slowly for last 6 months . dermatological examination revealed a 2.5 x 2 cm diameter pink , mildly infiltrated tumoral lesion with two pigmented papular lesions on left arm ( figure 1 ) . dermoscopic examination revealed pink - white cristalline structures and blue - grayish ovoid globules ( figure 2 ) . the lesion was totally excised with 3 mm margins with the initial diagnosis of basosquamous carcinoma , amelanotic melanoma and basal cell carcinoma . histopathological examination was consistent with myxoid melanoma with breslow thickness 11.6 mm , clark level v ( figure 3a , b ) . there was no vascular , lymphatic or perineural invasion histologically , and mitoses 2/1 per mm . strong positivity was detected with s100 and hmb45 staining , and widespread positive staining was detected with pas - alcian blue for mucin deposition ( figure 4a , b ) . laboratory tests , including complete blood count , routine biochemistry , lactate dehydrogenase and beta-2 microglobulin and imaging test for metastasis , including pet - ct and sentinel lymph node biopsy , were all clear . the patient is currently well and clinically free of recurrence 18 months after the diagnosis . the presence of myxoid stroma in malignant melanoma was first published by bhuta et al in 1986 in four metastatic malignant melanomas . clinically these tumors were reported to be amelanotic , but in some cases melanogenesis was also shown with fontana masson preparations . histologically , this rare variant of melanoma is characterized by large malignant melanocytes and a basophilic mucinous matrix . in all cases , the myxoid stroma is comprised of mesenchymal acidic mucopolysaccharides , as opposed to neutral epithelial mucins . mucinous material is usually located around the tumor cells , as in our case , but not within the tumor cells as in cytoplasmic localization , confirming that the myxoid matrix is produced as a response to the stromal cells in the tumor rather than being a product of the tumor cells . myxoid changes are more often reported in metastatic tumors than in primary malignant tumors , but in our patient due to the completely intradermal location of tumor cells with no junctional component , ith a nodular architecture , features of malignancy , no ulceration , the absence of peripheral nerves , and an absence of preexisting melanocytic nevus , he was diagnosed as primary dermal melanoma . additionally , based on the histopathologically myxoid stromal changes , he was diagnosed as primary myxoid melanoma . also no other metastatic or primary malignant lesion typically , s100 staining is strongly positive , but immunostaining with hmb-45 is less uniform and both positive and negative results were reported in the literature . in our patient both s100 and hmb45 staining were strongly positive . the differential diagnosis of myxoid melanoma is broad , including several other benign and malignant myxoid neoplasms of soft tissue as well as epithelial cancers such as myxoid liposarcoma , myxoid malignant fibrous histiocytoma , low - grade fibromyxoid sarcoma , myxoid chondrosarcoma , myxoid peripheral nerve sheath tumors , dermatofibrosarcoma protuberans and metastatic adenocarcinomas . the clinical and the prognostic significance of mucin deposition in melanomas is challenging due to presence of myxoid material both in primary benign or malignant tumors . some authors claimed that mast cells and secretion of transforming growth factor beta stimulates fibroblast secretion of mucin contributing to the tumor s invasive potential ; however , in the absence of compelling contrary data , the current series of cases suggests that myxoid stroma is more significant in diagnosis rather than prognosis of myxoid melanoma . although the real importance of myxoid changes in tumors is not well known , awareness of this stromal pattern in malignant melanomas may prevent misdiagnosis and therapeutic errors .

myxoid melanoma is a rare variant of melanoma , which is characterized by atypical spindle cells and dense mucin deposition in dermis . this tumor is usually seen in elderly people with a similar progress in other variants of melanoma.a 28-year - old male presented to our outpatient clinic with a 6-month history of a slowly growing asymptomatic pink lesion on his arm . dermoscopic examination revealed pink - white cristalline structures and blue - grayish ovoid globules . the lesion was totally excised with initial diagnosis of basosquamous carcinoma , amelanotic melanoma and basal cell carcinoma . histopathological examination was consistent with myxoid melanoma.we present this case due to the rarity of myxoid melanoma and occurrence at such a young age .

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unilateral absence of pulmonary artery ( uapa ) is a rare congenital abnormality , with an estimated prevalence of 1 in 200,000 . some patients with uapa are totally asymptomatic while others may have severe pulmonary hypertension , hemoptysis , congestive heart failure , and cyanosis . we report a 2.5-year - old girl with congenital absence of the right pulmonary artery with associated congenital cystic adenomatoid malformation ( ccam ) of the right lower lobe , patent ductus arteriosus ( pda ) , and atrial septal defect ( asd ) , who presented with ortner 's syndrome due to severe pulmonary hypertension . a 2.5-year - old girl presented with hoarseness of voice noticed since 3 months of age , breathlessness for the past 15 days and failure to thrive . she had been admitted elsewhere for lower respiratory tract infection at 3 months of age . the child was noticed to have a weak cry and an exaggerated suck - rest - suck cycle and sweating over the forehead while feeding . pulse oximetry in room air showed an oxygen saturation varying between 60% and 70% in all four limbs . weight and height were 7 kg and 77 cms , respectively both below the third percentile for age and sex . an ejection systolic murmur grade 3 was heard in the right parasternal region in the fourth and fifth intercostal spaces . the liver was palpable 2 cm below the right costal margin in the mid - clavicular line . chest x - ray [ figure 1 ] revealed cardiomegaly with a cardio - thoracic ratio of 0.6 and deviation of the trachea and mediastinum to the right . two - dimensional ( 2d ) echocardiography showed decreased flow in the right pulmonary artery , a small asd with a right - to - left shunt , and a dilated right atrium and right ventricle with severe tricuspid regurgitation , suggestive of severe pulmonary hypertension . multiple detector computerized tomography aortogram [ figure 2 ] confirmed the findings of asd with pulmonary hypertension , absent right pulmonary artery and hypoplastic right lung with small cystic lesions suggestive of ccam in the right lower lobe . chest x - ray posterioranterior view showing cardiomegaly and shift of trachea and mediastinum to the right multiple detector computerized tomography aortogram showing absent right pulmonary artery , and cystic lesions in the right lower lobe suggestive of congenital cystic adenomatoid malformation ( arrow ) of lung at admission , the patient was started on oxygen , furosemide , enalapril and intravenous cefotaxime , on a provisional diagnosis of congenital cyanotic heart disease with increased pulmonary blood flow , lower respiratory infection , and congestive cardiac failure . despite these measures , there was persistent hypoxemia and worsening of cardiac failure in the form of tachycardia and bilateral basal crepitations . the child was intubated and ventilated and started on pressors , but sustained a cardiac arrest on the 4 hospital day from which she could not be resuscitated . complete arrest of pulmonary artery supply results in the arrest of early bronchial development leading to agenesis of the affected lobe or segment . pulmonary agenesis is usually unilateral , right sided absence of pulmonary artery being more common . more than 50% of children with pulmonary agenesis have associated congenital anomalies that involve the cardiovascular ( pda and patent foramen ovale ) , gastrointestinal , skeletal , and genitourinary systems . our patient had asd and pda . while some patients with uapa are totally asymptomatic , others may have severe pulmonary hypertension , hemoptysis , congestive heart failure , and cyanosis . chest x - ray may show an absent hilar shadow , a shrunken affected lung , and shift of the mediastinum to the affected side . an early interruption in the development of the pulmonary artery could result in the continued development of the primitive capillary supply to a region of the lung with resultant abnormal development of the supplied region . the magnitude of the insult would determine the exact development of the affected lung tissue and final blood supply , resulting in pulmonary sequestration , ccam , or a combination of the two lesions . the incidence of pulmonary hypertension in patients with isolated uapa varies between 18% and 44% . in patients with uapa with pda as in our patient , the incidence of pulmonary hypertension was observed to be as high as 86% , those with pulmonary hypertension dying at an early age . ortner syndrome or cardiovocal syndrome refers to hoarseness of voice due to recurrent laryngeal nerve paralysis secondary to cardiovascular disease . this syndrome was first described by ortner in 1897 in two patients who had mitral stenosis and left recurrent laryngeal nerve paralysis . the syndrome has since been described in adults with various cardiovascular disorders , but reports in children are less common . the pulmonary artery , enlarged due to pulmonary hypertension , has been implicated as the main mechanism of nerve injury . fetterolf and norris studied the anatomic relations of the left recurrent laryngeal nerve in cadavers and concluded that the nerve must be squeezed between the enlarged left pulmonary artery and the aorta or ligamentum arteriosum . unilateral absence of pulmonary artery should be considered in the differential diagnosis of children who present with cyanosis , pulmonary hypertension , or recurrent lower respiratory tract infections .

we report a 2.5-year - old girl who presented with hoarseness of voice since 3 months of age and failure to thrive . chest x - ray showed cardiomegaly with a deviation of the trachea and mediastinum to the right side . two - dimensional echocardiography showed decreased flow across the right pulmonary artery , a small atrial septal defect ( asd ) with a right - to - left shunt , and a dilated right atrium and right ventricle with severe tricuspid regurgitation suggestive of severe pulmonary hypertension . a silent large patent ductus arteriosus was also seen . multiple detector computerized tomography aortogram confirmed the findings of absent right pulmonary artery and hypoplastic right lung with small cystic lesions suggestive of congenital cystic adenomatoid malformation in the right lower lobe . hoarseness of voice was due to the left vocal cord palsy probably secondary to severe pulmonary hypertension ( ortner 's syndrome ) .

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stevens johnson syndrome ( sjs ) is thought to be a hypersensitivity complex affecting the skin and the mucous membranes . a new anticonvulsant , oxcarbazepine , which is structurally related to carbamazepine ( cbz ) , was introduced for use in patients with epilepsy . drug hypersensitivity reactions can occur with most drugs , although the frequency , severity , and clinical manifestations vary . drug hypersensitivity can be defined as an inappropriate immune response leading to tissue damage from an otherwise nontoxic agent . drug hypersensitivity reactions to benzodiazepines often fall into the category of type b ( or bizarre ) adverse drug reactions , according to the classification proposed by rawlins and thompson . the incidence of hypersensitivity varies according to the drug , the disease being treated , and the ethnicity of the patient . drug response ( including drug hypersensitivity ) is a multifactorial and multigenic process , dependent on a complex interaction between multiple genes and the environment [ figure 1 ] . each gene contributes to the risk of developing the hypersensitivity reaction , but each individual gene is neither necessary nor sufficient by itself to cause the reaction . a female patient of 38 years with a history of drug allergy was complaining of fever , sore throat , and fatigue , at the time of admission . she was administered oxcarbazepine for the management of right partial bronchial seizure due to left parasagittal mass lesion following which she developed papular rashes all over the body and diagnosed as sjs . in this case , during the first week , we used 600 mg / day oxcarbazepine for seizure control , and then increased the dose after 3 days to 900 mg / day . after 10 days of treatment , ulcers and other lesions begin to appear in the mouth and lips along with the genital and anal regions . ulcers in the mouth are usually extremely painful and reduce the patient 's ability to eat or drink . high fever and multiple maculopapular rashes were found over the patient 's face and neck initially on the 12 day after taking oxcarbazepine . she was brought to our emergency department and admitted under the presumed diagnosis of sjs . laboratory investigations showed leukocytosis ( wbc , 14660/l ; reference value , 4,000 - 10,000/l ) , and elevated c - reactive protein ( 59.30 g / ml ; reference range , 0 - 5 g / ml ) . after obtaining informed consent , we carried out genotyping and took photos of the patient . there was marked liquefactive degeneration in the lower half of the epidermis with some dyskeratotic keratinocytes . the dermis showed predominant cd8 + lymphohistiocytic infiltration around the blood vessels and scanty eosinophils . after corticosteroids ( iv dexamethasone 1.0 mg / kg body weight ) and antihistamine treatment for 10 days , the patient improved and was discharged from the hospital . anticonvulsant hypersensitivity syndrome is a potentially fatal drug reaction with cutaneous and systemic reactions ( incidence , 1 in 1000 to 1 in 10,000 exposures ) to the arene oxide - producing anticonvulsants phenytoin , cbz , and phenobarbital sodium . cbz and its derivatives are widely used anticonvulsants that can cause rashes in up to 10% of patients , and in occasional cases , this may be the precursor to the development of a hypersensitivity syndrome characterized by systemic manifestations such as fever and eosinophilia . the diagnosis of sjs is based on clinical manifestations with acute onset of rapidly expanding targetoid erythematous macules , necrosis and detachment of the epidermis along with erythema , erosions , and crusting of two or more mucosal surfaces . our patient had skin targetoid erythematous rashes and mucosa involvement 2 weeks after starting oxcarbazepine treatment . during these 2 weeks the skin pathology finding revealed lymphohistiocytic infiltration around the blood vessels and scanty eosinophils , which was consistent with sjs . the patients usually develop a hypersensitivity reaction between 2 and 12 weeks after starting medicine . it has been postulated that metabolites and not the parent drug are the causal agents . one of the first reports showed that hla - b*1502 was present in 100% of cbz - induced patients with sjs but in only 3% of patients tolerating cbz and in 9% of the general population . other studies have confirmed these results in han chinese and in the thai population . in this case , we used a dose of 600 mg / day and then titrated it to 900 mg / day . it has been reported that higher daily doses of drugs are associated with an increased risk of sjs than lower doses , which is the case for allopurinol . however , lam et al . found that the early use of short - term systemic steroids for 35 days lacked any significant side effects and did not increase mortality or morbidity . no sequels were found during 3 months of follow - up . compared with other categories of drugs , such as antibiotics and nsaids , antiepileptic therapies are associated with a high incidence of sjs and toxic epidermal necrolysis ( ten ) . case reports by chen et al . suggest that identification of such genetic factors is important , not only to realize the prospect of developing preventive strategies but also to learn about the mechanisms of these reactions , which may ultimately lead to other preventive strategies through better drug design and to better treatment strategies for patients who develop the reactions .

stevens johnson syndrome ( sjs ) is a rare immunologic reaction that may involve skin or various mucosal surfaces . the etiology may range from multiple pharmacologic agents to viral infections . associated findings can range from minimal skin and mucosal involvement to extensive dermal exfoliation , nephritis , lymphadenopathy , hepatitis , and multiple serologic abnormalities . we report a female patient of 38 years with a history of drug allergy who was administered oxcarbazepine for the management of right partial bronchial seizure due to left parasagittal mass lesion following which she developed papular rashes all over the body and diagnosed as sjs . although carbamazepine ( cbz ) is the most common cause of sjs , a new anticonvulsant , oxcarbazepine , which is structurally related to cbz , has been shown to induce sjs .

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therapeutic hypothermia has been shown to provide neuroprotection against ischemic injury after cardiac arrest in in vitro and in vivo models . in the previous issue of critical care , meybohm and colleagues demonstrate that cardiac arrest triggers the release of cerebral inflammatory cytokines in pigs ' cerebral cortex . . the combination of hypothermia with sevoflurane post - conditioning does not confer additional anti - inflammatory effects compared with hypothermia alone . cardiac arrest remains the leading cause of death in the us and europe , with an out - of - hospital cardiac arrest survival - to - discharge rate of less than 10% . in - hospital cardiac arrest presents a dismal prognosis . according to a large in - hospital registry , the survival - to - discharge rate is 18% , whereas that of a developing country is 6.9% . without prompt care when immediate care is available and victims are successfully resuscitated , the majority of these initial survivors subsequently suffer crippling neurologic injury or die in the few days following the cardiac arrest event . thus , improving survival and brain function after initial resuscitation from cardiac arrest remains a critical challenge . therapeutic hypothermia , introduced more than six decades ago , remains an important neuroprotective factor in cardiac arrest . laboratory studies have demonstrated that cooling after resuscitation from cardiac arrest improves both survival as well as subsequent neurologic and cardiac function and has few side effects . these findings have been reproduced using a variety of cooling techniques in different species , including rats , dogs , and pigs . however , physician use of hypothermia induction in patients resuscitated from cardiac arrest is low . in 2003 , abella and colleagues reported that 87% of us physicians did not use therapeutic hypothermia following cardiac arrest . various reasons for non - use were cited : 49% felt that there were not enough data , 32% mentioned lack of incorporation of hypothermia into advanced cardiovascular life support protocols , and 28% felt that cooling methods were technically too difficult or too slow . in 2002 , a european group demonstrated an improvement in survival - to - discharge rate with favorable neurologic status in cooled patients , compared with normothermic patients surviving after cardiac arrest ( 53% versus 35% , respectively ) , and with no significant adverse events from cooling ; thereafter , induced hypothermia was considered the best practice for patients following cardiac arrest . in 2005 , the american heart association recommended the consideration of therapeutic hypothermia for unconscious adult patients with return of spontaneous circulation following out - of - hospital cardiac arrest due to ventricular fibrillation . in 2008 , binks and colleagues reported that 85.6% of intensive care units in the uk were using hypothermia as part of post - cardiac arrest management . clinical observation demonstrated that tumor necrosis factor - alpha ( tnf ) and interleukin-6 ( il-6 ) protein were increased in cerebrospinal fluid following cardiac arrest . animal studies showed that inflammatory markers were unregulated in rats ' hippocampus tissue and pigs ' serum and myocardial tissue after cardiac arrest [ 8 - 10 ] . meybohm and colleagues go further to demonstrate anti - inflammatory and anti - apoptosis effects of therapeutic hypothermia via the reduction of the upregulation expression of il-1 , il-6 , il-10 , tnf and intercellular adhesion molecule-1 , bcl-2 , and bax mrna and il-1 protein in cerebral cortex after cardiac arrest in a pig model . small reductions in core temperature lead to vaso - constriction and shivering , effectively hindering hypothermia . thus , prevention of vasoconstriction and shivering has become a major goal during induction of therapeutic hypothermia . sevoflurane pre - conditioning and early post - conditioning reduced both cerebral infarct size and neurological defect score , reduced impairment of hippocampus long - term potentiation resulting from myocardial ischemia , and increased nuclear factor inhibitory kappabalpha content in thp-1 cells [ 11 - 13 ] . sevoflurane pre - conditioning preserves myocardial function in patients undergoing coronary artery bypass graft surgery under cardiologic arrest . an in vivo study showed that combination hypothermia with sevoflurane attenuates the inflammatory response during endotoxemia . however , meybohm and colleagues could not provide evidence to support the view that sevoflurane post - conditioning confers additional anti - inflammatory effects in pigs ' cerebral cortex after cardio - pulmonary resuscitation . in summary , meybohm and colleagues provide useful evidence to support the clinical use of therapeutic hypothermia for cardiac arrest , but they did not study the anti - inflammatory effects of sevoflurane in this model . it is even possible that in the setting of clinical practice , anesthetics may not provide significant neuroprotection beyond that which is already being produced by therapeutic hypothermia . thus , at this time , it is difficult to recommend anesthetics for the purpose of neuroprotection in cardiac arrest .

in the previous issue of critical care , meybohm and colleagues provide evidence to support hypothermia as a kind of therapeutic option for patients suffering cardiac arrest . although anesthetics had been used to induce hypothermia , sevoflurane post - conditioning fails to confer additional anti - inflammatory effects after cardiac arrest . further research in this area is warranted .

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formulations were prepared using different polymers kollidone sr , cellulose acetate , acrycoat s 100 , methocel k4 m , methocel k15 m , methocel k100 m . drug and polymer in proportion 1:2 , ( drug : polymer ) were dissolved in organic solvent ( ethanol and acetone ) . this clear solution was poured slowly as a thin stream in oil phase ; about 100 ml of liquid paraffin solution with continuous stirring at a speed of 500 rpm using mechanical stirrer at room temperature until complete evaporation of solvent took place . the floating microspheres were collected by decantation , while the non floating microspheres were discarded along with any polymer precipitates . several preformulation trials were undertaken for various proportions of drug and polymer by variation of the ethyl acetate - acetone ratio and dichloromethane - ethanol ratio . kollidone sr , acrycoat s 100 and cellulose acetate were selected as matrixing agent considering its widespread applicability and excellent gelling activity in sustain release formulations and also having the ph - independent and reproducible drug release profile . it was found that kollidone sr microspheres show desirable high drug content , yield , floatation and adequate release characteristics and hence was suitable for development of a controlled release system . in the present study , in vitro release studies of the floating microspheres were carried out in 0.1 n hydrochloric acid at 37c for a maximum period of 12 hours . at different time intervals , samples were withdrawn and cumulative % drug release was calculated . the percentage drug release of all the formulations is presented in figure 1 . out of 9 formulations tried , the formulation k1 was found to be satisfactory ; since it showed prolonged and complete release with 94.75% at end of 12 h. % drug release of all nine formulations multi unit gastroretentive drug delivery system has additional advantage of absence of dose dumping as in single unit drug delivery . the present investigation described the influence of the drug : polymer ratio on hydrochlorothiazide release . the release and drug entrapment efficiency of the microspheres were affected by the different polymers . it was found that the kollidone sr had a dominant role in the drug release from microspheres rather than acrycoat s 100 and cellulose acetate . and it can be given in hard gelatin capsule form . therefore , it may be concluded that drug loaded floating microspheres in combination with kollidone sr are a suitable drug delivery system for hydrochlorothiazide .

the present investigation described the influence of viscosity and drug : polymer ratio on hydrochlorothiazide release . floating microspheres loaded with hydrochlorothiazide were prepared by emulsion solvent evaporation method . the prepared microspheres were evaluated by micromeritics properties , in vitro drug release , floating ability and drug entrapment efficiency .

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the purpose of bone scintigraphy is to portray areas of new bone formation within the skeleton . this is useful in imaging reaction of bone to tumor , fracture , and infection . since approximately half of the administered radioisotope is excreted through renal filtration , abnormalities of the urinary system are also frequently noted during bone scintigraphy . in such cases , to reach an accurate diagnosis , the interpreting physician must first recognize which structures are involved in the uptake and the significance of the uptake . we describe a rare case in which extraosseous bone scan tracer accumulation was noted in a renal calculus . a 60-year - old man , who had difficulty in passing urine for the past 6 months and a complaint of lower back pain presented to our department . on ultrasound imaging , he was found to have an enlarged prostate . transrectal ultrasound - guided biopsy revealed adenocarcinoma of prostate , gleason 's score 3 + 3=6 . his psa was normal ( 1.3 ng / ml ) and serum alkaline phosphatase was elevated 58 u / l ( normal range 30 - 50 u / l ) . a 99 m technitium methylene diphosphonate ( 99 m tc - mdp ) bone scan was performed . the scan [ figures 1 and 2 ] showed mildly increased tracer uptake in lumbar vertebrae and focal accumulation of tracer in lower pole of the left kidney . fusion imaging [ figure 3 ] , using single photon emission computed tomography along with x - ray computed tomography ( spect - ct ) of the lumbar spine was performed to characterize the vertebral tracer uptake . lasix 40 mg was given 45 minutes prior to spect - ct to monitor the renal tracer accumulation . high tracer uptake in a lower pole of the left kidney was found localized in a calculus in the lower calyx of kidney . the ct attenuation factor was 1060 hounsfield unit , compatible with that of a renal calculus . ( a ) anterior and ( b ) posterior views , showing mildly increased tracer uptake in the lumbar vertebrae ( black arrow ) and intense , focal localization of the tracer in the lower pole of left kidney ( red arrows ) . ( a - b ) lateral and ( c - d ) oblique views clearly demonstrate the focal tracer uptake outside the skeletal structures in left kidney ( arrows ) . ( a ) coronal , ( b ) sagittal and ( c ) transaxial ct images ; ( d ) coronal ( e ) sagittal and ( f ) transaxial spect images ; ( g ) coronal , ( h ) sagittal and ( i ) transaxial spect - ct images . the ct attenuation factor was 1060 hounsfield unit , compatible with that of a renal calculus . we concluded this as extra - skeletal bone scintigraphy tracer uptake in a renal calculus . bone scintigraphy is a valuable diagnostic tool in the evaluation of patients with a variety of osseous abnormalities . however , accumulation of bone scan tracer outside the skeleton can pose a difficulty in reporting for a nuclear medicine physician , especially if only planar imaging is performed and the tracer uptake is overlapping or is in close vicinity of the skeleton . localization of bone scan tracer in a renal calculus has been reported in the past and its use in preoperative in vivo localization of the renal calculus has been explored . retention of bone scan tracer on ureteric calculi and bladder calculus has been reported in the past.[35 ] in one of the studies , autoradiography of ureteric calculus demonstrated peripheral tracer distribution within the calculus . accumulation of the radionuclide due to sluggish flow and its absorption onto the crystal surface , within the calculus , were suggested as possible mechanisms of tracer uptake . the other common causes of urinary system localization of the bone scan tracer are dilation of urinary - collecting system , bladder diverticulum , and presence of an ureterostomy bag.[69 ] conditions causing extraskeletal accumulation of bone scan tracer must be kept in mind while reporting a bone scan . our case demonstrates a rare occurrence of extraosseous bone tracer accumulation in a renal calculus . it also highlights important role played by spect - ct in localizing the extraskeletal tracer uptake . spect - ct can be used effectively when an extraskeletal uptake is encountered on planar bone imaging .

extraosseous localization of radioisotope , used in bone scan , in a variety of physiological and pathological conditions is a well - known phenomenon . the causes of extraosseous accumulation of bone - seeking radiotracers should be kept in mind when bone - imaging studies are reviewed to avoid incorrect interpretations . we report an extremely rare occurrence of extraosseous accumulation of bone scintigraphy tracer in a renal calculus , in a patient with adenocarcinoma of prostate , that was demonstrated by single photon emission computed tomography and computed tomography ( spect - ct ) fusion imaging .

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malignant hematologic disorders ( multiple myeloma , myelodysplasia , chronic lymphocytic leukemia , and hodgkin 's and non - hodgkin 's lymphoma ) and idiopathic aplastic anemia that occur in the population with an incidence of 0.5 - 5/100,000 increase the surgical operative risk due to coagulation defects , changes of blood viscosity , immunosuppression , and bone marrow insufficiency ( 1 ) . pancytopenia associated with idiopathic aplastic anemia may pose an increased risk for postoperative bleeding and infection ( 2 ) . here , we report a patient with severe aortic valve insufficiency undergoing bioprosthetic aortic valve replacement who suffered from idiopathic aplastic anemia . a 66-yr - old man was admitted to the hospital because of shortness of breath . his dyspnea was classified by the new york heart association ( nyha ) as functional class iii . the patient 's history included treatment for severe aplastic anemia with anti - thymocyte globulin and prednisone for 5 yr prior to admission . echocardiography revealed reduced left ventricular function and severe aortic valve regurgitation ( grade iv ) with left ventricular end diastolic dimension measuring 87 mm . the presence of severe dyspnea and echocardiographically documented severe aortic valve insufficiency led us to schedule a surgery for elective aortic valve replacement . on admission , laboratory testing showed leukocytopenia ( white blood cells 1,900/l ) , anemia ( hemoglobin 6.7 mm / l ) , and thrombocytopenia ( platelets 34,000/l ) . the differential blood count revealed 42.9% neutrophils ( 50 - 70% ) , 42.3% lymphocytes ( 25 - 40% ) , 6.9% monocytes ( 2 - 8% ) , and 4.3% eosinophils ( 2 - 4% ) . preoperative preparation included two weeks of granulocyte colony - stimulating factor ( neupogen 300 g , amgen , subcutaneously three times a week ) , transfusion of four units of packed red blood cells ( rbcs ) , and ten units of platelet concentrations ( pcs ) one day before the surgical procedure resulting in a hemoglobin level of 9.8 mm / l , a leukocyte level of 10,300/l , and a platelet level of 120,000/l preoperatively . the patient received aortic valve replacement with a 21-mm bioprosthetic aortic valve ( carpentier - edwards , edwards lifesciences , irvine , ca , u.s.a . ) by a standard median sternotomy ; 2 milrione units of aprotinin were added to the cardiopulmonary bypass circuit priming solution . on the second day after surgery , atrial fibrillation deveolped and was successfully converted with intravenous amiodarone . antibiotic prophylaxis was given using ceftriaxon ( rocephin , roche , basel , swiss ) 2 g intravenously over 6 days . the patient was discharged from the intensive care unit 4 days after surgery and was discharged from the hospital postoperative on day 11 with a leukocyte count 4,300/l , hemoglobin of 6.2 mm / l , and a platelet count of 62,000/l . after the surgery , the patient received 5 units of rbcs and 20 units of pcs until discharge . perioperative laboratory findings are summarized in table 1 . during the 6 months of follow - up , the patient did well with a functional class of nyha class i. the chest radiography at 3 months post surgery showed a decreased cardiac size . according to the definition of the severity of aplastic anemia ( 5 ) , the patient 's aplastic anemia could be categorized as non - severe aplastic anemia . regardless of the severity of aplastic anemia , ( 1 ) , two questions have to be considered when cardiac surgery is indicated in patients with a hematologic disorder causing pancytopenia . given the increased operative risk and the potential benefit with regard to the life expectancy and quality of life , aggressive surgical treatment has to be compared with conservative treatment and the natural course of both , the hematologic disorder and the cardiac disease . our patient suffered from severe dyspnea on exertion ; the patient had stable idiopathic aplastic anemia and was under hematological supervision . progressive deterioration of ventricular function was documented on serial echocardiographic examinations . both symptomatic aortic regurgitation and impaired left ventricular function led us to the decision to perform aortic valve replacement for this patient . another important issue for this specific subset of patients is perioperative management to decrease morbidity and mortality ( 1 ) . potential complications are caused by a decreased number or impaired function of blood cells ; this condition is aggravated by the cell damaging properties of extracorporeal circulation . in addition to meticulous surgical hemostasis , substitution of various blood products is important to reduce the risk of bleeding complications . total amount of the blood product transfused to this patient is regarded to be acceptable considering the higher risk of bleeding in these specific subset of patients . aprotinin has been shown to decrease blood loss and reduce the use of blood products by at least 30% , in open heart surgery ( 4 ) . thoracic drainage in our patient was 785 ml , which was similar to that of other patients who required cardiac surgery with a variety of malignant hematological disorders ( 1 , 3 ) . another important aspect of perioperative care in these patients is the increased risk for infections . it is of great importance to perform surgery in aseptic conditions and to avoid transmission of pathogenic microorganisms during the perioperative period . the patient received warfarin sodium 2 days after surgery , and inr was maintained between 1.5 to 2.0 . follow up , we have noticed neither thrombotic nor hemorrhagic complications related to the anticoagulation therapy . ( 3 ) , cardiac surgery with extracorporeal circulation is feasible in patients with idopathic aplastic anemia , and it was not associated with excessive complications that might be expected in a patient with this disorder .

major surgery in a patient with pancytopenia might be associated with increased surgical risks , especially for bleeding and infection . a 66-yr - old man was admitted to the hospital due to shortness of breath . his dyspnea was classified by the new york heart association ( nyha ) as functional class iii . prior to admission , he had a 5-yr history of medical management for idiopathic aplastic anemia . the severity of aplastic anemia of the patient was graded as non - severe aplastic anemia . echocardiography revealed reduced left ventricular function and severe aortic valve regurgitation ( grade iv ) with left ventricular end diastolic dimension measuring 87 mm . because of dyspnea and echocardiographically documented aortic valve insufficiency , the patient underwent elective aortic valve replacement . although extracorporeal circulation for valve operations might be associated with aggravation of impaired blood cell function , the patient recovered from surgery uneventfully . here , we report a successful cardiac surgery with extracorporeal cardiopulmonary bypass in a patient with severe aortic valve insufficiency and concomitant idiopathic aplastic anemia .

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greater than 80% of pavms are congenital , and approximately 50% of are associated with hereditary hemorrhagic telangiectasia ( hht ) . in addition to common complaints , such as dyspnea and epistaxis , a pavm can cause hemoptysis , hemothorax , and serious neurologic complications , such as stroke , seizures , and brain abscesses . ebstein 's anomaly ( ea ) is a congenital cardiac malformation characterized by downward displacement of the attachment of the septal and posterior leaflets of the tricuspid valve . ea is also a rare disorder , and patients with ea may have various additional cardiovascular anomalies . although the causes of a pavm and ea have not been established , embryogenic or genetic factors might give some contributions . however , there are no reports concerning the co - existence of pavms or hht with ea . we present a case of a pavm with ea that is suspected to have hht . she had experienced spontaneous recurrent epistaxis for the last several years , with no other remarkable medical history . a chest radiograph showed cardiomegaly and a well - demarcated , 12-mm nodular opacity in the left lung field ( fig . the electrocardiogram showed tall and broad p waves , as well as an incomplete right bundle - branch block . on transthoracic echocardiography , the displacement index ( distance between the mitral annulus and tricuspid annulus , divided by the body surface area ) was 15.6 mm / m , and it fulfilled the displacement index criteria for the diagnosis of ea ( 8 mm / m ) . moreover , tethering of the septal and posterior leaflets of the tricuspid valve was observed with central coaptation failure and severe regurgitation . a portion of the right ventricle was atrialized because of apical displacement of the tricuspid valve . she was diagnosed with ea , and agreed to undergo surgical treatment . to evaluate the nodule in the left lung , a 12-mm serpiginous vascular structure connecting the left interlobar pulmonary artery to the left inferior pulmonary vein was detected in the superior segment of the left lower lobe ( fig . no part of her medical history could explain the causes of secondary pavm , such as chest trauma , thoracic surgery , long - standing hepatic cirrhosis , metastatic carcinoma , mitral stenosis , or infections . three - phase contrast ct scans of the liver and mr angiography of the brain were performed to identify other visceral arteriovenous malformations . a liver ct showed diffuse dilatation of the hepatic arteries and veins with multifocal arteriovenous malformations , suggesting hht ( fig . the brain mr showed no evidence of vascular malformation . on the 7th day of admission , after discharge , she has remained stable and comfortable , and will undergo percutaneous embolotherapy of the pavm in the near future . a pavm is a rare pulmonary vascular anomaly with an incidence of 2 - 3 per 100,000 population.1 ) pavms may be single or multiple , and the left lower lobe of the lung is the most common location of solitary pavm.2 ) greater than 80% of pavms are congenital , and approximately 50% of these are associated with hht , also known as rendu - osler - weber disease.2)3 ) a single pavm < 2 cm in diameter on chest radiography usually does not cause symptoms.4 ) the most common complaint is epistaxis . a pavm can cause dyspnea , cyanosis , hemoptysis , hemothorax , and serious neurologic complications , such as strokes , seizures , and brain abscesses.4)5 ) patients with co - existing hht tend to have multiple arteriovenous malformations , rapid disease progression , and a higher complication rate.2 ) the brain , lungs , and liver are the most frequently involved organs in patients with hht.6 ) ea is a congenital cardiac malformation characterized by downward displacement of the attachment of the septal and posterior leaflets of the tricuspid valve.7 ) ea is also a rare disorder occurring in 1 per 200,000 live births.7 ) most cases are sporadic , and the embryologic and genetic contributors are unclear,8 ) but the ea gene might be located on chromosome 9.9 ) the genetic linkages to hht are located on chromosome 9 or 12.10 ) patients with ea may have various additional cardiovascular anomalies.8)11 ) however , there are no reports concerning the co - existence of a pavm or hht with ea . although it is unclear whether or not a concurrent pavm and ea has an embryologic or genetic relationship , we report a patient with a pavm and ea who was suspected to have hht according to the curaao criteria.12 ) further genetic and embryonic studies are needed to detect a possible relationship between the two medical conditions .

a pulmonary arteriovenous malformation ( pavm ) is a rare pulmonary vascular anomaly presenting as dyspnea or recurrent epistaxis . ebstein 's anomaly ( ea ) , a congenital cardiac malformation , is also a rare condition . there have been no reports concerning the co - existence of pavm with hereditary hemorrhagic telangiectasia ( hht ) and ea . a 40-year - old woman was admitted with a 2-month history of increasing dyspnea and several years of recurrent epistaxis . on transthoracic echocardiography , she was diagnosed with ea and agreed to undergo surgical treatment . a chest ct angiography showed a 12-mm serpiginous vascular structure suspicious for a pavm and a liver ct suggested htt . although it is unclear whether or not a concurrent pavm and ea have an embryologic or genetic relationship , we report a case of a pavm with ea . further genetic and embryonic studies are needed to identify a possible relationship of the two medical conditions .

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bloc excision is mandatory but can represents a real technical challenge , expecially if tumor involves both the skull and the dura . the term primary intraosseous meningioma ( pim ) is used for an extradural meningioma that arises from the cap cells staying in the bone . they are mostly met at the periorbital and frontoparietal regions and they are , generally , devoid of neurological findings . other tumors that can involve the skull are hemangiomas , metastases , many type of carcinoma and sarcoma arising directly by bone or by contiguous tissue ( cutis , muscle , dura ) and invading secondarily the skull . the degree of resection is the most important factor correlating with recurrence and clinical long - term outcome . the aggressive surgery that is needed in these cases could bring a large bone defects to fill . the removal of a tumor involving the skull and the intracranial space could present a technical challenge , especially if the tumor has a dural and/or brain attachment . with a standard craniotomy , the maneuver of separating the intraosseus portion of the tumor from the intracranial portion might produce hemorrhage , traction and injury to the underlying brain . we describe a technique where the tumor involving the bone is before left in situ , normal dura is exposed around the tumor , and the tumor can be afterwards removed en bloc with direct vision and minimally traction of the brain surface . 8 patients , with different tumors involving both skull and dura , were treated with these technique . the histological examination of these patient s specimens showed : atypical meningioma , pim and anaplastic meningioma . below , we describe this technique used in the last patient treated , a man aged 45 with no neurological signs . patient underwent dcc , total surgical excision of pim and cranioplasty with peek custom made prothesis ( synthes ) . a 45 years old man , b.v . , presented to our department of neurosurgery with a mass arising from the right frontoparietal bone . he had not neurological signs and symptoms . computed tomography ( ct ) scan showed a homogeneous expansive bone lesion at the right fronto - parietal region , without contrast enhancement . on magnetic resonance imaging ( mri ) , the lesion was hypointense on t1- and t2-weighted images with no contrast enhancement after intravenous gadolinium administration , but contrast enhancement was seen at the adjacent dura . in the differential diagnosis , were first considered a fibrous dysplasia and primary bone tumors . patient undergoes tumor removal and cranioplasty with peek custom made prothesis ( synthes ) . under general anesthesia , the patient was positioned with the head turned approximately 30 toward the contralateral side , to expose the skull mass , with a pillow under the ipsilateral shoulder . the temporal muscle was splitted , the muscolar part that covering the tumor was left in situ for a completely radical resection and the remaining part was overturned . one only burr hole was performed laterally to the midline and we performed the first circle craniotomy around the lesion . then , using the same burr hole , a second and more enlarged circular concentric craniotomy was performed . there was no tumor in the bone ring and the dura beneath the ring was not involved . the inner surface of the dura was amply involved by the tumor , and it was dissected by the underlying brain surface under direct vision thanks to the space guaranteed by bone ring removal . the dural defect was repaired with pericardium bovinum patch and a peek custom made cranioplasty was performed ( fig 3 ) . tumor involving many tissues should be resected en bloc for minimizing risk of seeding and recurrency . in fact , meningioma with calvarial hyperostosis is commonly associated with an increased rate of recurrence if limited bone resection is performed . although radical excision of tumor involving the skull is the gold standard of oncological treatment , elevating a bone flap when the tumor involves both the skull and the dura could be very hard and represents a technical challenge . with a standard craniotomy this maneuver , done without direct vision , could produce hemorrhage and unwanted traction on the underlying brain surface . we have described a technique , a dcc , that permit to expose the normal dura surrounding the tumor and permit the resection of entire specimen with a direct vision of the underlying brain . in our opinion also have described a similar procedure for the removal of nail that was hammered in both the skull and the brain . sekhar et al . have utilized the concentric craniotomy technique for a fronto - orbital approach and ibarra - de la torre et al . for a posterior fossa craniotomy to protect the underlying dura and vascular structures . we believe that dcc is an optimal alternative on standard technique because permits the total removal of tumor involving both skull and dura structures , minimizing tractions on the underlying brain and permitting the direct vision during the dissection .

highlightsmany tumors can involve the skull . radical excision is the golden standard of treatment.elevate a bone flap when the tumor involves both the skull and the dura present a technical challenges.double concentric craniotomy is a safe and effective technique to remove tumor involving both skull and dura structures under direct vision .

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the authors declare that there is no conflict of interests regarding the publication of this paper . this is a case report , patient written consent has been obtained and will we available on request . written informed consent was obtained from the patient for publication of this case report and accompanying images . a copy of the written consent is available for review by the editor - in - chief of this journal on request . asp and kh performed the documentation of the patient and follow up care , assisted in operation and draft writing .

highlightshaemophilic pseudotumour was caused by repetitive bleeding.this is a bilateral iliopsoas pseudotumour with neurologic deficit and contracture.after excision , there was improvement in muscle power from score 2/5 to 4/5.recurrent bleeding will cause joint contractures chronic pain and nerve compression.surgery should be performed in major center by an integrated surgical team .

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modern drugs have made great contributions to better quality of life , less disabling symptoms , decreased demands of health care and a better prognosis . despite this , one can nowadays notice an increasing proportion of negative side effects and adverse drug reactions due to extensive pharmacological treatment . to examine if patient - focused drug surveillances were associated with a higher quality of drug treatment at nursing homes ? physicians were told to systematically focus on the patient 's basic health status as a fundamental starting point for further continuous medication , with the aim to obtain a rational drug usage . mortality , health care consumption and number of drugs . health status and evaluation of drug therapy . evaluation of medications showed significant differences favouring the intervention group . due to existing polypharmacy there was a significant reduction of number of drugs in the intervention group vs. a significant increase in the control group . the intervention seemed to have achieved significant positive results in quality of drug treatment , although it has shown immense lacks of monitoring the health status of frail elderly .

backgroundmodern drugs have made great contributions to better quality of life , less disabling symptoms , decreased demands of health care and a better prognosis . despite this , one can nowadays notice an increasing proportion of negative side effects and adverse drug reactions due to extensive pharmacological treatment.study objectivesto examine if patient - focused drug surveillances were associated with a higher quality of drug treatment at nursing homes?methodsphysicians were told to systematically focus on the patient 's basic health status as a fundamental starting point for further continuous medication , with the aim to obtain a rational drug usage.settingan intervention study in nursing homes in sweden.outcomesmortality , health care consumption and number of drugs . health status and evaluation of drug therapy.resultsno significant difference in mortality rate . extensive health care consumption in both groups . significant differences of examinations like weight , blood pressure and renal function . monitoring and evaluation of medications showed significant differences favouring the intervention group . due to existing polypharmacy there was a significant reduction of number of drugs in the intervention group vs. a significant increase in the control group.conclusionsthe intervention seemed to have achieved significant positive results in quality of drug treatment , although it has shown immense lacks of monitoring the health status of frail elderly .

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one of the methods of partial ureter substitution is the use of free bladder autologous mucosa flaps . this method has been in use for about 50 years . despite previous reports describing the benefits of this method , in reconstructive surgery , up to now , it has not been used in clinical practice . our poor morphologic results with the use this method prompted us to perform microangiography of the newly constructed part of the ureter with this method to definitively resolve the problem of neovascularization in this area . the experiments were carried out on 29 mongrel dogs , as previously described [ 14 ] . all animals were operated on in the same manner ( figure 1 ) , with general endotracheal anesthesia . after grafting , four of the experimental animals were additionally subjected to perfusion of the vascular bed with a radiopaque medium called micropaque ( barium sulphuricum ) ( nichols roche , france , distributed by schering germany ) . this was done to ascertain neovascularization in the supplemented part of the ureter by a tube constructed from the free mucosa flap of the urinary bladder . microangiography was performed after a longitudinal median laparotomy of 34 cm over the kidney vessels . the aorta was ligated and a cannula connected with the perfusion set ( figure 2 ) was inserted into the aorta . to avoid errors in performing the operation and interpreting our results , we carefully studied the literature [ 512 ] and consulted with other investigators about the principles of this kind of experiment . the perfusion of 500 ml 30% micropaque ( barium sulphuricum ) ( nichols roche , france , distributed by schering germany ) , with 0.9% nacl was transfused and subsequently 30% micropaque ( barium sulphuricum ) ( nichols roche , france , distributed by schering germany ) with 10% buffered formalin . perfusion was performed under constant pressure of 140/80 mm hg , at the temperature of 37c , for 30 min . after the procedure , all the experimental animals were euthanized and the samples were harvested and stored for 6 h at 20c . then , a segment of ureter measuring 10 cm in length was excised ( 5 cm above and 5 cm below the graft suture site ) , showing the surface vessels filled with micropaque ( figure 3 ) . at the end of the procedure , the specimen was stitched on the celluloid plate and harvested with 10% formalin , buffered to 7.6 ph . after 14 consecutive days , the specimens were irradiated to show the visible net of the new vascular bed ( figure 4 ) . then , they were cut into 2-mm - thick slices using a microtome and were irradiated by direct exposure and visualized on celluloid film . exposure time was 10 min , electric voltage 20 kv , and current intensity 5 ma . we used single - sided celluloid film type tn-12 , ( bydgoszcz manufacturing fotochemic plant , poland ) , which allowed us to obtain image of capillaries of up to 100200 , because these celluloid films contain a one - sided photosensitive layer . a stepwise process of the ureter substitutions from 1 dog is shown in figures 13 . microscopic evaluations showed the complete regeneration of the mucosa of the supplemented ureteral segment [ 14 ] . the defects in the ureters were not bridged by the smooth muscle [ 14 ] . the experimental findings showed a massive periureteral fibrosis , due to reparative or reactive process , which was considered as the main reason for the failure of our interventions [ 14 ] . one of the most common causes of scarring or stricture of the ureters is ureter surgery . the reason for this is that the arteries going to the uterus are very close to the ureters . during surgery , the amount of scarring and inflammation that occurs after surgery can be very dense and as a consequence leads to hydronephrosis and destroying the kidney . previous reports described the benefit of free autologous bladder mucosa graft for the reconstruction of the ureter . however , unpublished observations have generated a significant interest and a need to re - evaluate autologous ureter grafts constructed from the free autologous mucosa flap ; therefore , this method has not been used in clinical practice . it should be noted that the results of our previous studies [ 14 ] and of the present study do not support a free bladder mucosa flap as a viable clinical therapy for ureteral reconstruction , in spite of the positive reports of the above - mentioned authors . the present animal study suggests that the surgical ureteral reconstruction using this method or grafted tissue is not suitable to achieve clinically desirable results .

backgroundthere is a paucity of data addressing the blood supply in the surgically reconstructed ureter , and complete lack of microangiographic studies of the reconstructed ureter with the use of a free bladder mucosa flap . the present study evaluated the blood supply in the reconstructed dog ureter after a 5-centimeter segment resection , supplemented by a tube constructed from a free bladder mucosa flap.material/methodsfemale mongrel dogs ( n=29 ) were used in this study . under general anaesthesia , a 5-centimeter autologous free bladder mucosa flap was used to construct a tube , which was afterwards grafted to replace a 5-centimeter ureter resection . after a period of 3 months ( n=2 ) and after 1 year ( n=2 ) , microangiography was performed to assess the revascularization of the grafted ureter.resultsin our study , we observed the continuity of the ureter , but the grafted reconstruction was narrowed by the cicatrization in about 86% ( n=25 ) of cases . this resulted in the development of hydronephrosis , as described in previous publications . the ureteral wall was covered by a normal urothelium , but consisted of fibrous connective tissue , which failed to restore a regular ( normal ) coat . the reconstructed segment showed no smooth muscle cells . a few smooth monocytes were found only at the border with intact portions of the ureter . the microangiography performed at the end of the experiments showed no vascularization of the restored segment of the ureter.conclusionsthe experiments showed a whole regeneration of urothelium in the transected and reanastomosed ureters . however , there was no regeneration of the muscular coat and a complete lack of revascularization .

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an increased incidence of hashimoto thyroiditis has been reported in patients with turner syndrome . in view of its pathogenic relationship with autoimmune thyroiditis we report the case of a 9-yr - old girl with turner syndrome who developed graves disease during recombinant human gh therapy , and we discuss previous reports of this association . the patient was the first child of healthy unrelated parents of normal stature , father 175.0 cm and mother 159.0 cm . the patient was born after an uncomplicated pregnancy at a gestational age of 37 wks and 1 d. her birth weight was 2490 g. on examination , lymphedema and webbed neck were detected . chromosomal analysis of peripheral blood lymphocytes ( n=26 ) showed complete deletion of the short arm of the second x chromosome ( karyotype : 45 , x ) . at the age of 5 yr and 2 mo , her height was 98.2 cm ( 2.25 sd ) and her weight was 14.4 kg ( 1.48 sd ) . a gh stimulation test was performed , and the peak gh values after stimulation with clonidine and arginine were 8.39 ng / ml and 5.56 ng / ml , respectively . of note is that diagnosis of complete gh deficiency is less than 5 ng / ml in this assay . recombinant human ( rh ) gh therapy ( 0.175 mg / kg / wk ) was initiated with success ( fig . thyroid function tests showed a tsh level of 0.01 u / ml ( normal , 0.50 to 5.50 u / ml ) , a free ( f ) t4 value of 6.00 ng / dl ( normal , 0.85 to 1.80 ng / dl ) , and a free t3 value of 21.76 pg / ml ( normal , 2.50 to 5.50 pg / ml ) . anti - thyroglobulin antibody was 3.4 u / ml ( normal , < 0.3 u / ml ) . anti - thyroid peroxidase antibody was 5.5 u / ml ( normal , < 0.3 u / ml ) . tsh receptor antibody ( trab ) was 52% ( normal , < 15% ) . these results are compatible with a diagnosis of graves disease and treatment with thiamazole was started . growth curve of the patient at the latest follow up ( 9 y old ) , the development of the patient s breast was tanner stage 1 . treatment with thiamazole was continued and the thyroid function has stayed within normal ranges . the association of turner syndrome with graves disease was reported in english about 1020 years ago ( 1,2,3,4,5,6,7,8,9,10 ) . table 1table 1 graves disease in turner syndrome summarizes the historical thirteen known cases of graves disease with turner syndrome , including our case . this table reveals that the clinical characteristics of graves disease with turner syndrome are similar to those known generally in patients without turner syndrome in terms of ages of the onset , symptoms and prognosis . treatment with rhgh was given to 4 patients in table 1 , and it was continued in 3 out of the 4 patients including our case . this continuation of gh treatment , together with the successful treatment of hyperthyroidism , suggests that a direct link between gh treatment and graves disease is unlikely . consistent with this hypothesis , the incidence of hyperthyroidism in japanese patients with turner syndrome receiving rhgh therapy is 0.40% , which is similar to overall incidence of hyperthyroidism in patients with turner syndrome , 00.5% ( 11 , 12 ) . it is well known that hyperthyroidism in patients with turner syndrome leads to acceleration of height velocity ( 6 , 13 ) . in our patient , the height velocity was not increased by hyperthyroidism , probably because her growth had been already accelerated by rhgh therapy before the onset of hyperthyroidism . it is important to monitor changes of thyroid function as well as growth parameters in patients with turner syndrome , as is recommended by sybert and mccauley ( 14 ) .

an increased incidence of hashimoto thyroiditis has been reported in patients with turner syndrome , but several cases of graves disease were also described ten to 20 years ago . we report the case of a patient with turner syndrome who developed graves disease , 3 years after successful treatment with recombinant human growth hormone ( gh ) . a diagnosis of graves disease was made and treatment with thiamazole was started , which resulted in normalization of the thyroid function . it is important to monitor thyroid function as well as growth parameters in patients with turner syndrome .

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a 28-year - old healthy male was seen in our clinic complaining of vision loss in his right eye ( od ) . best - corrected visual acuity ( bcva ) was od : 20/200 and left eye ( os ) : 20/20 . mild vascular abnormalities were detected in the temporal aspect of right optic nerve , but no conclusive information was obtained . oct using the stratus oct 3 model ( carl zeiss meditec , jena , germany ) was indicated for the evaluation of the macular areas , rnfl thicknesses , and optic nerves . the fast macular protocol revealed subtle macular thickness beyond normal in the superior and nasal quadrants of both maculae . no visible alterations in the internal microarchitecture of the retina were observed using several retinal lines and optic disc protocols . a slight incremental thickening of the rnfl was observed in the superior and nasal quadrants of the os . in order to evaluate the patient 's visual field ( vf ) and retinal sensitivities , a frequency doubling technology perimetry ( fdt ) , using the commercially available ( matrix ) device , was performed in both eyes . the threshold 30 - 2 strategy revealed the presence of junctional scotoma composed by a central scotoma in od associated with superior temporal quadrantanopia in the fellow eye . the pattern detected in the vf suggested the presence of an expansive mass at the level of the optic chiasm [ fig . 1 ] . magnetic resonance imaging ( mri ) was performed which disclosed the presence of a tumor , compatible with the diagnosis of pituitary adenoma [ fig . 2 ] . after a complete neurological evaluation , medical treatment with cabergoline ( a dopamine agonist ) , after 8 months , a significant reduction of tumor volume was achieved . moreover , the patient experienced a full recovery of bcva and vf [ fig . 3 ] . right : superior temporal quadrantanopia in os central nervous system mri . left : pituitary dependent macroadenoma . visual loss associated with a junctional scotoma is a well - known clinical sign related with the presence of a compressive mass in the chiasmal area . monteiro described a generalized reduction of the rnfl thickness in the retinas of patients with band atrophy secondary to pituitary tumors . lederer and colleagues reported on the usefulness of the macular map thickness , while studying patients with glaucomatous optic nerve alterations . due to a relative incremental change in the ganglion cells axonal concentration within the macular area interestingly , the patient described here showed a thickening in the nasal and superior and nasal aspects of both maculae . this tomographic sign , not visible on the biomicroscopic fundus examination and in the fa , was interpreted as an early retinal manifestation produced by the presence of the tumor . the physiopathology of this localized , incremental thickness is merely speculative , but could be produced by an alteration in the axonal transport in ganglion cells . after obstruction of the axoplasmic flow , an early enlargement ( intracellular edema ) of the nerve fibers followed by a chronic atrophy could occur . though no direct evidence exists corroborating this phenomenon , at least three indirect clinical issues support this theory : ( 1 ) the retinal thickness increment was revealed anatomically by the oct , but no evidence of dye leakage was seen in the fa . ( 2 ) the presence of a topographic correlation between the junction scotoma and the macular thickening . ( 3 ) the visual loss was ipsilateral to the main tumor location . furthermore , the macular thickness enlargement was symmetric while the visual alteration was roughly asymmetric . this most likely could be indicative of the potential for intracellular edema preceding the visual function alteration and this thickening could be an early sign that could be detected before consolidated fiber atrophy occurs . frequency doubling technology perimetry ( matrix fdt ) has shown to be a suitable method for studying and detecting abnormalities in patients with neurological visual field deficits . during the functional evaluation of this patient , the fdt revealed the presence of a junctional scotoma compatible with the tumor location . even with an asymmetric vf defect , the oct was able to show symmetric involvement , with respect to macular thickness . we speculate that the anatomical alteration revealed by the oct precedes the functional deficit found in the vf . thus , we propose that detection of bi - superonasal macular edema ( me ) could be an early sign of chiasmal pathology . with an early diagnosis and subsequent treatment , a complete restoration of visual function was achieved in this case , stressing the importance of early recognition of the disease . we are unaware of previous reports regarding localized bi - superonasal me secondary to a pituitary adenoma and could find no references to it in the medical literature . studies including a cohort of patients are warranted for the evaluation of these tomographic findings . in conclusion , it is important to suspect pituitary adenoma in cases of vf deficit and oct macular alterations .

a 28-year - old healthy male complaining of vision loss in his right eye was discovered to have localized bi - nasal macular edema in the presence of a pituitary adenoma . the presence of a junctional scotoma composed by a central scotoma in the right eye associated with superior temporal quadrantanopia in the fellow eye was seen . the pattern detected in the visual field suggested the presence of an expansive mass at the level of the optic chiasm . optical coherence tomography findings also revealed subtle macular thickness beyond normal in the superior and nasal quadrants of both maculae . this report illustrates the importance of suspecting a pituitary adenoma in the light of uncharacteristic retinal alterations .

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six species of echinococcus have been recognized , but four of them are of public health concern ( 1 ) . the greatest prevalence of hydatidosis in human and animal hosts is found in sheep - raising countries , including north america and south america , the entire australia , new zealand , europe , central asia , china , and parts of africa ( 1 , 2 , 46 ) . various parts of body may be involved with hydatid cyst but the liver and lungs are the main locations ( 7 ) . skeletal muscle infection is rare , and reported 0.5% 4.7% of all cases of echinococcosis ( 8) . patients with hydatid cyst are asymptomatic and present at an advanced stage of hydatidosis , when lesions have become extensive ( 7 ) . here we report a case of this rare entity of an isolated hydatid cyst of the muscle of the thigh . a 70-years old housewife living in ardoghesh , a village in neyshabur , northeast of iran , was admitted to general surgery clinic in feb 2014 because of a painless mass in the back of her left thigh . she had a history of removing hydatid cyst surgery in her left thigh from three years ago . laminated layers of hydatid cysts were observed and two round masses was successfully removed ( fig . hydatid cyst with laminated layer ( original ) gross pathology showed two soft cystic masses ( 134/54/5 cm and other 1744 cm in diameter ) that contained gelatinous material and multiple daughter cysts . in histopathologic examination cross section of a hydatid cyst with laminated layer and germinal layer , brood capsules containing multiple protoscolices , hooklets are considered as diagnostic keys ( h&e ) . pathological examination confirmed diagnosis as hydatid cyst of thigh ( fig . 2 , 3 ) . hydatid cyst in muscle ( original ) hydatid cyst in muscle showing germinal layer ( original ) hydatid disease is a very serious problem in sheep - raising countries caused by tapeworms belonging to the class cestoda , in the family taeniidae , of the genus echinococcus . they measure 3 mm to 6 mm long when mature and lives in intestine of carnivores , particularly dogs and other canines , as definitive hosts . many mammals may serve as intermediate hosts , but herbivorous species are most likely to become infected by eating eggs on contaminated herbal material . humans are seldom involved as accidental intermediate hosts in these cases and infected by accidentally ingestion of echinococcus spp . hydatidosis is a serious public health problem in some parts of the world ( 4 , 10 ) . khorasan province , located in the northeastern part of iran had the highest incidence rate for hydatidosis ( 11 ) . although the incidence of hydatidosis has decreased because of education and control programs , there are still concerns in some parts of the world ( 12 ) . hydatid cyst is most commonly found in the liver and lung , while they can occur in other organs including muscle , brain , eye , spleen , kidney , orbit , lymphatic glands , myocardium , tonsil , pancreas , skin , ovary , uterus and parotid glands ( 2 , 1317 ) . hydatidosis are usually asymptomatic until adolescence due to the slow growing process of the parasite in tissues such as muscle and bone , although it can be acquired at any age ( 18 ) . incidence rate of musculoskeletal hydatidosis is not clear . some reports showed an incidence of musculoskeletal echinococcosis including involvement of subcutaneous tissue as 0.5%4.7% among all cases of hydatid disease and soft - tissue hydatid cysts occur in 2.3% of cases reported from endemic areas ( 8 , 19 ) . muscles provide a poor environment for the parasite because of the presence of lactic acid and mechanical factors , such as contractile activity , may make encystment less likely ( 1820 ) . it may mimic any soft tissue tumor such as abscess , synovial cyst , and malignant tumor . before biopsy of cyst , diagnosis of hydatidosis should be confirmed to avoid leakage of cyst contents and the accompanying risks of anaphylaxis ( 20 ) . hydatidosis in soft tissues may present with a variety of patterns and recognizing them is necessary in diagnosis ( 18 ) . in our presented case , preoperative diagnosis was malignant neoplasm and after surgery , hydatidosis was confirmed . rokni yazdi et al . noted left thigh hydatidosis in a 50-years - old housewife living in a village in zanjan , northwestern iran ( 18 ) . noted left thigh hydatidosis in a 50-year - old woman from rural area around rasht city with no history of trauma , fever , or weight loss ( 20 ) . in conclusion , the hydatid cyst can present in any part of the body and no part is protected . the infestation may resemble a soft tissue tumor in the muscle and therefore in endemic area of hydatidosis , hydatid cyst should be considered as differential diagnosis of any soft tissue mass . in this case ,

hydatid cyst is an important endemic zoonosis in iran . it may be seen in various organs of body . musculoskeletal system is rarely involved by hydatid cyst , the larval form of echinococcus granulosus . on clinical basis , it may resemble any soft tissue tumor . a 70-years old housewife living in ardoghesh , a village in neyshabur , northeast iran , was admitted to general surgery clinic because of a painless mass in the back of her left thigh . this case emphasizes that hydatidosis should be included in differential diagnosis of any soft tissue mass especially in regions where hydatidosis is endemic .

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a 36-year - old man presented with sudden severe occipital headache , and neck stiffness in may 1999 . after several weeks , the headache subsided , however , blurred vision subsequently developed . t1-weighted ( 665/14/2 [ repetition time / echo time / excitation ] ) and t2-weighted ( 4200/99/2 ) mr images showed a large cystic mass with a small solid portion in the left temporal lobe ( figs . gd - dtpa enhanced t1-weighted images showed marked enhancement of a thick peripheral wall and solid portion ( fig . 1c ) . on diffusion - weighted mr images ( b value=1000 sec / mm , isotropic image ) , the mass was surgically excised , and histopathologic examination revealed a typical adamantinous craniopharyngioma with anastomosing epithelial islands and a palisaded layer of cells , as well as an area of keratinization and numerous calcifications ( fig . craniopharyngiomas are generally considered to be epithelial tumors arising from remnants of the craniopharyngeal duct , which connects the stomodeal ectoderm with the evaginated rathke 's pouch , which in turn forms the adenohypophysis ( 7 , 8) . this theory suggests that craniopharyngiomas can arise anywhere along the migration of rathke 's pouch , which extends from the vomer and the roof of the nasopharynx , through the midline sphenoid bone to the floor of the sella turcica . thus , craniopharyngiomas can potentially arise in unusual locations such as the nasopharynx ( 2 , 8) , sphenoid bone ( 9 ) , third ventricle ( 3 ) , and posterior fossa ( 10 ) . however , the above hypothesis does not explain the development of craniopharyngiomas either in the pineal gland ( 5 ) or the temporal lobe as in our case . there is no clear embryological reason for craniopharyngiomas to originate from the pineal gland or temporal lobe . ( 6 ) suggested that they might originate from totipotential or multipotential cells that reside in the pineal gland . however , metastasis has never been described in this benign neoplasm , and therefore we think that the former hypothesis is more reasonable than the latter . craniopharyngioma would not usually be included in the differential diagnosis of a temporal lobe mass . given the well - defined cystic mass with peripheral dense enhancement , our preoperative diagnosis was pilocytic astrocytoma . craniopharyngiomas can be classified into two histopathologically and clinically distinct subtypes ( i.e. adamantinous and squamous - papillary variants ) ( 8) . the adamantinous type consists of a predominantly cystic lobulated tumor , which is often observed in an intrasellar / suprasellar location in children . these cysts contain various amounts of cholesterol , triglycerides , methemoglobin , protein , desquamated epithelium , and watery fluid content . squamous - papillary craniopharyngioma , on the other hand , consists of a predominantly solid or mixed solid - cystic spherical tumor in a suprasellar location in adults . the solid tumor parts have an inhomogeneous but intense enhancement with small necrotic areas , and calcifications are rare . the combination of papillary and adamantinous tumor parts within the same neoplasm has been described in 15% of these tumors . in summary , although our case did not show any specific radiologic finding permitting the differentiation of craniopharyngioma , to the best of our knowledge , this is the first case of a craniopharyngioma originating in the temporal lobe . it does not appear to be embryologically derived from ectopic embryonic remnants of the craniopharyngeal duct .

herein , we report on an unusual case of craniopharyngioma arising in the temporal lobe with no prior history of surgery and with no connection to the craniopharyngeal duct . mr images showed a cystic tumor with a small solid portion . to the best of our knowledge , this is the first case of a craniopharyngioma occurring in the temporal lobe .

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a 22-year - old female reported with complaints of multiple missing teeth in upper and lower anterior region . on examination , patients skin was dry and soft on face and increased thickness of nails ; palm and sole were normal . the patient had 18 , 15 , 12 , 22 , 27 , 28 , 38 , 45 , and 48 missing teeth with generalized microdontia . the canines had cone shaped clinical crowns , and molars appeared malformed [ figure 1 ] with obliterated occlusal tables . vertical dimensions were reduced due to a lack of occlusion and vertical stop causing protuberant lips . the panoramic radiograph confirmed presence of retained primary teeth in both the arches . with the above findings and orthopantomogram ( opg ) figure , we came to a conclusion and diagnosed as haed . to restore the missing teethto increase the vertical dimensionto correct size of teethalignment of teeth . to restore the missing teeth to increase the vertical dimension to correct size of teeth alignment of teeth . treatment plan included compensating microdontia by increasing tooth size using fpd retained by implant . to use implants , enough space was required ; so orthodontic alignment of teeth was required . as teeth were distributed unevenly , it had to be aligned to gain space for implants . retained deciduous teeth were extracted and orthodontic correction [ figure 2 ] done with aligning and leveling . orthopantomograph during orthodontic correction treatment two implants [ figure 3 ] were placed in region of 12 , 13 and one in the region of 45 . final fpd [ figure 4 ] was given and orthopantomograph [ figure 5 ] taken posttreatment . intraoral photograph posttreatment orthopantomograph posttreatment the prosthetic rehabilitation provides the patient with esthetic , functional , and phonetic benefits , as well as a better outlook towards her social life and peers oral hygiene instructions , were given to the patient , and he was asked to report for follow up and assessment of her fixed prosthetic appliance after 6 months . to restore the missing teethto increase the vertical dimensionto correct size of teethalignment of teeth . to restore the missing teeth to increase the vertical dimension to correct size of teeth alignment of teeth treatment plan included compensating microdontia by increasing tooth size using fpd retained by implant . to use implants , enough space was required ; so orthodontic alignment of teeth was required . as teeth were distributed unevenly , it had to be aligned to gain space for implants . retained deciduous teeth were extracted and orthodontic correction [ figure 2 ] done with aligning and leveling . orthopantomograph during orthodontic correction treatment two implants [ figure 3 ] were placed in region of 12 , 13 and one in the region of 45 . final fpd [ figure 4 ] was given and orthopantomograph [ figure 5 ] taken posttreatment . intraoral photograph posttreatment orthopantomograph posttreatment the prosthetic rehabilitation provides the patient with esthetic , functional , and phonetic benefits , as well as a better outlook towards her social life and peers oral hygiene instructions , were given to the patient , and he was asked to report for follow up and assessment of her fixed prosthetic appliance after 6 months . the typical treatment of a patient with ed is achieved in several phases , depending on the craniofacial development , thus requiring a comprehensive and multidisciplinary approach . the different treatment modalities are dictated by manifestation of level of hypodontia and resulting malocclusions . numerous clinical reports have demonstrated importance of prosthetic dental treatment in ed patients for psychological and psychosocial reasons . a number of reviews regarding treatment of ed has showed that implant therapy in early infancy or adolescent age while little information is present in literature for ed patient above 18 years of age . prosthodontic treatment of ed can include fixed , removable , or implant supported prosthesis of which implant and fixed partial prosthodontics are the most frequent modalities . in removable prosthesis , frequently the oral mucosa is dry due to a decrease in the number of mucous glands and lesser quantity of saliva which makes retention of removable prosthesis more difficult . overdentures can be used to restore ideal occlusion and usually allow preservation of existing dentition , but these require rigorous oral hygiene regimes to avoid development of caries and periodontal problems . this case report describes a routine method for fixed prosthodontic treatment of a patient with ed . as the patient was older than 18 years , the growth period was assumed to be over that would have resulted in the failure of fpds as shown by hogeboom . a study by guckes et al . , showed that patients younger than 18 years had a hazard ratio of 2.8 compared to the patients older than 18 years . so for young patients , fpds should be avoided as they could interfere with jaw growth and implants should be recommended and vice versa in patients > 18 years . the dental appearance of patients affected by ed has to be improved for physiologic and psychologic reasons , so that they can lead a normal social life with self - esteem . this clinical report demonstrates the implant retained fpd as a suitable treatment while treating an adult patient with ed . this treatment option gives excellent result , which not only improves masticatory function but also enhances esthetics , allowing patients to lead a normal social life .

ectodermal dysplasia represents a group of rare inherited conditions in which two or more ectodermally derived anatomical structures fail to develop . early dental intervention can improve patients appearance , thereby minimizing associated emotional and psychological problems in these patients . treatment requires a teamwork by medical personnel along with dental professionals of various specialties . here , a rare case of a young female patient is presented with prosthetic management with implant supported fixed partial denture .

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the lingual tonsils develop at 6.5 weeks between the second and third arches ventrally while palatine tonsils develop at 8 weeks from second pouch ( ventral and dorsal ) . tonsils are predominantly b - organs and b - lymphocytes comprise 5060% of tonsillar lymphocytes . ample evidence shows that tonsils are involved in inducing secretory immunoglobulin production [ 4 , 5 ] . both adenoids and tonsils are favourably located to mediate immunologic protection of the upper aerodigestive tract as they are exposed to air borne antigens . tonsils are particularly designed for direct transport of foreign material from the exterior to the lymphoid cells . involution of the tonsils begins after puberty , resulting in a decrease of the b cell population and a relative increase in the ratio of t to b cells . the commonest indication for tonsillectomy is recurrent tonsillitis , which results in shedding the immunologically active cells and decreasing antigen transport function with subsequent replacement by stratified squamous epithelium [ 6 , 7 ] . we investigated the common pathogens causing this condition in our hospital and report here our findings . patients presenting at our clinic with signs and symptoms of chronic tonsillitis were enrolled for the study . the study was explained to them and where children were involved , to their parents . before the operation began , the laboratory was informed and a technician stood by to collect the tonsil as soon as it was removed . sterile wide - mouthed container was provided and the excised tonsils were aseptically put into them and carried immediately to the laboratory for processing . as soon as the tonsil reaches the laboratory , it is cut into two with a sterile surgical blade ; the inner surfaces were swabbed with sterile cotton swab , and inoculated onto two blood agar plates , one macconkey agar and one chocolate agar plate . one blood agar plate was incubated anaerobically , the chocolate plate in 510% co2 while the rest of the plates were incubated aerobically . the aerobic plates and the co2 plate were examined after 24 hours ; if no growth , they were reincubated for a further 24 hours after which if still no growth , they were discarded as negative . the anaerobic cultures were examined at 72 hours and if no growth they were reincubated for a total of 7 days . there were 34 bacterial isolates from 52 patients , giving a percentage positivity of 65.38% . thirty isolates were gram - positive bacteria and only four were gram - negative , made up of two genera , klebsiella and pseudomonas . staphylococcus aureus was the predominant isolate ( 15/34 , 44.1% ) , followed by group b streptococcus ( 12/34 , 35.3% ) . others were , streptococcus pyogenes ( group a streptococcus ) , 1/34 , 2.94% ; and untypable streptococcus spp . 2/34 , 5.88% . the gram - negative bacteria consist of klebsiella pneumoniae 3/34 , 8.82% and pseudomonas aeruginosa 1/34 , 2.94% ( table 1 ) . there were no growths in 7 patients while 11 yielded growth of normal flora only . all the cases were chronic and most of them took antibiotics before presenting to us . the ratio of anaerobic to aerobic bacteria in saliva is approximately 10 : 1 because of variations in oxygen concentration throughout the oral cavity . invargsson et al . revealed that streptococcus pneumoniae was recovered in 19% of healthy children , hemophilus influenzae in 13% , group a streptococcus in 5% , and moraxella ( branhamella ) catarrhalis in 36% . the frequency of pathogens decreases with age , possibly because of increased immunity . because the oropharynx is colonized by many organisms , most infections of wadeyer 's ring are polymicrobial . in our study , but in contrast with other researchers who found beta - hemolytic streptococci to be the predominant isolate [ 14 , 15 ] . these authors also found that streptococcus pyogenes was isolated more frequently in recurrent tonsillitis while in the tonsillar hypertrophy , streptococci beta - hemolytic non a group predominated . it has been suggested that fine - needle aspiration can be used in identifying tonsil core bacteriology in clinical settings . methicillin resistant staphylococcus aureus ( mrsa ) , has been isolated from the surface and core tonsils in children . we did not encounter any mrsa in our study ; and all the staphylococcus aureus isolated were sensitive to augmentin and vancomycin . it would appear from our results , that augmentin should be our drug of choice in future treatment of tonsillitis from this centre . the findings of kuhn et al . supported the etiologic role of hemophilus influenzae and staphylococcus aureus in hypertrophic tonsils with or without inflammation [ 18 , 19 ] .

objectives . tonsillitis is a common infection in all age groups , especially under the age of five . organisms causing this condition vary from place to place . our aim is to find out the main causative agents of this condition in our hospital . patients and methods . fifty - two consenting patients who needed tonsillectomy in khamis civil hospital , kingdom of saudi arabia , between september 2006 and april 2007 , were enrolled for the study . swabs were taken from their inner surfaces and cultured for anaerobes and aerobes according to standard microbiological techniques . results . fifty - two patients , consisting of 30 males and 22 females were enrolled . their mean age was 9.81 6.47 . nearly 65% of patients had positive cultures while 35% were negative . the commonest bacteria isolated were staphylococcus aureus ( 44.1% ) ; and group b streptococcus ( 35.3 % ) . two gram - negative bacteria , klebsiella pneumoniae , ( 8.82% ) , and pseudomonas aeruginosa ( 2.94 % ) , were also isolated . no anaerobe was isolated . conclusion . gram - positive cocci , consisting of staphylococcus aureus and group b streptococcus ( streptococcus agalactiae ) , are the major causes of tonsillitis requiring surgery in our hospital . antibiotic treatment of this condition should be directed largely against these organisms .

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guillian barre syndrome ( gbs ) is associated in 45 - 75% of cases with cranial nerve involvement . facial nerve is the commonest to be involved followed by extra ocular muscles and lower cranial nerve involvement . only two cases has been reported till date with gbs with total paresis of motor cranial nerves . here a thirteen year old boy presented with acute progressive areflexic flaccid quadriparesis associated with motor cranial nerve involvement with bilateral facial and bulbar weakness . he had an upper respiratory infection one week preceding the motor weakness which started from the lower limbs . on day seven after the onset of motor weakness of limbs , the child developed significant bulbar weakness , difficulty in talking and could not move the tongue . he was totally anarthric . on day nine , he had significant respiratory muscle weakness requiring mechanical ventilatory support . on day fourteen , the nerve conduction studies ( ncs ) were suggestive of severe demyelinating motor sensory polyradiculoneuropathy . his anti - ganglioside antibody panel in the blood showed positive igm gm2 , gt1b , igg gm1,2,3 and gt1b antibodies . his csf showed albuminocytological dissociation and his antinuclear antibody was negative . his magnetic resonance image ( mri ) he was treated with intravenous immunoglobulin at a daily dose of 0.4 g / kg for five days . he showed gradual improvement and started swallowing after eight weeks of the onset of illness . the wasting of the tongue also improved gradually at 3 months after the onset of illness [ figure 2 ] . at 6 months repeat ncs was suggestive of motor sensory demyelinating radiculoneuropathy with improvement in conduction velocities and compound muscle action potential amplitudes compared to baseline study . tongue wasting noted at 2 weeks after the onset of motor weakness improvement in tongue weakness and wasting noted at 6 months twelfth nerve involvement , either isolated or as a part of multiple cranial nerve involvement is quite uncommon and only two cases has been reported till date as a part of multiple motor cranial nerve involvement in a case of gbs . , reported a case of fulminant gbs with quadriplegia and total paresis of motor cranial nerves and polo et al . , reported a case of atypical gbs with multiple cranial neuropathies including xii cranial nerve involvement twelfth nerve involvement , either isolated or as a part of multiple cranial nerve involvement is quite uncommon and only two cases has been reported till date as a part of multiple motor cranial nerve involvement in a case of gbs . , reported a case of fulminant gbs with quadriplegia and total paresis of motor cranial nerves and polo et al . , reported a case of atypical gbs with multiple cranial neuropathies including xii cranial nerve involvement gbs with multiple cranial nerve involvement is a known entity but the involvement of xii cranial nerve is extremely rare . our patient had multiple cranial neuropathies with involvement of vii , ix , x and xii nerves . he had a severe form of gbs with areflexic quadriparesis along with respiratory muscle involvement requiring prolonged ventilatory support . his mri brain was normal supporting that the tongue weakness is due to xii cranial nerve involvement . twelfth nerve involvement , either isolated or as a part of multiple cranial nerve involvement is quite uncommon and only two cases has been reported till date as a part of multiple motor cranial nerve involvement in a case of gbs . tan et al . , reported a fulminant case of gbs with quadriparesis with all motor cranial nerve involvement and their ncs showed segmental demyelination which was responsible for such severe involvement and possibly the pathophysiology in this case and our case are similar . described a 23 years gentleman who developed a progressive illness over ten days with diplopia , facial diplegia and a nasal voice . subsequently , the patient also developed weakness of the neck and tongue muscles , dysphagia , abolition of reflexes of the left arm and right triceps but without involvement of the respiratory muscles or other limbs . however , the same patient had involvement of reflexes of left arm and right triceps only without involvement of respiratory muscles and lower limbs . hence it was thought to an atypical variant of gbs in contrast to our case where he had all the typical features of gbs . our patient showed anti - gm2 antibody positive which is usually seen in post cmv infection with or without gbs . usually going on to ventilatory support requirement and the same is found in our patient . the two previous case reports did not have the autoantibody levels to corroborate the clinical and electrophysiological findings . the cranial neuropathy in our patient showed gradual recovery and tongue atrophy recovered over next three months . at 6 months gbs with multiple cranial nerve involvement can rarely involve the xii nerve as well causing significant tongue weakness and such a presentation may be indicative of a severe nature of the disease and it usually recovers over a period of time .

guillian barre syndrome ( gbs ) is associated with cranial nerve involvement . commonest cranial nerves involved were the facial and bulbar ( ixth and xth ) . involvement of twelfth cranial nerve is rare in gbs . we present a case of gbs in a thirteen years old boy who developed severe tongue weakness and wasting at two weeks after the onset of gbs . the wasting and weakness of tongue improved at three months of follow up . brief review of the literature about xiith cranial nerve involvement in gbs is discussed .

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hyperparathyroidism ( hpt ) occurs when there is an abnormal increase in parathyroid hormone ( pth ) production by the parathyroid gland [ 13 ] . primary hpt ( phpt ) is caused by parathyroid adenomas in 85% of cases , leading to hyperplasia and over - secretion of pth . in most individuals in western society , it is diagnosed at an asymptomatic stage , without signs or symptoms of ( pth ) calcium excess . initial symptoms are nonspecific , such as weakness , malaise , fatigue and possible mood disturbances . if not diagnosed and left untreated , it leads to devastating consequences including nephrolithiasis , nephrocalcinosis , renal failure , osteopenia and osteoporosis . a 22-year - old male student presented to our hospital with a 2-year history of progressive lower extremity weakness . one year prior to the admission , he had been seen at another medical clinic because of fatigue and difficulty walking . his radiographs showed no fractures , and he was referred to physiotherapy for muscle strengthening . further questioning revealed that , despite physiotherapy , his weakness had progressed and he had to quit school as he was having considerable difficulty walking up a flight of stairs . a thorough investigation was performed and showed a serum calcium level of 3.46 mmol / l ( normal : 2.12.6 mmol / l ) and a pth level of 138 pmol / l ( normal : 1.69.3 pmol / l ) . he was referred to our general surgery service where he was admitted for excision of the parathyroid adenoma . upon admission , one day prior to the scheduled parathyroidectomy , he had a simple , low - energy fall while he was walking on his own to the hospital . radiographs revealed a displaced subcapital fracture of his left hip , a segmental fracture of his right humerus and extreme osteopenia ( fig . 1 ) . figure 1:(a ) left femoral neck displaced subcapital fracture and ( b ) right humeral shaft segmental fracture . ( a ) left femoral neck displaced subcapital fracture and ( b ) right humeral shaft segmental fracture . one day after sustaining this injury , the patient underwent an uncemented left total hip arthroplasty ( tha ) ( fig . 2 ) , and acetabular fixation was supplemented with multiple screws , and he maintained touch - down weight bearing for 6 weeks postoperatively . postoperatively , the pth level had decreased to 4.9 pmol / l , and the calcium level had returned to normal at 2.42 mmol / l . the calcium level continued to fall , and 2 weeks after the parathyroidectomy , it had reached 1.70 mmol / l . after a further 20 days , it started to normalize and then continued to remain stable , as shown in fig . 3 . figure 2:(a ) left thr and ( b ) right humerus in a brace . ( a ) left thr and ( b ) right humerus in a brace . a graph for calcium level from admission to 7 weeks after surgery we report this case to illustrate the importance of early detection and diagnosis of phpt , which was possible in this young and presumed healthy individual if proper assessment and management were done . furthermore , sending him for physiotherapy without a clear diagnosis increased the likelihood of missing the diagnosis and progression of the disease . solitary parathyroid adenoma is the most common cause of phpt , representing 8590% of cases . symptomatic phpt with skeletal , renal , abdominal and neuro - psychiatric manifestations has become exceedingly rare in developed countries . our patient was unusual in that he was not diagnosed early and presented with skeletal manifestations resulting in multiple fractures including femoral neck fracture ( fnf ) , which was treated with total hip replacement ( thr ) . at this young age , thr would affect his lifestyle and put him at risk of revision surgery early in his life . the literature clearly shows that the earlier hpt is detected , the more reversible the disease . we believe that , despite the young age of our patient , thr was still the preferred treatment in this situation owing to his poor bone quality , which could have led to failure of fixation ; in addition , severe fracture displacement carries a high risk of femoral head avascular necrosis . yang et al . found that salvage tha for failed internal fixation following fnf is a more technically demanding procedure with prolonged operative time and larger amounts of postoperative drainage ( within 24 h ) and that patients are at increased risk of developing hip complications compared with primary thr for acute displaced fnf . french et al . reported a case of a 21-year - old male with parathyroid adenoma who had presented with a left femoral shaft fracture and right fnfs . although these were treated with fixations , the right side required conversion to thr within a year of his presentation .

we present the case of a 22-year - old male with longstanding progressive fatigue , weakness and pain around his hips due to an undiagnosed parathyroid adenoma . the resultant primary hyperparathyroidism ultimately caused pathologic fractures . he was admitted to the hospital for further assessment and excision of the parathyroid adenoma . a few days after admission , he fell down while walking and was referred to our team . x - rays showed a displaced left femoral neck fracture ( fnf ) and right humeral shaft fracture with poor bone quality . his humeral fracture was treated conservatively , and the fnf was treated with total hip replacement . three days later , he underwent parathyroidectomy . this case demonstrates the importance of a thorough investigation of progressive weakness even in a young individual and illustrates the importance of early diagnosis of parathyroid adenoma to avoid the devastating end results of this condition .

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furthermore , the site of lesion could indicate surgical excision to prevent continuous microtraumas [ 13 ] . surgical excision could determine loss of substance due to the dimension of the nevus that could not be easily directly repaired . the foreskin is a good autologous full - thickness skin graft in several conditions . the authors report the use of foreskin as skin graft to repair a loss of substance due to excision of an interdigital nevus of the foot . a four - year - old boy presented a 2 cm 1.5 cm congenital compound nevus entirely covering the plantar surface of the second finger of his left foot ( figure 1 ) . paediatric dermatologist 's indication was a radical excision because of the site and the dimension of this melanocytic lesion . primary closure of the skin defect secondary to radical excision of the lesion was not indicated because of the large loss of substance and the risk of retractive scar . then we performed circumcision and a radical excision of the nevus ( figure 3(a ) ) ; foreskin , trimmed in a rectangular shape ( figure 2 ) , was sutured into the residual defect ( figure 3(b ) ) . , the patient has normal use of the foot finger with no evidence of contracture ( figure 4 ) . congenital melanocytic nevus is a frequent condition in childhood ( 0,21% ) [ 1 , 2 ] . the role of these lesions in increasing incidence of cutaneous melanoma is discussed and the prophylactic removal of all congenital melanocytic nevi is not supported : however , the most congenital melanocytic nevi are removed on preventing criteria . the selective excision of suspicious nevi is indicated when the features of a possible malignancy are faced . these features can include change in size or colour , irregular borders , or development of ulcerations . other features that can justify excision are site and extension of the lesion , multinodular aspect , and the presence of other risk factors ( immunodeficiency , dysplastic nevus syndrome , and xeroderma pigmentosa ) . excision of larger lesions require the use of local plasty , free tissue skin graft , or even the prior use of a tissue expander .graft should be harvested from hairless areas where the skin is redundant ( groin , volar wrist crease , volar elbow crease , and ulnar side of the hypothenar eminence ) . foreskin as a source of skin graft most often been used in urethral reconstruction for congenital or acquired penile defects [ 5 , 6 ] , in burn reconstruction , most commonly for eyelid resurfacing , and in syndactyly repair [ 9 , 10 ] . newborn circumcision remains controversial ; this procedure has potential medical advantages ( decreased risk of cancer of the penis and urinary tract infections ) as well as disadvantages and risks ( bleeding , infection , meatitis , and scarred phimosis ) . in italy , neonatal circumcision is not routinely performed ; this intervention is electively carried out until three years of age to repair congenital phimosis and at all ages in cases of scarred phimosis , recurrent balanoposthitis , and urinary infections . therefore foreskin is frequently available as tissue graft in paediatric population . in our case , dimension and site ( difficult to control ) of melanocytic foreskin was available because the boy was also affected by congenital phimosis , so we did not look for another source of skin graft . the most common problem reported after the use of prepuce as donor skin is hyperpigmentation . in our case , hyperpigmentation was not a contraindication for the use of foreskin as skin graft because the lesion was hidden localizated . foreskin provides a skin of good elastic quality avoiding secondary retraction with a favourable rate of graft intake . therefore , this source of graft gives the advantage of the absence of scar prejudice at the donor site .

we report a four - year - old boy with a nevus covering all the plantar side of his second finger on the left foot . he was also affected by congenital phimosis . surgical excision of the nevus was indicated , but the skin defect would have been too large to be directly closed . the foreskin was taken as a full - thickness skin graft to cover the cutaneous defect of the finger . the graft intake was favourable and provided a functional repair with good aesthetic characteristic .

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patients with coronary artery disease ( cad ) and diabetes have higher mortality and morbidity than patients without diabetes . data from studies such as the uk prospective diabetes study suggest that very good glycemic control is associated with fewer cardiovascular events . hypoglycemia is a very common side effect of insulin therapy and , to a lesser extent , of treatment with sulfonylureas . risk factors for severe hypoglycemia include age , duration of diabetes , strict glycemic control , sleep , impaired awareness of hypoglycemia , renal impairment , c - peptide negativity and previous history of severe hypoglycemia . acute hypoglycemia provokes pronounced physiological responses , the important consequences of which are to maintain the supply of glucose to brain and promote hepatic production of glucose . hypoglycemia and the rapid changes in blood glucose have been shown to increase counter - regulatory hormones such as epinephrine and nor - epinephrine , which may induce vasoconstriction and platelet aggregation , thereby precipitating myocardial ischemia . autonomic activation , principally of the sympatho - adrenal system , results in end - organ stimulation and the profuse release of epinephrine which precipitates hemodynamic changes like tachycardia , increased peripheral systolic blood pressure , decreased central blood pressure and increased myocardial contractility with an increased ejection fraction . the increased activity of sympathetic nervous system and secretion of other hormones and peptides such as the potent vasoconstrictor endothelin have pronounced effects on intravascular coagulability and viscosity . increased plasma viscosity occurs during hypoglycemia because of an increase in erythrocyte concentration , while coagulation is promoted by platelet activation and an increment in factor viii and von - willebrand factor . endothelial functions may be compromised during hypoglycemia because of an increase in c - reactive protein , mobilization and activation of neutrophils and platelet activation . the catecholamine - induced increased myocardial contractility may induce ischemia in the myocardium in patients with cad . the greater oxygen demand is not met because of not only the rigid vessels , but also endothelial dysfunction with failure to vasodilate . several studies have shown that the hypoglycemia is associated with a significant lengthening of the corrected qt interval ( qtc ) in subjects with and without diabetes . these changes are likely seen because of increased catecholamine release during hypoglycemia , and qtc prolongation , in particular , could lead to a high risk of ventricular tachycardia and sudden death . hyperinsulinemia and increased secretion of catecholamines may lead to hypokalemia during hypoglycemia , thus potentiating cardiac repolarizing abnormalities . effects of antecedent hypoglycemia on cardiac autonomic regulation may contribute to the occurrence of adverse cardiac events . abnormalities in high - frequency and low - frequency heart rate variability have been associated with hypoglycemia and increased catecholamine release . however , other studies did not find any associations between heart rate variability , hypoglycemia and increased catecholamine release . episodes of hypoglycemia have been found to be associated with rise in inflammatory cytokines including interleukin ( il)-6 , il-8 , tumor necrosis factor ( tnf)- , c - reactive protein and endothelin-1 . these inflammatory cytokines result in endothelial injury and abnormalities in coagulation , resulting in rise of cardiovascular events . inflammatory cytokines like il-1 have also been shown to increase the severity of hypoglycemia , thus perpetuating a positive feedback cycle . vessel wall stiffness was found to be increased during hypoglycemia in patients with type-1 diabetes of longer duration than those with shorter duration of diabetes . thus , hypoglycemia may increase the risk of cardiovascular events , especially in subsets of patients with longer duration of diabetes . inflammation and endothelial dysfunction could potentially be the aggravating factors that contribute to increased cardiovascular risk with severe hypoglycemia , especially in the subset of patients with pre - existing cardiovascular disease , diabetes , and severe autonomic neuropathy . a direct relationship between hypoglycemia and fatal cardiovascular event is difficult to demonstrate as blood glucose and cardiac monitoring are seldom performed simultaneously . in the accord study , excess of deaths was noted in the intensive treatment arm , which led to discontinuation of study . in the smaller study of veterans with type-2 diabetes , veterans affairs diabetes trial ( vadt ) , ecg changes , including ectopic activity , flattening of t - wave , st depression , ventricular tachycardia , and atrial fibrillation , have been reported in cases of low plasma glucose . sudden death during sleep has been described in patients with type-1 diabetes , the mechanism being a significant cardiac arrhythmia induced by nocturnal hypoglycemia . many of these patients have no evidence of severe hypoglycemia - induced neuronal damage at autopsy , implying that a cardiac arrhythmia had been triggered by hypoglycemia , resulting in sudden death . despite the high frequency of nocturnal hypoglycemia in young patients with type-1 diabetes , sudden nocturnal death ( dead in bed syndrome ) is rare . evidence is accumulating that severe hypoglycemia can provoke adverse cardiovascular outcomes such as myocardial ischemia or cardiac arrhythmia . episodes of severe hypoglycemia are common during intensive therapy in type-1 and type-2 diabetes in the out - patient as well as in - patient setting . larger clinical trials are required to look specifically at the association between hypoglycemia and cardiovascular events and to determine the mechanism further . the challenge to the physicians is to lower blood glucose to normal values to decrease the risk for long - term complications and at the same time minimize hypoglycemia and hypoglycemia - associated morbidity and mortality .

hypoglycemia is a very common side effect of insulin therapy and , to a lesser extent , of treatment with oral hypoglycemic agents . severe hypoglycemia can precipitate adverse cardiovascular outcomes such as myocardial ischemia and cardiac arrhythmia . these are mainly secondary to autonomic activation which results in hemodynamic changes , vasoconstriction and rise in intravascular coagulability and viscosity .

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a 72-year - old man presented with a two - week history of a red painful right eye . he was a soft contact lens wearer and wore his lenses for one month at a time , taking them out at the end of each day and disposing of the lenses at the end of the month . he had no past ocular , medical , or drug history of note . on examination he had an inflamed eye with a central corneal epithelial defect 3.0 mm 3.5 mm in size , with surrounding superficial and midstromal infiltration . a corneal scrape of the right eye was performed for gram staining and cultures were obtained on blood , chocolate , and sabouraud s agar , as well as non - nutrient agar overlaid with escherichia coli . he was started on empirical antimicrobial treatment with topical ofloxacin hourly by day and night . there was no growth of any other organisms , and the topical ofloxacin was discontinued . his treatment was switched to topical , ie , hourly polyhexamethylene biguanide 0.02% , hourly propamidine isethionate 0.1% ( brolene ) , prednisolone 0.5% four times daily , and atropine 1% three times daily . over the following three months , his treatment was slowly tapered to topical , ie , polyhexamethylene biguanide four times daily , brolene four times daily , and prednisolone 0.1% twice daily as his clinical picture improved . at three months after initial diagnosis he complained of the right eye being red and sore again for several days . on examination the gram stain showed gram - positive cocci in chains and he was empirically commenced on topical penicillin hourly , which was added to his prophylactic treatment for acanthamoeba . after 48 hours , the blood agar grew streptococcus viridans which was shown to be sensitive to penicillin . topical penicillin was tapered down to twice daily as his condition improved over the following month . four months after initial diagnosis , he again complained of a red painful right eye for one week . on examination fine debris and hairs were noted in the base of the abscess and were removed by scraping ( figure 1 ) . the penicillin drops were stopped and he was treated with topical ofloxacin initially hourly and then tapered off over one month . five months after initial diagnosis he complained of a one - day history of a painful right eye . fine strands were noted on the surface of a central corneal abscess and were removed during the corneal scrape . he was treated with topical ofloxacin and gentamicin , to which the organism was found to be sensitive . the organism was resistant to ciprofloxacin , chloramphenicol , penicillin , cefuroxime , and fusidic acid . as he improved , fine fibrils were again seen in the healing cornea and were removed and sent for microscopy . this confirmed that the fibrils were synthetic and were not observed in previous / present cultures , and presumed to be derived from tissues which the patient was constantly using to wipe his eye . the cornea continued to heal and topical treatment for acanthamoeba ( including steroids and ofloxacin ) were continued and tailed off over a further three months . in all episodes of infection , antibiotic sensitivities were determined using disc diffusion susceptibility testing . in all episodes of reinfection , scraping for acanthamoeba was performed to exclude reactivation of the initial infection . this patient had acanthamoeba keratitis secondary to contact lens wear and then developed three episodes of bacterial keratitis . microbial keratitis results from the interaction of a broad spectrum of pathogens and a diverse range of host responses . recurrence is rare in the absence of predisposing factors , such as contact lens wear , ocular surface and corneal disease , corneal anesthesia , exposure , trauma , or previous corneal surgery.1 when suspected , corneal scraping is mandatory in order to provide material for a microbiological diagnosis , debride necrotic tissue , and enhance antibiotic penetration . corneal biopsy may be necessary in some cases of recurrence . the initial bacterial infection in this patient presumably was facilitated by the underlying acanthamoeba keratitis and the use of corticosteroids.2,3 the presence of synthetic fibrils on the ulcer base presumably contributed to the infection recurrences by acting as a nidus for organisms and by interfering with corneal healing . it is noteworthy that the cornea finally healed with no further infections once the patient was instructed not to wipe his eye with a paper tissue . unfortunately it was not possible to obtain cultures directly from the tissue fibrils to confirm their direct association with the infective organisms . the first two bacterial infections were due to virulent organisms , such as pseudomonas and streptococcus . the culture of stenotrophomonas maltophilia from the third corneal abscess was feasible because this is an opportunistic organism and infections typically occur in patients with compromised ocular surface and trauma.4,5 it has been reported previously in patients following penetrating keratoplasty.3,6,7 this is the first case report of acanthamoeba keratitis occurring in a contact lens wearer with preceding protozoal and bacterial keratitis . the characteristically resistant antibiogram of s. maltophilia may limit the therapeutic options . fortunately in this patient treatment

recurrence of microbial keratitis in the presence of protozoal infection is very rare and infrequently reported unless predisposing factors are present . the association of recurrent microbial keratitis and synthetic microfibrils has never previously been reported to our knowledge . this single interventional case study describes the clinical course and treatment of a contact lens wearer who was treated for acanthamoeba keratitis with superinfection from bacterial organisms in the presence of synthetic microfibrils . the presence of synthetic fibrils on a corneal ulcer base may act as a nidus for pathological organisms and interfere with normal corneal healing . this may result in infection recurrence and the growth of resistant opportunistic organisms .

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pilomatricoma usually appears in the first two decadesusually , an asymptomatic lesion with an occasional inflammation . pilomatricoma usually appears in the first two decades usually , an asymptomatic lesion with an occasional inflammation . usually , they present as a solitary nodule / cystic lesion distributed on head , neck , and upper trunk area . sometimes , there is associated inflammation . a 36-year - old obese , hypothyroid female patient presented in surgery outpatient department of a tertiary care medical college of eastern bihar with a bluish - red , nodular lesion over the upper back [ figure 1 ] . she gave a history of 68 months duration and complained of some sticky material coming out from the lesion intermittently whenever there was pain and redness . she was treated outside with intermittent antibiotic and anti - inflammatory agents with an apparent resolution of the symptoms followed by recurrence in a few weeks . on examination , we found a violaceous nodulo - cystic lesion on the upper back with a size of 2 cm 1.5 cm ; the center of the lesion was eroded , and there was a visible yellow - colored cheesy discharge from that area [ figure 2 ] . depending on the site , the nodulo - cystic nature of the lesion , the recurrent history of inflammation and discharge , a provisional diagnosis of the ruptured epidermal cyst was placed . the patient was managed conservatively till discharge subsided , and the epithelialization of central area was complete . nodule covered with yellow cheesy discharge and crust in scanner view , the epidermis was acanthotic . in the dermis , there was a nodular basaloid cell proliferation extending from papillary to reticular dermis [ figure 3 ] . there was also the proliferation of squamoid cells with the disappearance of nucleus leaving behind a shadow ( ghost cells / shadow cells ) [ figures 4 and 5 ] . the dermis was covered by mononuclear inflammatory infiltrate along with the formation of foreign body type giant cells [ figures 6 and 7 ] . in view of these findings h and e ( scanner view ) : basaloid cells h and e ( scanner view ) : ghost cells ghost cells with nuclear shadow , ( h and e , 40 ) stromal inflammation , ( h and e , 10 ) foreign body giant cells , ( h and e , 40 ) in scanner view , the epidermis was acanthotic . in the dermis , there was a nodular basaloid cell proliferation extending from papillary to reticular dermis [ figure 3 ] . there was also the proliferation of squamoid cells with the disappearance of nucleus leaving behind a shadow ( ghost cells / shadow cells ) [ figures 4 and 5 ] . the dermis was covered by mononuclear inflammatory infiltrate along with the formation of foreign body type giant cells [ figures 6 and 7 ] . in view of these findings h and e ( scanner view ) : basaloid cells h and e ( scanner view ) : ghost cells ghost cells with nuclear shadow , ( h and e , 40 ) stromal inflammation , ( h and e , 10 ) foreign body giant cells , ( h and e , 40 ) in scanner view , the epidermis was acanthotic . in the dermis , there was a nodular basaloid cell proliferation extending from papillary to reticular dermis [ figure 3 ] . there was also the proliferation of squamoid cells with the disappearance of nucleus leaving behind a shadow ( ghost cells / shadow cells ) [ figures 4 and 5 ] . the dermis was covered by mononuclear inflammatory infiltrate along with the formation of foreign body type giant cells [ figures 6 and 7 ] . in view of these findings h and e ( scanner view ) : basaloid cells h and e ( scanner view ) : ghost cells ghost cells with nuclear shadow , ( h and e , 40 ) stromal inflammation , ( h and e , 10 ) foreign body giant cells , ( h and e , 40 ) usually , they appear in early age , the average age of onset being childhood to adolescence . they usually present as a solitary firm nodule or cyst distributed on any nonglabrous area , but mostly restricted to head - neck and upper trunk . sometimes , the lesion shows an inflammatory sign . in histology , usually there is a cyst formation with matrical keratinization . there are two kinds of cells ; outer basaloid matrical cells and central squamatized cells with the remnant of nuclear outline ( shadow or ghost cells ) . in the early stage , particularly , in the elderly population , the basaloid cells predominate , and these tumors are called proliferating pilomatricoma . the treatment is basically surgical excision ; recurrence has been reported following the surgical excision . in our case , the pilomatricoma appeared at 36 years of age , and clinically mimicked ruptured epidermal cyst . the goal of this presentation is to highlight the fact that pilomatricoma may appear late in life and matrical cyst of pilomatricoma can rupture and give rise to a clinical picture mimicking ruptured epidermal cyst .

pilomatricoma is a benign tumor arising from the hair matrical cells . most pilomatricomas appear in the first two decades of life as a solitary skin to a bluish colored nodule on head - neck area with an occasional sign of inflammation . here , we present a case of pilomatricoma which appeared at 36 years of age with a history of recurrent inflammation and discharge mimicking ruptured epidermal cyst .

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systemic chemotherapy can lead to a variety of ocular complications , such as cicatricial ectropion , nasolacrimal duct stenosis , conjunctivitis , keratitis , cataract , macular edema , retinopathy , and optic neuropathy ( 1 , 2 ) . although bulbar perforation with orbital cellulitis has been reported in an immunocompromised patient , corneal perforation has not been documented in patients undergoing systemic chemotherapy ( 3 ) . we report a case of corneal perforation with preseptal cellulitis in a patient treated with systemic chemotherapy for acute lymphocytic leukemia ( all ) . a 17-yr - old female patient undergoing systemic chemotherapy for all was referred to our hospital due to swelling and pain of the right upper lid for two days . laboratory examination showed leukocytes 40 cells/l , erythrocytes 3.610 cells/l , hemoglobin 10.8 g / dl , hematocrit 30.6% , and thrombocytes 3310 cells/l . the patients received induction chemotherapy ( vincristine , prednisolone , daunorubicin , and l - asparaginase ) . there were no abnormal findings , other than diffuse swelling of the right upper eyelid . artificial tear eye drops were used for the treatment of superficial punctate erosions . on the 12th day , spontaneous bloody and purulent discharge from the upper palpebral conjunctiva occurred in the right eye . systemic antibiotics ( meropenem ) were maintained per the antibiotic sensitivity test . on the 16th day , periorbital swelling decreased , however , corneal melting and perforation with iris prolapse was noted in the right eye ( fig . seven months after surgery , visual acuity in the right eye was 20/300 , and intraocular pressure was 14 mmhg . ocular complications associated with systemic chemotherapy can be divided into complications in the adnexa , anterior segment , and posterior segment . previously reported complications in the anterior segment include conjunctival injection , conjunctivitis , corneal edema , keratitis , and corneal opacity ( 1 , 2 ) . anticancer agents such as cytosine arabinoside , 5-flurouracil , carmustine , deoxycoformycin , and tamoxifen have been known to cause corneal toxicity ( 1 , 2 , 4 , 5 ) . in our case , vincristine , prednisolone , daunorubicin , and l - asparaginase were used for combination chemotherapy . to the best of our knowledge , corneal toxicity associated with these anticancer agents has not been reported . serratia marcescens , a motile , gram - negative coccobacillus , is an emerging opportunistic pathogen noted for causing urinary , respiratory , blood stream , and ocular infections ( 6 ) . preseptal cellulitis in the present case was caused by s. marcescens because the patient was immunocompromised . extracellular protease produced by s. marcescens is considered a major corneal destructive factor . in an experimental study of rabbit cornea , serratia protease preparations have caused rapid and extensive liquefactive corneal necrosis , descemetocele formation , and corneal perforation ( 7 ) . it is possible that s. marcescens in the purulent discharge directly invaded the cornea with superficial punctate erosions and caused corneal melting and perforation in the immunocompromised patient . this hypothesis is supported by two facts : first , s. marcescens is a virulent organism . second , corneal melting and perforation occurred in the area in direct contact with the infected upper palpebral conjunctiva . in conclusion , physicians should consider the possibility of serious ocular complications , such as severe bacterial keratitis or corneal perforation in cases of preseptal cellulitis caused by a virulent organism , particularly in immunocompromised patients .

we report a case of corneal perforation with preseptal cellulitis in a patient with acute lymphocytic leukemia ( all ) . a 17-yr - old female patient who was undergoing combination chemotherapy for all was referred due to upper lid swelling and pain in the right eye for 2 days . visual acuity in the right eye was 20/20 . initial examination showed no abnormal findings , other than swelling of the right upper eyelid . computed tomography showed a finding of preseptal cellulitis . microbiologic study of bloody and purulent discharge revealed serratia marcescens . corneal melting and perforation with iris prolapse were detected in the right eye on the 16th day . emergent tectonic keratoplasty was performed . seven months after surgery , visual acuity in the right eye was 20/300 , and the corneal graft was stable .

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cervical spine spondylolysis is the most commonly caused by high energy trauma to the upper cervical spine in the form of a " hangman 's " fracture of c2 vertebrae7 ) . however , congenital cervical spine spondylolysis is discovered by incidental radiographic finding , neck pain after minor trauma , a clunking or clicking neck and rarely neurologic compromise1 - 3,6,8,10 ) . this rare condition potentially signifies an unstable cervical spine . we will report a case of congenital isthmic defect of cervical spine and describe a cause of cervical isthmic defect , diagnostic radiographic finding and appropriate treatment of cervical spondylolysis or spondylolithesis a 23-year - old female suffered from a hyperextension injury to cervical spine after a traffic accident . following this injury , she experienced mild to moderate degree of nuchal and back pain . plain radiograph of the cervical spine showed bilateral pars interarticularis defect at the c6 level with cleft - bow - tie configuration , hypoplastic pedicle , grade i spondylolisthesis of c6 on c7 and spinal bifida at c6 ( fig . 1 ) . computed tomography of the c6 vertebrae showed bilateral corticated defects of the c6 articular mass , bilateral hypoplastic pedicles , lamina and spinal bifida of c6 and apparent neural foraminal enlargement at c6 - 7 ( fig . the magnetic resonance image of spine showed grade i spondylolisthesis c6 - 7 , degenerative change of c6 - 7 disc and acute compressed fracture at 4 thoracic vertebrae . she was subjected to controlled flexion - extension views of the cervical spine , which confirmed stability of the segment with less than 10 degrees of anterior angulations ( fig . she was treated with the semihard - orthosis brace immobilization for 8 weeks followed by repeated lateral cervical spine radiography . after 10 months to injury , she was symptom free with full range of motion of cervical spine . reported cervical spondylolysis are incidentally discovered except the " hangman 's " fracture1 - 3,6,8,9,10 ) . cervical spondylolysis is less prevalent than lumbar spondylolysis , only about 100 cases have been reported in the literature worldwide , mainly in adults1 ) . there is often no clear uniform mechanism of the injury , or the force causing the injury is often not strong enough to explain all of observed cervical anomaly . incidentally discovered cervical spondylolysis could combine with another anomaly like spinal bifida , dysplastic lamina or transverse process ( table 1 ) . the etiology of cervical spondylolysis is unknown , but congenital basis is asserted because no history of trauma is elicited in the majority of cases2,3,5 ) . another basis is nonunion of vertebra after birth , infantile fracture or repetitive microtrauma1,6,9 ) . the vertebral body will articulate with the vertebral arch at the neurocentral joints at birth , with fusion occurring between the ages of 5 and 8 years5,7 ) . if congenital defect is invalid , problem of ossification or chondrification might occur during vulnerable period from birth to fusion at neurocentral joint . proposed that this condition was caused by repetitive microtrauma resulting in stress fractures to the pars region similar to the process proposed in lumbar spondylolysis5 ) . they further proposed c6 to be the commonest site to be affected as , this being a transitional vertebra , it was subjected to stress more often . some of these theories can be put together and it may be suggested that a dysplastic spine is more susceptible to trauma or stress and leads to spondylolysis6 ) . we could clarify that our case was associated with congenital basis because of well corticated margin in defected area combined with another congenital lesion and no trauma history . congenital cervical spondylolysis , an embryologic developmental defect , occurs the most commonly at c61,2,6,9,10 ) . the overall frequency is unknown , and most cases are detected incidentally ( table 1 ) conventional radiolographic findings of cervical spondylolysis included a deformed articular mass , hypoplastic or dysplastic pedicle , spinal bifida and anterolisthesis1 - 3,6,8 - 10 ) . the computed tomography is the most useful image modality , because it shows the exact bony structures and three dimensional images . the magnetic resonance image can aid identification of combined soft tissue damage , cord injury and bony contusion at trauma lesion . however , magnetic resonance image is not useful for diagnosis of cervical spondylolysis . in our case , cervical magnetic resonance image showed acute compressive fracture at 4 thoracic vertebrae and degenerative disc change . misdiagnosis occurred for traumatic unilateral interfacet dislocation , a chronic nonunited articular mass fracture or congenitally absent pedicle4,10 ) . for example , unilateral interfacet dislocation is characterized by spondylolisthesis greater than 3 mm , rotation without a superimposed articular mass , and misalignment of the spinous process4 ) . the cervical spondylolysis can cause variable symptoms like neck pain , muscle spasm or neurologic deficit . symptomatic cervical spondylolysis need adequate treatment because previously existing cervical spondylolysis is vulnerable to trauma . the treatment of cervical spondylolysis depends on instability . if the cervical spine is stable , conservative approach is desirable for treating symptomatic cervical spondylolysis . our case was stable but needed hard orthosis because of the fracture of 4 thoracic vertebrae .

we report a case of rare cervical isthmic spondylolisthesis of c6 - 7 combined occult spinal bifida at c6 , and review the radiologic finding , different diagnosis and treatment . a 23-year old female presented nuchal , back pain after traffic accident . radiologic finding showed the 6th cervical isthmic defect , spondylolisthesis and dysplasia . the patient was conservatively treated about 8 weeks , and 10 months after injury , she was symptom free with full range of motion of cervical spine and she was followed up . cervical spondylolysis is a very rare condition . this clinical importance is vulnerable to trauma . for whatever reasons , symptomatic patients need to be treated by conservative or surgical option .

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l.c.c . owns equity in , receives compensation from , and serves on the board of directors and scientific advisory board of agios pharmaceut - icals . agios pharmaceuticals is identifying metabolic pathways of cancer 135 cells and developing drugs to inhibit such enzymes in order to disrupt tumor cell growth and survival . was partially supported by a pancan - aacr pathway to leadership award and a dale f. frey award for breakthrough scientists from the damon runyon cancer research foundation ( dfs-09 - 14 ) .

abstractserine metabolism is likely to play a critical role in cancer cell growth . a recent study reports the identification of a novel small - molecule inhibitor of serine synthesis that targets 3-phosphoglycerate dehydrogenase ( phgdh ) , the first enzyme of the serine synthesis pathway , and selectively abrogates the proliferation of phgdh overexpressing breast cancer cells .

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colonic necrosis has been described as a rare complication after the administration of kayexalate [ 1 , 2 ] . in this case study , we present a case of calcium polystyrene sulfonate - induced colonic necrosis and perforation to remind clinicians of this rare , but dangerous , toxicity associated with this commonly used medication . a 78-year - old woman with stage 4 chronic kidney disease ( ckd ) due to chronic pyelonephritis , and a right hypoplastic kidney was presented to our emergency department with a 2-day history of abdominal pain . her medical history included epilepsy that was treated with carbamazepine , hypertension and hyperlipidaemia , for which she received carvedilol and atorvastatin . because of the persistent hyperkalaemia , she was treated with calcium polystyrene sulfonate at 30 g / day ( anti - potassium granule ; assos drug , istanbul , turkey ) . as a result of the ckd , she was prescribed calcitriol for a mineral and bone disorder , darbepoetin alpha for anaemia and sodium hydrogen carbonate for acidosis . upon initial physical examination , her abdomen was non - distended with normoactive bowel sounds , but there was diffuse tenderness in her abdomen . significant laboratory values at the time of admission included sodium 138 meq / l , potassium 4.6 meq / l , ph 7.20 , bicarbonate 13 meq / l , blood urea nitrogen 71 mg / dl , creatinine 2.6 mg / dl , leukocytes 15 500/mm , eosinophils 110/mm , haematocrit 32.7% , platelets 221 000/mm and c - reactive protein 10.9 mg / dl . on the second day of hospitalization , the abdominal pain worsened and free air under the diaphragm was found on abdominal radiography . her sigmoid colon was found to be necrotic and perforated . a biopsy was performed and the perforated segment was repaired by primary closure . the surface of the deep ulcer contained necroinflammatory debris and various sized fragments of basophilic crystalloid material with angulated margins on microscopic examination ( figure 1a and b ) . also , there were no features of chronic colitis , including inflammatory bowel disease or chronic ischaemic colitis . ( a ) the basophilic crystalloid material in the necroinflammatory background of the ulcer surface ( haematoxylin and eosin , original magnification , 100 ) . ( b ) the basophilic crystalloid material in the necroinflammatory background of the ulcer surface ( haematoxylin and eosin , original magnification , 400 ) . sodium polystyrene sulfonate can also bind intraluminal calcium , leading to constipation , fecal impaction and subsequent bowel obstruction or perforation . gerstman et al . reported a 0.27% overall incidence , with a higher incidence ( 1.8% ) during the postoperative period . sorbitol enemas , along with experimental evidence suggesting that the necrosis was due to sorbitol rather than the kayexalate in presence of uraemia . extensive transmural necrosis was noted in rats receiving enemas of sorbitol or kayexalate in sorbitol in both the uraemic and non - uraemic groups . as in this case report , renal failure may be an important facilitating factor in the pathogenesis of the necrosis . in contrast to the experimental data , our case was treated with calcium polystyrene sulfonate orally , not rectally . rashid et al . noted that kayexalate in sorbitol given as an enema or orally to treat hyperkalaemia has been reported to induce intestinal necrosis in uraemic patients . they studied clinical and pathologic features of 15 patients and observed kayexalate crystals in tissue specimens from surgical resections and endoscopic biopsies . one possibility is elevated renin levels , commonly seen in renal insufficiency , that predispose the patient to non - occlusive mesenteric ischaemia via angiotensin - mediated vasoconstriction . one gram of kayexalate possesses a theoretical in vitro exchange capacity of 23.1 meq of potassium and in vivo capacity of 1 meq . emmett et al . reported that in vivo potassium - binding capacity may be lower than previously estimated , more on the order of 0.40.8 meq / g of kayexalate resin . in contrast to other minor digestive complications associated with kayexalate treatment , colonic perforation results in significant morbidity and mortality . as a result , potassium exchange resins may , although rarely , induce a colonic perforation , and this diagnosis should be considered in a patient treated as such in case of acute abdomen . the clinicians must be aware of the possible rare and serious complications of potassium exchange resins .

sodium or calcium polystyrene sulfonate ( kayexalate or analog ) is an ion - exchange resin commonly used to treat hyperkalaemia in patients with chronic kidney disease . it is known to cause digestive complications , such as nausea , vomiting and constipation . although rare , colonic necrosis and perforation are very severe complications associated with the medication . in this case report , we present a case of calcium polystyrene sulfonate - induced colonic necrosis and perforation to remind clinicians of this rare , but dangerous , toxicity associated with this commonly used medication .

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development of spontaneous pneumopericardium is a very rare complication of tuberculosis with coexisting human immunodeficiency virus ( hiv ) infection . to the best of our knowledge only three cases of pneumopericardium with pulmonary tuberculosis concomitant with hiv infection[13 ] have been reported so far . a 30-year - old hiv - positive male smoker on antiretroviral therapy ( art ) presented with complaints of cough with expectoration and fever for two and a half months . cough was moderate in intensity and was present throughout the day with worsening at night . patient had intermittent fever with an evening rise . there were associated complaints of decreased appetite and loss of weight . bp was 110/70 mm of hg and temperature was 99.2f with a respiratory rate of 20/min . a thin hyper - lucent line was present lining the lateral cardiac borders suggesting possibility of pneumopericardium [ figure 1 ] . the air or gas in x - ray chest did not rise above the upper level of pericardium in standing erect position differentiating it from pneumothorax and pneumomediastinum . computed tomography ( ct ) scan of the chest confirmed the presence of pneumopericardium [ figure 2 ] . it also showed a well - defined thick walled cavity with necrotic areas in apicoposterior segment of left upper lobe and bilateral infiltration with minimal bronchiectatic changes [ figure 3 ] . x - ray chest pa view showing patchy infiltration in both the upper zones with pneumopericardium ct scan showing bilateral infiltration with pneumopericardium ct scan showing cavitary lesion on the left side with pneumopericardium barium swallow fluoroscopy ruled out any esophagopericardial fistula . the patient did not have any symptoms related to pneumopericardium and its detection was purely incidental . patient was started on anti - tubercular treatment ( att ) as per the guidelines of revised national tuberculosis control program . pneumopericardium was first described by bricheteau in 1844 who named bruit de moulin ( water wheel sound ) associated with pneumopericardium . pneumopericardium occurs typically because of the breech of the pericardium by traumatic or nontraumatic causes . in adults , 60% of pneumopericardium results from trauma either sharp or blunt . iatrogenic factors include thoracocentesis , thoracic surgery , endotracheal intubation , sternal bone marrow puncture , or positive pressure mechanical ventilation . noniatrogenic causes can be underlying disease processes like infected fluid or gas producing organisms in the pericardial sac , fistulous communication between pericardium and other air containing structures such as bronchus , esophagus , or stomach and amebic liver abscess . other causes also include foreign body aspiration , physical exertion , parturition , severe cough , acute asthma , cocaine inhalation , chlorine gas exposure , and forceful emesis . the possible mechanism of pneumopericardium in this case could either be a severe bout of cough or a fistulous communication of pericardium with an infected contiguous organ . the rise in intra - alveolar pressure above atmospheric pressure due to increased bout of coughing may lead to rupture of alveoli and the released air moves to hilar area , mediastinum , and through pericardial reflections on the pulmonary vessels in the pericardial cavity . the parietal pericardium is reflected on the visceral pericardium near ostia of the pulmonary veins , the weakest histological area . nectrotizing pulmonary process like tuberculosis in the setting of hiv could have resulted in the fistulous communication between the lung and pericardium leading to pneumopericardium in this patient . pneumopericardium may be symptomatic or asymptomatic depending upon the quantity of air in the pericardium . the patient with a small pneumopericardium may be asymptomatic and the cardiac examination may be normal . it may only be diagnosed incidentally on a chest radiograph and the gas usually does not rise above the upper limit of the pericardium in the erect position , which differentiates the pneumopericardium from pneumomediastinum as in this patient . in large pneumopericardium the patient may be symptomatic and the note may be tympanic and heart sounds may be metallic . this complication is thought to be caused by a ball valve mechanism preventing air from leaving the pericardial space . pneumopericardium unlike pneumothorax often does not require any specific treatment and is usually self - limiting as in the present case . treatment is required in cases of large and symptomatic pneumopericardium or in patients of tension pneumopericardium , which can be a needle aspiration or tube decompression . oxygen therapy in high concentrations as in management of pneumothorax can also be helpful in absorption of air .

pneumopericardium is defined as a collection of air or gas in the pericardial cavity . it is a rare entity and spontaneous pneumopericardium is even rarer . it is a rare complication of tuberculosis and human immunodeficiency virus and just three cases have been reported so far .

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it is caused by trauma , stress , sepsis , adrenal tumors , anticoagulation , hemorrhagic disorders and pregnancy . idiopathic adrenal hematoma. the symptoms vary from subclinical to clinical , such as nausea , abdominal pain , fever and hypotension due to circulatory collapse . adrenal tumors associated with hemorrhage primarily include pheochromocytomas , adrenocortical cancers and metastatic lesions from other organs . from a clinical perspective , whether the lesion is benign or malignant is an important issue but is difficult to determine prior to surgery . a 59-year - old japanese man was referred to our hospital for evaluation of a 7.0 cm mass in the posterior segment of the liver or right adrenal gland in november 2009 . this mass was discovered when abdominal ultrasonography was obtained to evaluate a weight loss of 8 kg from may to november 2009 . his body temperature was 37.0c , his blood pressure was 124/73 mm hg , and his pulse was regular at 67/min . hormonal examination revealed a slightly elevated metanephrine level ( 0.35 mg / day ; normal range 0.040.19 ) . tumor marker levels , including serum -fetoprotein , carcinoembryonic antigen and carbohydrate antigen 19 - 9 were all within normal ranges . only pivka - ii was slightly elevated at 53 mau / ml ( normal < 37 ) . abdominal computed tomography ( ct ) demonstrated a well - demarcated , 7.0 cm lesion in either the posterior segment of the liver or in the right adrenal gland . the peripheral region was slightly enhanced and the central part was hypovascular , indicating central necrosis ( fig . there was no evidence of lymph node enlargement or distant metastasis . on magnetic resonance imaging ( mri ) , , the mass showed arterial enhancement and a ct filling defect in the right superior and inferior adrenal arteries and arterial portography , respectively ( fig . i - meta - iodo - benzylguanidine ( mibg ) scintigraphy demonstrated no aberrant accumulation in the mass , which ruled out the possibility of a pheochromocytoma . for the purposes of securing a definite diagnosis and treatment , an open right adrenalectomy was planned for march 2010 . the resected specimen was 7.0 5.5 cm in size , and the cut surface of the mass was heterogeneous and dark red and white in appearance ( fig . hematoxylin - eosin staining revealed hemorrhage , necrosis and hemosiderin deposit with normal adrenal tissue . the patient had an uneventful postoperative course and was discharged home on postoperative day 12 . small adrenal branches from the three main adrenal arteries form a subcapsular plexus , and the gland is drained by relatively few venules . for example , stress increases adrenal vascularity and increases adrenal venous pressure due to vasoconstriction , resulting in intraglandular hemorrhage . idiopathic adrenal hematomas are difficult to diagnose prior to surgery . in japan , koizumi et al . described 14 cases of idiopathic adrenal hematomas from 1983 to 2010 . of these 14 cases , 13 had been suspected to be adrenal tumors including malignant lesions preoperatively , but were then found to be idiopathic adrenal hematomas on pathological examination after surgery . only 1 patient was diagnosed with an idiopathic adrenal hematoma without surgery . the adrenal hemorrhage was not suspected to be caused by an adrenal tumor on the basis of ct , hormonal assay and mibg results , and this mass spontaneously regressed after 2 weeks . although imaging modalities such as ct and mri are helpful in detecting adrenal hemorrhage , it is difficult to determine whether the hemorrhage is associated with tumors or not . adrenal hemorrhage associated with pheochromocytoma ( 48% ) was the most frequently observed situation , and the second most frequently observed was a malignant lesion ( 20% ) such as adrenocortical cancer or metastasis from another organ . hematomas derived from pseudocysts or adenomas comprised 17% of all cases . therefore , even if pheochromocytoma is ruled out by hormonal evaluations and imaging studies , the possibility that the mass may be a malignant lesion remains approximately 50% . furthermore , the size of adrenal incidentalomas , which are adrenal gland masses discovered serendipitously on imaging , is an important factor in differentiating benign tumors from malignant lesions . nieman recommended routine surgical resection for adrenal incidentalomas > 4 cm in diameter without a clear - cut diagnosis . in our case , metastatic liver tumors , hemorrhaged hepatic cellular carcinoma with central necrosis and adrenal tumor were raised as differential diagnoses at the time of the initial consultation because of ring enhancement with hypovascularity on enhanced ct and a slightly elevated pivka - ii level . subsequently , abdominal angiography revealed the origin of the mass to be the right adrenal gland . although vital signs , laboratory data and mibg scintigraphy ruled out the possibility of a pheochromocytoma , the possibility of another malignancy could not be completely ruled out . therefore , surgery was strongly recommended for a definite diagnosis as well as for treatment . the reason for the patient 's weight loss and the increase in the pivka - ii in our case was not clear , but may have been related to exhaustion during the summer season from may until november that kept him from eating sufficiently . in fact , his weight loss ceased after he consulted our hospital , and the pivka - ii value also normalized prior to surgery . in conclusion , we report herein a case of idiopathic adrenal hematoma . an accurate diagnosis of idiopathic adrenal hemorrhage is quite difficult to make prior to surgery . some imaging modalities are useful in generating a differential diagnosis , but if the potential for malignancy is not excluded , surgical resection should be taken into consideration .

we report herein a case of idiopathic adrenal hematoma . a 59-year - old japanese man was referred to our hospital for evaluation of a 7.0 cm mass in the right upper abdominal cavity . the tumor was suspected to originate from either the posterior segment of the liver or the right adrenal gland . his chief complaint was weight loss of 8 kg over the previous 6 months . he had no past medical history and took no medications , including no anticoagulants . laboratory data were almost normal except for a slight elevation of pivka - ii . the origin of the tumor was found to be the adrenal gland , as angiography revealed the blood supply to the mass to derive from the right superior and inferior adrenal arteries . a fine needle biopsy of the lesion was unable to confirm the diagnosis . open right adrenalectomy was performed . the histopathological findings of the surgical specimen revealed a hematoma with normal adrenal tissue . in the absence of any obvious etiology , the diagnosis was idiopathic adrenal hematoma .

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congenital scrotal anomalies are unusual and include penoscrotal transposition , bifid scrotum , ectopic scrotum , and accessory scrotum ( as ) . among these , as is the least frequent , with only 42 cases reported in the english literature . as is characterized by additional scrotal tissue lacking a testis , besides a normally developed scrotum . various associated anomalies have been reported . in particular , contiguous subcutaneous tumor is the most frequently associated abnormality and is reported to be related to the etiology of as . although prenatal screening techniques have advanced , most reported cases of congenital perineal mass have been identified after birth . a 28-year - old woman was referred to our hospital for the evaluation of a fetal perineal mass at a gestational age of 31 weeks . prenatal ultrasonography and magnetic resonance imaging ( mri ) showed a mass of 1.0 1.2 cm located posterior to the scrotum in a male fetus . 1 ) the likely diagnosis was lipoma and the mass maintained a stable appearance until delivery . the male newborn was delivered vaginally at 38 weeks of gestation and his body weight was 2208 g. there were no specific symptoms after birth . arrowheads indicate a 1.0 1.2 cm mass located posterior to the scrotum . on physical examination , the soft peduncular mass , measuring 2.0 cm in diameter , was attached to a midperineal skin tag . there was also a rugged pigmented swelling on the mass , measuring 0.7 cm in diameter , which resembled the scrotum . the perineal mass showed high signal intensity on t1- and t2- weighted images and the signal intensity was suppressed on fat - suppressed t1-weighted images . mri revealed no associated abnormalities of the intraabdominal organs , musculoskeletal system , or genitourinary system . a. a soft peduncular mass with a rugged and pigmented swelling is located posterior to the normally developed scrotum . c. the mass is attached at the midperineum with a skin tag . the preoperative diagnosis was as with perineal lipoma , and we completely excised the mass under general anesthesia at one month of age . the postoperative course was uneventful , and the patient was discharged on the day after the operation . there was no recurrence or functional sequelae within a follow - up period of six months . a histological examination revealed that the peduncular mass consisted of mature adipose tissue . in this case , it was difficult to distinguish between lipoma and normal adipose tissue pathologically . however , our clinical diagnosis was lipoma because the peduncular mass was separated from normal perineal region by the skin tag . the rugged swelling on the peduncular mass showed smooth muscle fibers in the subcutaneous layer , which represented the tunica dartos . 3 ) the swelling was definitively diagnosed as as . histological examination . b. the peduncular mass smooth muscle fibers in the subcutaneous layer of the rugged swelling represent the tunica dartos . these swellings appear at four weeks of gestation and migrate to the caudal portion after 12 weeks of gestation . abnormal migration or early division of the labioscrotal swellings is possibly related to the etiology of congenital scrotal anomalies . the least frequent congenital scrotal anomaly is as , characterized by additional scrotal tissue without a testis , besides a normally developed scrotum . ( table ) characteristics of accessory scrotum various anomalies associated with as have been reported . in particular , contiguous subcutaneous tumor has a high incidence ( 72.5% ) of association with as . histologically , one case of subcutaneous tumor was lipoblastoma , three cases were hamartoma , and the others were consistent with lipoma . ( table ) it is assumed that the contiguous subcutaneous tumor is related to the etiology of as . sule hypothesized that as develops when intervening mesenchymal tissue ( i.e. , the developing subcutaneous tumor ) disrupts the continuity of the developing caudal labioscrotal swelling . however , the complete etiology of as is not explained by this hypothesis because as can occur with no contiguous tumor . takayasu hypothesized that as develops from the early division and teratoid growth of pluripotential labioscrotal tissue elements . however , two cases associated with skeletal abnormalities were located in the pubic area , and one case was located on the distal penile shaft . our case was detected at a gestational age of 31 weeks , and the other two reported cases were detected at 24 weeks and 32 weeks of gestation . a congenital perineal mass is unusual in itself , and most reported cases have been diagnosed after birth . however , detection is possible with careful prenatal screening . the differential diagnosis of a fetal perineal mass includes lipoma , lipoblastoma , infantile hemangioma , hamartoma , and choristoma . if a fetal perineal mass is detected during the antenatal period , it is important to look for any associated congenital anomalies . the prognoses of surgically treated patients are good , and only one has died , from an associated anomaly before surgery . the reported ages at surgery range from four days to 46 years ( median , nine months ) , and three adult cases are recorded in the literature . our patient was operated upon in the neonatal period because the mass was considered to be excisable without complications and the associated subcutaneous tumor had a low probability of malignancy . although a fetal perineal mass is difficult to diagnose , it can be detected with careful prenatal screening . many ass are associated with contiguous subcutaneous tumors , which are assumed to be related to the etiology of as .

abstractwe report a case of accessory scrotum ( as ) in the perineal region with peduncular lipoma , diagnosed prenatally . a male fetus of 31 weeks gestation was referred to our department with a perineal mass . prenatal ultrasonography and magnetic resonance imaging showed a mass of 1.0 1.2 cm located posterior to the scrotum . no other abnormalities were noted during pregnancy . the patient was delivered vaginally at 38 weeks of gestation . on physical examination , a soft peduncular mass with a rugged and pigmented swelling was located between the normally developed scrotum and the anus . there were no specific symptoms or any other associated congenital anomalies . we completely excised the mass at one month of age . a histological examination revealed lipoma , with tissue suggestive of scrotum , so a definite diagnosis of as was made . as is a rare congenital anomaly of the scrotum . we review the literature .

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the recognition that elevated intracranial pressure ( icp ) is transmitted through the optic nerve and its sheath has been known for many years . this physiological process is the basis for the physical exam finding of papilledema on fundoscopic examination . recently , interest has turned to measurement of the optic nerve sheath diameter ( onsd ) through non - invasive imaging technologies to provide surrogate markers for early elevated icp . in this issue of critical care , geeraerts and colleagues present their research correlating magnetic resonance imaging ( mri ) measurements of onsd with icp . in a retrospective review of 38 patients with traumatic brain injury requiring both invasive icp monitoring and mri , they found a significant positive relationship between onsd measured by mri and icp ( r = 0.71 ) . the best cut - off value to detect an icp > 20 cm h2o based on a receiver operating characteristic curve was found to be onsd = 5.82 mm with a sensitivity of 90% and a specificity of 92% . the optic nerve is surrounded by cerebrospinal fluid ( csf ) , which is contiguous with intracranial csf . increased icp is transmitted through this subarachnoid space causing distention of the dural optic nerve sheath , especially the retrobulbar segment . the optic nerve and its surrounding sheath can be imaged and measured on mri using a fat - suppressed t2-weighted sequence . mri has been used to demonstrate increased onsd in idiopathic intracranial hypertension , and interestingly , decreased onsd in csf hypotension . the onsd has also been shown on mri to decrease after drainage of subdural hematomas . the research presented by geeraerts and colleagues is unique in its comparison of onsd with simultaneous direct measurements of icp through invasive monitoring . their findings generally correlate with a growing body of research using bedside ultrasound measurements of onsd to detected elevated icp . original research with lumbar intrathecal infusions performed by hansen and helmke demonstrated rapid changes in the onsd with alteration of csf pressures . in emergency department patients with traumatic brain injury , the onsd correlates with signs of elevated icp on computed tomography scans . more recently , researches have compared bedside ultrasound measurements of onsd to invasive icp [ 11 - 13 ] . while there is some variation in the optimal cut - off value , the correlation between onsd and icp remains consistent . in their current article , geeraets and colleagues provide further evidence of this physiological relationship and an intriguing possibility for non - invasive assessment of icp using mri . the obvious drawbacks to mri include its expense , long acquisition times , need for patient transport , and limited availability . however , some research has shown that mri may provide more precise measurements then ultrasound . geeraerts and colleagues used a conventional t2 sequence with relatively large slice thickness and interslice spacing , resulting in an overall feasibility of measuring the onsd in 95% of patients . greater accuracy and reliability would be expected in coronal t2 slices with thinner slices . as mri becomes more accessible and faster , non - invasive mri measurements may prove to be useful in certain clinical settings and as a potential reference standard for further research . continued research with larger studies is required to confirm the precision and accuracy of mri measurements of onsd , as well as the optimal measurement technique . additionally , the time course of onsd distention and reduction needs to be further delineated . currently , non - invasive assessments of icp do not obviate the need for invasive icp monitoring . invasive monitoring detects minute to minute variations in icp and , in the case of intraventricular drains , can also be therapeutic . however , non - invasive screening tests may be useful in select populations who would not otherwise require invasive monitoring and could undergo mri scans , such as patients with liver failure , meningitis , stroke , and moderate traumatic brain injury . in summary , the study by geeraerts and colleagues adds to a growing body of research demonstrating a correlation between increased onsd and elevated icp . by demonstrating the correlation of mri measurements of the onsd with invasive icp monitoring , they illustrate the potential of yet another non - invasive method to screen for elevated icp . while this technique will not replace invasive icp monitoring , it may be useful in select patient populations that would not otherwise have invasive monitoring but are at high risk for elevated icp . further research is required before we can use measurements of the onsd to predict exact values of icp , but it may be useful as a screening test to estimate the probability of elevated icp . csf : cerebrospinal fluid ; icp : intracranial pressure ; mri : magnetic resonance imaging ; onsd : optic nerve sheath diameter .

the current gold standard for the diagnosis of elevated intracranial pressure ( icp ) remains invasive monitoring . given that invasive monitoring is not always available or clinically feasible , there is growing interest in non - invasive methods of assessing icp using diagnostic modalities such as ultrasound or magnetic resonance imaging ( mri ) . increased icp is transmitted through the cerebrospinal fluid surrounding the optic nerve , causing distention of the optic nerve sheath diameter ( onsd ) . in this issue of critical care , geeraerts and colleagues describe a non - invasive method of diagnosing elevated icp using mri to measure the onsd . they report a positive correlation between measurements of the onsd on mri and invasive icp measurements . if the findings of this study can be replicated in larger populations , this technique may be a useful non - invasive screening test for elevated icp in select populations .

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children spend from 31 to 60% of their school day either writing or performing other fine motor tasks2 . the preparatory skills for writing are coordination of multiple joints , visual perception , vision - motor integration , and proprioception3 . together with stereognosis , proprioception allows smooth joint movement when vision is impaired or absent5 . children with poor proprioception have problems with handwriting legibility because their grip on a pen is too strong or too weak4 . generally , proprioception can be measured with joint position sense ( jps ) and kinesthetic sense ( ks)6 . while there are many studies investigating children s handwriting , quantitative studies on the association between handwriting and proprioception involving jps and ks are lacking7 . the aim of this study was to investigate the association between proprioception , involving jps and ks , and handwriting legibility in children . nineteen healthy children ( 15 boys and 4 girls ) with an average age of 9.7 0.36 years participated in this study . prior to the study , the children and their parents were informed about the purpose of the study and the general procedures to be undertaken . all children and the study was approved by the kaya university of human health science studies committee . the investigator moved the child s right arm passively through 80 of flexion at the elbow ( from 30 to 110 ) . the children were then directed to repeat this motion 10 times following a metronome set at 1 s intervals . their movements were recorded by a compact measuring system ( cms ) 10 for 3d motion analysis ( using the winarm software zebris medical gmbh , germany ) . cms markers were placed at the greater tubercle and lateral epicondyle of the right humerus and at the right wrist . angles of deviation from the targeted range of flexion were analyzed using matlab version 2014a ( the match works inc . , 2014 ) . ks was measured by the kinesthesia item in the sensory integration and praxis tests ( sipt , wps , torrance , ca , usa ) . the investigator moved a finger of the subject passively along a line from beginning to end , allowing the child to learn the line direction and distance . then , blindfolded and after one trial , the children were directed to trace five lines for each hand . only the results of the right hand were analyzed in this study . the investigator then measured the distance between the test endpoints and the real endpoints of the lines . legibility was evaluated using form , alignment , space , size , and slope8 . the legibility score was calculated as the ratio of the number of clearly written words to the total number of words ( score for legibility ( % ) = number of letters that received 5 points/30 100 ) . all data were analyzed using ibm spss statistics 20.0 ( ibm corp . , armonk , ny , usa ) . spearman s rank correlation was used to determine the relationships between legibility of handwriting and jps and ks , with significance defined as p<0.05 . values obtained for jps and ks in relation to handwriting legibility are shown in table 1table 1.handwriting legibility and proprioception test results ( n=19)mean sdjoint position sense 110 ( degree)22.530 10.063joint position sense 30 ( degree)14.256 10.408kinesthetic sense ( cm)2.044 0.703handwriting legibility ( % ) 33.521 15.852*standard deviation . there was no correlation between writing legibility and jps at either 30 or 110 of elbow flexion ( p>0.05 ) . a high ks correlated significantly with legible handwriting ( p<0.05 , table 2table 2.correlation between handwriting legibility and proprioception ( n=19)handwriting legibility ( % ) joint position sense 110 ( degree)0.016joint position sense 30 ( degree)0.009kinesthetic sense ( cm)0.370*p<0.05 ) . this study investigated the association between the legibility of handwriting and jps and ks in young children . children aged between 7 and 8 years are expected to be proficient in building up their speed of handwriting , ensuring consistency in size and proportions of letters as well as the spacing between letters and words9 . the average age of the participants in this study was 9.7 years old . our results showed that a highly accurate ks was associated with higher legibility scores . ks provides ongoing error information and memory storage to be recalled when writing is repeated . a high ks leads to programmed error correction , and the upgraded program generates better writing legibility . the results of this study support the hypothesis that ks reinforces the linkage between visual and motor control required for clear handwriting15 . we studies jps at the elbow joint because proprioception at the elbow joint is necessary for performing fine manipulative tasks including handwriting16 . studies have reported that proprioception at the wrist joint17 and finger joints18 influences handwriting legibility . we looked at children within a limited age range between 9 and 10 years of age . further , we did not control for other factors that affect writing legibility , such as fine motor control and visual - motor integration19,20,21 . finally , although handwriting quality is measured in terms of legibility and speed , this study investigated only writing legibility .

[ purpose ] this study investigated the association between proprioception , including joint position sense and kinetic sense , and handwriting legibility in healthy children . [ subjects and methods ] assessment of joint position sense , kinetic sense , and handwriting legibility was conducted for 19 healthy children . joint position sense was assessed by asking the children to flex their right elbow between 30 to 110 while blindfolded . the range of elbow movement was analyzed with compact measuring system 10 for 3d motion analysis . kinetic sense was assessed using the sensory integration and praxis test . the children were directed to write 30 words from the korean alphabet , and the legibility of their handwriting was scored for form , alignment , space , size , and shape . to analyze the data , descriptive statistics and spearman correlation analysis were conducted using ibm spss statistics 20.0 . [ results ] there was significant negative correlation between handwriting legibility and kinetic sense . a significant correlation between handwriting legibility and joint position sense was not found . [ conclusion ] this study showed that a higher kinetic sense was associated with better legibility of handwriting . further work is needed to determine the association of handwriting legibility and speed with joint position sense of the elbow , wrist , and fingers .

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the adenoid cystic carcinoma ( acc ) is a relatively rare epithelial tumor of the salivary glands . it accounts for about 5 - 10% of all salivary gland neoplasms , representing 2 - 4% of malignant occurrences of the head and neck area . approximately , 31% of lesions affect minor salivary glands , particularly the palate , although they can also be observed in the sub - mandibular and parotid glands . the frequency reported in the tongue is 19.8% , with 85% observed at the base of the tongue . we report one such rare case of tongue neoplasm which turned out to be acc in a middle aged lady . a 45-year - old - female patient presented with an asymptomatic growth of the tongue , which was perceived just 2 weeks before consultation . the intra - oral examination at that time revealed a mass in the dorsum of the tongue with light pain to pressure , without any evidence of cervical lymphadenopathy . the mass was firm , same color as that of the surrounding mucosa and asymptomatic otherwise [ figure 1 ] . as a pre - operative assessment of the lesion , a fine needle aspiration was done and the smear revealed a salivary neoplasm consisting of well delineated , tightly cohesive clusters of basaloid cells surrounding mucoid , hyaline globules , or clear spaces also forming honeycomb ( cribriform ) pattern [ figure 2 ] . at places dense aggregates of monomorphic small cells with uniform round to oval hyperchromatic nuclei and scanty cytoplasm were seen . smears also showed individual tumor cells with high n : c ratio and nuclear moulding . macroscopically , the mass had firm consistency with an irregular form and surface , brown color and measured 2.5 1.5 1.0 cm . the histopathologic study revealed multiple pseudocystic spaces of variable sizes surrounded by cuboidal cells with scarce cytoplasm and oval nuclei , filled with eosinophilic material and hence was consistent with the diagnosis of acc [ figure 3 ] . however , there was no evidence of perineural infiltration on serial sections . clinical photograph showing a swelling on the dorsum of the tongue cytological smears show well - delineated clusters of basaloid cells surrounding hyaline globules with uniform round to oval hyperchromatic nuclei and scanty cytoplasm ( papanicolaou stain , 400 ) histopathological section showing multiple pseudocystic cavities of variable size composed of cuboidal cells with scarce cytoplasm and oval nuclei ( h and e , 400 ) minor salivary gland neoplasms occur less commonly than the major salivary gland tumors and tongue is a relatively uncommon site for salivary gland neoplasms acc is a malignant neoplasm that originates in both the minor and major salivary glands , characterized by slow growth , diffuse invasion , and potential to produce distant metastases , mainly to the lungs and bones . it is an infrequent lesion , as it represents approximately 1 - 2% of all malignant neoplasms of the head and neck , and up to 10 - 15% of all malignant salivary gland neoplasms . the most common intra - oral site for minor salivary gland tumors is the hard palate , followed by the base of the tongue where up to 96% of all tumors are malignant , and acc represents 30% of them . on the other hand , one of the least frequent sites of presentation for acc is the mobile tongue , as several authors have reported an incidence of only approximately 3% of the cases . , analyzed 178 cases of salivary gland tumors , out of which only six cases were located on the tongue . cytologically , cribriform variety of acc can be diagnosed by hypercellular smears composed of clusters of small , relatively monomorphic epithelial cells with hyperchromatic nuclei . these appear bright magenta in may - grunwald giemsa mgg stains and pale blue with papanicolaou stain . finger - like process of similar material can also be found in between the groups of cells in tubular variety . the solid variant of acc also exhibits the same material and the cells resemble that of small cells of anaplastic carcinoma . the globules of amorphous material surrounded by the monomorphic hyperchromatic cells was a clue to the diagnosis of acc in our case , but since the hyaline globules are also found in other tumors like basal cell adenoma , pleomorphic adenoma , polymorphous low grade adenocarcinoma , epithelial myoepithelial carcinoma etc . it is important to distinguish the adenomas from acc because of the conservative mode of management in case of adenomas . we ruled out the adenomas because of the nature of the globules and the cytological morphology . unlike the adenomas , the hyaline globules were dense and stained intensely with mgg and the cells were relatively monomorphic , hyperchromatic with coarse chromatin and irregular nuclear membrane like that of acc . of three histologic variants - tubular , cribriform and solid ; in our case cribriform pattern was the dominant one without any evidence of perineural infiltration . the main factors associated with patient survival were tumor location , clinical stage , and the observed histologic variable . conversely , spiro et al . , have not found histologic classification to be of any benefit , and deny a correlation between microscopic appearance and prognosis . due to the slow growth pattern of the tumor however , due to local recurrence and late metastasis , surgery remains the mainstay of management with or without radiotherapy .

adenoid cystic carcinoma is a relatively rare epithelial tumor of the salivary glands accounting for about 5 - 10% of all salivary gland neoplasms . approximately , 31% of salivary gland neoplasms affect minor salivary glands particularly the palate . it involves tongue in only 19.8% of cases and even rarely the dorsum of the tongue . we report such a rare case that affected dorsum of the tongue in a 45-year - old - female patient .

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the use of synthetic material such as prolene mesh in prolapsus surgery has become a popular approach . it has been proven to be very effective and became a standard of care in the treatment of most cases with severe prolapsus . sacropexy is a surgical repair technique that restores pelvic anatomy by attaching synthetic graft material into the vagina and sacrum . erosion of vaginal wall or bowel can be seen as a long term complication . in this paper , we present a patient suffering from mesh migration into the rectum after abdominal sacral colpopexy . a 69-year - old woman was admitted to the hospital with a complaint of sensation of fullness and a feeling of a foreign material protruding during defecation . she had been diagnosed with uterine prolapsus and stress incontinence in 2008 and underwent total abdominal hysterectomy , bilateral salpingo - oophorectomy and sacral colpopexy with prolene mesh . the patient was admitted to our clinic five years after the procedure with complaints mentioned above . there was a foreign material palpated in rectum with digital examination . prolene mesh was detected in sacral region but resection of the mesh could not be conducted because of high levels of adhesions in that region . genital prolapse or genital hernia is described as the protrusion of pelvic organs along the vagina . it is one of the common gynecological conditions that affect the quality of life in women . it may be seen in up to 50% of multipara women , and its incidence increases with age . high rates of recurrence with traditional techniques led to the development of new surgical techniques . the use of synthetic mesh has become more popular surgical approach in cystocele and rectocele repair . mesh migration is a well - known clinical pathology and have been reported in literature . yolen and grossman suggested that intra - abdominal foreign bodies ( like mesh ) transmigrate into the small or large bowel by triggering an inflammatory reaction . persistent inflammatory reaction causes an opening into a hollow organ assisted by the peristaltic movement of the bowel . insufficient fixation of a mesh is another factor for migration of synthetic materials in some patients . larger pores greater than 75 nm permit the migration of macrophage and leukocyte migration and reduce the infection rate . large pores also improves flexibility of the mesh and cause tissue ingrowths and healthy collagen deposition . complications reported after sacropexy include ileus , intraoperative vessel injury , ureter injuries , recurrent descensus and mesh tearing . the use of a mesh as a graft material results in higher success rates but also causes a higher number of complications , such as mesh erosions or chronic infections . taoka reported a case of rectal migration of mesh in a 64-year - old woman who presented with a recto - cutaneous fistula 11 months after a tension - free vaginal ( tvm ) repair ; the patient was treated by removal of the infected mesh and closure of the rectal wall defect under cover of a temporary colostomy . by contrast with the troublesome symptoms reported in such patients in the literature , the only presenting complaint of our patient was protrusion of foreign material from the rectum . in conclusion , mesh migration is a serious complication after sacral colpopexy . sometimes surgical resection of migrated mesh with laparotomy can be difficult due to dense adhesions . scu was responsible for writing , conception and design of the study ; ob contributed toward analysis and interpretation of data ; nas , oa , aa- performed acquisition of data ; bk drafted the manuscript .

introductionpelvic organ prolapse ( pop ) is a common gynecological problem . repair with synthetic materials such as prolene mesh has become a popular approach in prolapsus surgery . migration of synthetic materials can cause serious complications.presentation of casea 69-year - old woman was admitted to the hospital with a complaint of sensation of fullness and a feeling of a foreign material protruding during defecation . the patient underwent exploratory laparotomy . prolene mesh was detected in sacral region but resection of the mesh could not be conducted because of dense adhesions causing frozen pelvis . the migrated prolene mesh was resected transanally.discussiongenital prolapse or genital hernia is described as the protrusion of pelvic organs along the vagina . it is one of the common gynecological conditions that affect the quality of life in women . mesh migration is a well - known clinical pathology.conclusionmesh migration is a serious complication after sacral colpopexy . surgical resection of migrated mesh can be difficult due to dense adhesions .

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hemangiomas are categorized as racemose , capillary , cavernous , and venous , according to the size of their vascular spaces . cavernous and venous hemangiomas have low blood flow , because they lack arterial or capillary components . histopathologically , venous hemangiomas contain dilated vessels with thick , fibrous walls , whereas cavernous hemangiomas have capillary - sized vessels lined by flat endothelial cells . we report the first case of a venous hemangioma of the pps and discuss its typical radiographic findings . a 49-year - old female patient was referred for evaluation of swelling in the right submandibular region . she had swelling for the first time 3 weeks earlier , and it had not improved after medical treatment . clinical examination revealed a soft mass in the right submandibular region and a bulge at the right lateral pharyngeal wall . a contrast - enhanced computed tomography ( ct ) scan revealed a cystic lesion , 4.63.0 cm , with contrast non - enhancement in the parapharyngeal space ( pps ) . mri showed a well - circumscribed non - enhancing mass with high signal intensity on t2-weighted images ( fig . the cystic lesion of the pps was surgically removed using a transcervical approach . during surgery the lesion bled profusely , but the mass was bluntly dissected from the surrounding structures . the surgical specimen , measuring 4.34.52 cm , showed a pale - to - dark brown soft cut surface with a blood - filled spongy vascular lesion . microscopic examination revealed blood - filled sinusoidal spaces with large irregular lumens and thick walls lined by endothelial cells ( fig . postoperative recovery was uneventful , and there has been no evidence of cranial nerve palsy or tumor recurrence after one year . pps has a complex anatomy and close proximity to vital anatomical structures , with which it may become involved by various pathological processes . presenting symptoms of pps tumors may be attributed to the size of the mass and compression of neighboring structures ( 1 ) . we had difficulty with the preoperative diagnosis , due to the absence of typical symptoms or signs of a vascular lesion in the pps , such as pulsation or bruit . fine - needle aspiration cytology ( fnac ) was also not helpful in reaching a diagnosis . although an exact tissue characterization of hemangioma in the pps could not be made on fnac , contrast - enhanced ct and magnetic resonance imaging ( mri ) should be included in differentiating these lesions from other tumors of the pps . in general , mr imaging is superior to ct imaging in its ability to ascertain the soft tissue characteristics of pps tumors ( 2 ) . changes in the blood flow dynamics within a hemangioma result in thrombus formation and phleboliths ( 3 ) . phleboliths are calcified nodules that can be regarded as a characteristic property of venous or cavernous hemangiomas . ct with contrast is an excellent imaging technique for revealing phleboliths . although mri is not sensitive for the detection of small amounts of calcification , large amounts of calcification show discrete low signal intensity on all pulse sequences mri can produce high signal intensities , representing the blood , as well as focal heterogeneities , representing areas of thrombosis , fibrosis , or calcification ( 4 ) . although mri is very useful for the detection of vascular lesions , the detectability of phleboliths on ct images is superior to that by mri . some reports have shown that plain x - ray films may also reveal calcified lesions . when imaging shows calcification in the pps , the differential diagnosis may include pleomorphic adenoma of the deep lobe of the parotid gland and metastatic thyroid carcinoma of the pps . pleomorphic adenoma with foci of chondroid or osteoid stroma can demonstrate opacities on imaging , but usually shows minute , scattered flecks . additionally , in cases of metastatic thyroid carcinoma of the pps , large flocculent calcification is seen on ct images ( 5 ) . venous hemangioma can not be clinically or radiologically differentiated from cavernous hemangioma , because of their similar characteristics . however , perfusion and blood pool scintigraphy has been demonstrated to have high sensitivity for detecting head and neck hemangioma , and can also differentiate between cavernous and venous hemangiomas ( 6 ) . they tend to be larger and less well circumscribed , and show no tendency to regress ( 7 ) . attention needs to be given to the tumor location , extent , growth rate , and accessibility as well as the patient 's age and esthetic concerns . the excision of large tumors in the pps can be challenging , given the risk for severe hemorrhage and nerve injury . a surgical approach should be chosen according to the tumor size and location , its relationship to the great vessels , and any suspicion of malignancy . generally , hemangiomas in the pps , including venous hemangioma , can be resected by using a transcervical approach . profuse bleeding often occurs during surgery , but after all of the pathology has been removed , the bleeding will cease . when surgery is impossible , the use of corticosteroids , cryotherapy , feeding vessel ligation , embolization , and fibrosing agents can be considered as alternatives ( 8) . in summary , we present the first reported case of venous hemangioma occurring in the parapharyngeal space . considering that a presumptive diagnosis of most pps tumors can be made based on imaging studies , preoperative imaging findings are very important in approaching these lesions . it may be that multiple , spotty , calcific nodules , 13 mm in size , within a cystic lesion on ct images indicate the pathognomonic finding of hemangioma in the pps .

a hemangioma of the parapharyngeal space ( pps ) is an extremely rare tumor and is responsible for 0.5 - 1% of all tumors occurring in the pps . we report a case of pps venous hemangioma in a 49-year - old woman presenting with diffuse swelling in the submandibular region . a preoperative computed tomography ( ct ) scan showed a cystic mass with multiple calcifications in the pps . the calcific nodules were round and about 2 mm in diameter . the hemangioma was completely resected via a transcervical approach . during surgery , we found several calcific nodules , which represented phleoboliths or areas of thrombosis with dystrophic calcification . despite its rarity , a venous hemangioma of the pps should be considered in a differential diagnosis when a cystic mass with calcification is found by ct scan . to our knowledge , this is the first reported case of a pps venous hemangioma ; we describe its pathognomonic findings on imaging .

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verruciform xanthoma ( vx ) , which has almost similar histologic features , is also a rare lesion usually found on the oral mucosa or the genital area . it is presumably associated with the inflammatory response to mucosal damage.1 however , xanthoma and vx of the esophagus are extremely rare . since the first report by remmele and engelsing2 only 13 cases of esophageal xanthoma have been reported,1,2,3,4,5,6,7,8,9 and since the report by herrera - goepfert et al.,10 only four cases of vx of the esophagus have been reported.10,11,12,13 the etiologies of both lesions are not understood . we describe herein a new case , including a review of all reported cases of xanthoma and vx of the esophagus . a 70-year - old man with an unremarkable medical history was hospitalized with a complaint of epigastric pain . serum total cholesterol , triglyceride , high density lipoprotein cholesterol , and low density lipoprotein cholesterol levels were 151 , 215 , 33 , and 102 mg / dl , respectively . multiple shallow gastric ulcers and a duodenal ulcer were detected and suspected to be the cause of the pain . besides the ulcers , in the upper esophagus 20 cm from the incisors , a 3-mm yellowish granular elevated mucosal lesion was found and a biopsy was performed ( fig . 1 ) . microscopically , large round cells were aggregated in the lamina propria immediately beneath the squamous epithelium . the etiologies are different , as xanthoma is caused by hyperlipidemia and vx arises presumably as a result of an inflammatory response to continuous mucosal damage.1 however , the etiologies of the two lesions arising in the esophagus are not understood . the characteristics of all reported cases of xanthoma and vx of the esophagus are summarized in table 1 . fourteen cases of xanthoma and four cases of vx of the esophagus have been reported . however , some reports loosely stratified vx into esophageal xanthoma , whereas others have excluded it.6,8 in terms of clinical data , both diseases were found predominantly in men than in women : 9 versus 3 in xanthoma and 3 versus 1 in vx . the median age was 59 years ( range , 37 to 74 ) in xanthoma and . the predominant location was the lower esophagus for xanthoma ( lower , 7 ; middle , 2 ; upper , 3 ) , whereas vx was not reported in the lower esophagus ( upper , 2 ; middle , 2 ) . the median size was not different : 3 mm ( range , 2 to 10 ) for xanthoma and 4 mm ( range , 3 to 20 ) for vx . the associated medical conditions were diverse ; however , two patients with malignant tumors were included in each group : hepatocellular carcinoma and ileocecal lymphoma in xanthoma , and gastric cancer and multifocal cancer ( cancer of the glottis , liver , and trachea ) in vx , although there was no definite association . vx is characterized by its histologic features , including papillomatosis , acanthosis , and hyperparakeratosis.11 also , the external morphology is verrucoid . nevertheless , findings of large round foam cells in the lamina propria under the squamous epithelium are the same as those in xanthoma . it is difficult to differentiate between the two lesions on the basis of gross examination when they arise on the esophagus . exophytic and verrucoid features seen in vx of the skin were not observed in the esophagus because most of the reported cases were small in size.10,12 considering that xanthoma and vx are nonneoplastic lesions , differentiating between them could be a waste of effort . however , these lesions have to be grossly distinguished from ectopic sebaceous glands and small subepithelial tumors such as carcinoid and granular cell tumor because most of the reported esophageal xanthomas are yellowish or white mucosal elevated lesions . in terms of microscopic findings , signet ring cell carcinoma , which contains round cells with abundant cytoplasm , while signet ring cell carcinoma has an eccentrically located nucleus because of the intracellularly abundant mucin , xanthoma has a centrally located and small nucleus . positive immunohistochemical staining for cd68 , which indicates a histiocytic origin , is another characteristic finding of xanthoma.5 moreover , esophageal cancer and ectopic sebaceous glands do not commonly stain with lugol 's solution ; thus , endoscopists need to be aware of these lesions for the differential diagnosis.6,14 with more case reports of esophageal xanthoma and vx of the esophagus , the characteristics of both lesions will be more clearly elucidated .

xanthoma is an uncommon nonneoplastic lesion resulting from the accumulation of histiocytes . it predominantly shows cutaneous manifestations associated with dyslipidemia . however , xanthoma of the esophagus is extremely rare . to the best of our knowledge , only 14 cases have been reported thus far . the clinical significance of this lesion has not been established . however , this lesion should be distinguished grossly from ectopic sebaceous glands and small subepithelial tumors such as carcinoid and granular cell tumor . moreover , signet ring cell carcinoma , which contains round cells with abundant cytoplasm and has similar histologic features to xanthoma , should be distinguished microscopically .

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soft tissue coverage for wounds remains a difficult management problem for patients sustaining traumatic injury and burns . there are several methods to achieve wound coverage secondary healing , primary suturing , skin grafting , and flap surgeries as described in reconstruction ladder . a skin graft is the most commonly used modality for coverage of wounds in reconstructive plastic surgery . the skin graft needs to undergo various stages of healing for a good take on the recipient bed . there are different methods employed to secure the graft to recipient bed for a few days with a basic idea to ensure that the graft is not elevated off the bed by formation of haematoma / seroma under it . repeated tie over dressings are required in situations where the dressing needs to be changed more frequently as in cases of infected raw area , bleeding tendency , patients on anticoagulant drugs , and in convex areas of body such as buttocks , breast , and the scalp , where the dressing is difficult to secure . in this novel method , a sterile sample container was cut at its upper part [ figures 1 and 2 ] . the skin graft was applied on the raw area and fixed with skin staplers and tie over sutures . once paraffin gauze and adequate padding is applied on the raw area , the tie over threads were passed from inside out of the container [ figures 3 and 4 ] and pulled at the appropriate tension to keep the dressing in place . the lid of the container was tightened to complete the dressing ensuring that the graft was maintained in close approximation with the wound surface . sterile plastic container upper part of the sterile container is cut tie over threads being passed from inside out the lid is tightened over the dressing the dressing can be changed repeatedly with sterile precautions depending on the requirement , as an outpatient procedure by unscrewing the lid [ figures 5 - 8 ] which can then be easily reapplied . the procedure can be used on wounds of any size by changing the size of the sterile container chosen . post - toilet mastectomy raw area covered with the skin graft the tie over dressing applied in ot tie over dressing repeated in an outpatient department we have used this method in eight cases of the post toilet mastectomy raw areas with very good results [ figures 9 and 10 ] . post - toilet mastectomy raw area skin graft once applied has to be covered with petrolatum gauze to avoid its separation from the wound bed at the time of change of dressing . an ideal method of graft fixation should be simple , rapid , repeatable , able to be performed in the outpatient department , prevent hematoma or seroma formation , soak the exudates well , and allow the graft bed to be inspected easily . there are multiple methods of securing dressings over the skin graft , some of them can be applied only once and some can be repeated . the dressings that can be applied only once are like foam , hydro cellular dressing ( highly absorbent and can be easily changed ) , negative pressure therapy dressing ( stabilises the graft , increases the vascularity of bed , takes away toxic chemicals ) , and gas bag ( transparent , can see graft and monitor any haematoma ) . these traditional methods can stabilize the graft till the first dressing post operatively . in some contaminated wounds , the dressing needs to be removed earlier , especially if there is drainage or foul smell . this approach may also be proper for graft , used to cover defects of some anatomical regions with increases risk of contamination , such as perineal , axillary , and genital or it can be used in areas where base of wound is difficult to immobilize like breast / pectoral region . repeated tie over dressings can be done by keeping interrupted sutures long to be used as tie over dressings . these ties over dressing can be made of sutures or rubber bands . when taking a tie over stitch , both the threads can be left long and only one thread is tied at a time , the other thread is left long for next time . these techniques are difficult for small dressings especially the bra hooks ; the silk loops method is very cumbersome and takes long time to do . the novel method being discussed has a very small learning curve and is very fast . it hardly takes 5 min in the hands of a plastic surgeon to complete the dressing . this dressing technique maintains the advantage of conventional tie over dressing with rapidity and repeativity . good graft take can be expected whether split - thickness or full - thickness with appropriate methods of stabilisation . we recommend a novel , low cost , simple , rapid method of graft fixation that can be used repeatedly and can be applied to a wound of any size .

tie overdressing is commonly used to secure the graft against the raw surface and prevent loss due to of hematoma or seroma . a conventional tie over dressing with silk sutures , is a useful method of securing the graft to raw area . refixation is difficult when repeated tie over dressings are needed . we assessed a low cost repeated tie over dressing method using sterile sample collection containers and silk suture threads in eight patients . after the graft is applied on the bed , tie - over stitches are taken , and paraffin gauze is applied over with adequate padding ; the tie over sutures are passed through the container and the lid is tightened over it to complete the dressing . the lid can be unscrewed easily at any time to inspect the graft and can be easily reapplied in the outpatient department . the skin graft take in all the patients was complete without any seroma or hematoma . a novel and low - cost tie over dressing that enables simple fixation of the dressing , to maintain proper position of grafts that require repeated fixation is reported here .

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since 2006 , surveillance physicians have listed and collected blood from those patients encephalitis , defined as fever or history of fever with axillary temperature > 38.5c ( 101.3f ) and altered mental status , new onset of seizures , or new neurologic deficit in patients admitted to 3 nipah surveillance hospitals : rajshahi , rangpur , and faridpur medical college hospitals . the institute for epidemiology disease control and research and us centers for disease control and prevention tested serum with an igm - capture enzyme immunoassay to detect niv igm , and we defined laboratory - confirmed niv encephalitis as niv igm in serum . during december 2012march 2013 , surveillance physicians interviewed accompanying caregivers of all hospitalized patients whose illness met the encephalitis case definition on admission in the inpatient ward . study physicians asked about patients consumption of raw or fermented date palm sap and contact with other persons with fever and altered mental status in the month before illness onset ; if caregivers were unaware of the patient s exposures , study physicians asked them phone the patient s friends and colleagues about exposures . hospital physicians used personal protection equipment and provided it to caregivers of each patient with encephalitis and a history of these exposures . as part of subsequent epidemiologic studies , we also conducted detailed case investigations at each niv encephalitis case - patient s household . we interviewed surviving patients directly , or appropriate proxies among family , friends and relatives for patients who died , about their exposures to encephalitis patients or to fresh or fermented date palm sap before illness . we calculated the sensitivity , specificity , positive predictive value ( ppv ) , and negative predictive value ( npv ) of the screening questions asked on admissions to hospitals by comparing with the niv igm results . we repeated the calculations for patients hospitalized during january and february , when the prevalence of niv encephalitis is highest . we compared the answers provided by caregivers during patient hospitalization with those provided during interviews in the community as part of our epidemiologic studies . icddr , b s ethical review committee reviewed and approved the protocol for niv surveillance and case investigation . they collected and tested blood samples from 328 ( 91% ) patients for niv igm . seventeen ( 5% ) had niv igm ( table 1 ) , of whom 15 ( 88% ) niv encephalitis case - patients were identified during january and february 2013 . of the 17 confirmed case - patients , family caregivers of 14 reported either a history of drinking raw or fermented date palm sap or contact with other persons with fever and altered mental status in the month before illness onset . therefore , the sensitivity of the screening questions was 82% , specificity was 86% , ppv was 24% and npv was 99% ( table 2 ) . the sensitivity during january february was 93% , specificity was 82% , ppv was 37% , and npv was 99% . * drinking raw or fermented date palm sap or having contact with encephalitis patients in month before illness onset . at admission , 3 ( 18% ) niv encephalitis case - patients had no reported history of drinking raw or fermented date palm sap or of contact with persons who had encephalitis ( table 1 ) . however , during the epidemiologic investigations in the community , family members of 2 case - patients reported that the patients drank fermented date palm sap in the month before illness onset . of the 14 niv encephalitis case - patients who , at admission , had reported 1 of the risk exposures , results were consistent with exposures reported during the epidemiologic investigation . screening patients with possible encephalitis at the time they seek hospital care regarding recent exposure to date palm sap and to other patients with encephalitis demonstrated high sensitivity and specificity for detecting niv encephalitis , particularly during peak months of niv encephalitis incidence . the high npv of the screening questions suggests that focusing infection control efforts toward patients with these exposures is an efficient use of scarce resources to prevent transmission . although three fourths of encephalitis patients had reported histories of exposure , they possibly could have had other infections , including other bat - borne viruses , that were transmitted through similar routes or could have lacked niv igm , despite having niv infection ( 13 ) . alternatively , recent consumption of date palm sap by these patients might have been purely coincidental because this practice is common in bangladesh during this season , but nipah infection is rare . for 2 niv encephalitis case - patients , caregivers did not report a history of drinking fermented date palm sap during hospital interview , but this behavior was reported in later community investigations . because 90% of bangladeshis are muslim , and consumption of alcohol is prohibited by islam ( 14 ) and illegal in bangladesh , patients might be reluctant to report drinking traditional liquor made of fermented date palm sap . therefore , caregivers should be asked about socially stigmatized behaviors privately and confidentially to increase the odds that these stigmatized behaviors are reported . exposure - based screening can detect patients at high risk for niv encephalitis in low - income , resource - constrained settings , such as bangladesh . we deployed screening questions on admission to inpatient wards but screening earlier , at triage in emergency wards , could further reduce risk . surveillance for other diseases with well - described exposures that put healthcare workers at risk , such as ebola virus infection , and where laboratory diagnosis is limited or delayed could also deploy this approach .

we measured the performance of exposure screening questions to identify nipah virus encephalitis in hospitalized encephalitis patients during the 201213 nipah virus season in bangladesh . the sensitivity ( 93% ) , specificity ( 82% ) , positive predictive value ( 37% ) , and negative predictive value ( 99% ) results suggested that screening questions could more quickly identify persons with nipah virus encephalitis .

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parasitic infections are increasing worldwide due to rapid urbanization of cities , global warming , and international traveling . the diagnosis of parasitic diseases of the respiratory system is challenging because clinical manifestation and radiological findings are not specific . the larvae can cause airway inflammation , whereas migration of the mature adult worm may cause mechanical obstruction . we present a case of interstitial pneumonitis caused by parasitic infestation , which was diagnosed on transbronchial lung biopsy ( tblb ) . a 54-year - old female presented with a history of exertional breathlessness and dry cough for two months . she was afebrile with spo2 of 91% on room air and bilateral crepitations on respiratory examination . l ( 46.4% ) , fvc of 1.45 l ( 45.2% ) , and fev1/fvc 83% with reduction in dlco 1.46 mmol / min / kpa ( 18% ) and corrected value with alveolar volume 0.49 mmol / min / kpa ( 30% ) . high - resolution computed tomography ( hrct ) of chest showed multiple ill - defined nodular opacities in both lung fields showing centrilobular distribution associated with ground glass opacities bilaterally . fine reticular densities were seen at places with segments of traction bronchiectasis [ figure 1 ] . total serum ige was 145 iu / ml and specific ige for pigeon droppings ( < 0.10 tblb showed thickened alveolar septa with mild lymphoplasmacytic inflammatory infiltrate in the interstitium with cross - sections of calcified parasitic larvae with foreign body giant cell reaction around it [ figure 2 ] . travel history revealed frequent travels in the himalayan belts yearly for 20 years during which she had frequent episodes of diarrhea . she was given albendazole 400 mg for three days and oral corticosteroids for 12 weeks . bronchoalveolar lavage ( bal ) for tuberculosis culture at 6 weeks was negative . at 6 weeks high - resolution computed tomography chest showing multiple ill - defined centrilobular opacities with ground glass opacities bilaterally before treatment histopathology image of transbronchial lung biopsy showing calcified larva with thickened alveolar septa with mild lymphoplasmacytic inflammatory infiltrate in the interstitium high - resolution computed tomography chest showing remarkable improvement after treatment diffuse lung diseases can be further divided into transient pulmonary infiltrates and alveolar or interstitial lung diseases . ascariasis , anchylostomiasis , and toxocariasis usually cause transient pulmonary infiltrates , whereas schistosomiasis , strongyloidiasis , and tropical pulmonary eosinophilia can cause diffuse interstitial changes , as in the current case . strongyloidiasis causes reticulonodular opacities because of secondary infection , hemorrhage , inflammatory pneumonitis , and bacterial abscess formation . schistosomiasis eggs that are not passed into bladder or intestinal lumen are the main cause of chronic lung diseases causing granulomatous reaction and fibrosis . tropical pulmonary eosinophilia typically results from a hypersensitivity reaction to wuchereria bancrofti and brugia malayi . tropical pulmonary eosinophilia is an immunological response to microfilariae rather than acute infection , which usually present as reticulonodular opacities . the diagnosis of parasitic infections is difficult on chest radiography or hrct of chest because of nonspecific presentations . bronchoscopy may be helpful for diagnosis of parasitic infestation by variety of ways like direct visualization , bal , brushing , and tblb . parasitic lung diseases can also show microscopic pulmonary calcification on lung biopsy . in the current report , transbronchial lung biopsy revealed showed thickened alveolar septa with mild lymphoplasmacytic inflammatory infiltrate in the interstitium with cross - sections of calcified parasitic larvae . tblb is performed for obtaining tissue specimen from peripheral lung masses and focal or diffuse lung infiltrates . the technique is useful in patients with suspected lung cancer , fungal and mycobacterial lung infections , unexplained infiltrates in immunocompromised hosts and in patients with suspected pulmonary sarcoidosis , lymphangitic carcinomatosis , and in selected cases of pulmonary langerhan 's cell histiocytosis , lymphangioleiomyomatosis , and cryptogenic organizing pneumonia . with increasing travels and worldwide migration , parasitic infections should be considered in differential diagnosis of interstitial lung diseases particularly in the evaluation of diffuse lung infiltrates . bronchoscopy and transbronchial lung biopsy can be useful in the diagnosis of diffuse lung infiltrates .

parasite infections are increasing worldwide due to increasing migration and traveling . parasitic infections can affect lungs and present as a focal or diffuse lung diseases . high index of suspicion and detailed history are most important . we present a case of interstitial pneumonitis caused by parasite infestation , which was diagnosed on transbronchial lung biopsy .

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recently , new oral anticoagulants have been approved as alternatives to warfarin for patients with atrial fibrillation . rivaroxaban is one of the novel anticoagulants , which is an oxazolidinone derivative and inhibits both free factor xa and factor xa bound with the prothrombinase complex . it is a highly selective direct factor xa inhibitor with oral bioavailability and rapid onset of action . there are some advantages of the new agents compared with warfarin including rapid anticoagulation after an oral dose and lack of dietary or drug - drug interaction . however , there are no specific antidotes for the anticoagulant effect of rivaroxaban in the event of a major bleeding , unlike warfarin . we present a case of spontaneous rectus sheath hematoma ( rsh ) during rivaroxaban therapy for atrial fibrillation in an elderly female patient . a 75-year - old woman presented to the emergency department with the complaints of fatigue and abdominal pain after coughing . the patient had been started on new oral anticoagulant agent rivaroxaban therapy for nonvalvular atrial fibrillation for 3 days . the dose of 20 mg / day rivaroxaban was started because the creatinin clearance of the patient was above 50 ml / min . she had a blood pressure of 70/40 mmhg and an irregular heart rate of 115 beats / min on admission . the patient had no history of any trauma or surgery ; she reported that the symptoms started after vigorous coughing . blood analyses revealed leukocytosis ( 26.5 k / ul ) accompanied by severe anemia ( 5.4 g / dl ) . platelet counts were within normal ranges and her international normalized ratio ( inr ) was 1.48 . her abdominal x - ray was normal and the stool occult blood test was negative . after the first treatment , the patient was transferred to the intensive care unit ( icu ) . repeated abdominal examination in the icu revealed increased tenderness and a palpable mass on the left side of the umbilicus . noncontrast abdominal computerized tomography scan showed a left - sided rsh , 102 45 mm in size [ figure 1 ] . a specific antidote for rivaroxaban is not available then the patient was treated with fluid resuscitation and packed red blood cells . computed tomography scan of the abdomen shows a left - sided rectus sheath hematoma ( arrow ) rivaroxaban is an oral anticoagulant agent that directly inhibits factor xa and interrupts both the intrinsic and extrinsic pathway of the coagulation cascade . rivaroxaban is currently indicated for use in patients for atrial fibrillation and prophylaxis of deep venous thrombosis . it does not require inr monitoring like warfarin . with the increasing use of the new anticoagulant agents like rivaroxaban in atrial fibrillation , bleeding complications due to these agents there are no specific antidotes for the anticoagulant effect of rivaroxaban and other new oral anticogulants unlike warfarin , thus the management of the bleeding complications include support and observation . currently , no available specific antidote exists for the management of rivaroxaban - associated bleeding events , but supporting therapy is useful which are likely to be effective for the majority of patients because of the short half - lives of these agents . recent studies showed that rivaroxaban was noninferior to warfarin for the primary endpoint of stroke and systemic embolism . there was no reduction in rates of mortality or ischemic stroke , but a significant reduction in hemorrhagic stroke and intracranial hemorrhage . the primary safety endpoint was the composite of major and clinically relevant nonmajor bleeding , which was not significantly different between rivaroxaban and warfarin but , with rivaroxaban , there was a significant reduction in fatal bleeding , as well as an increase in gastrointestinal bleeds and bleeds requiring transfusion . the main causes of the rsh include anticoagulant therapy , hematological disorders , trauma , excessive physical exercise , coughing , sneezing , and pregnancy . especially in elderly patients the risk of rsh may be increased due to the impaired functional status and weakened rectus muscle . early recognition , rapid assessment and treatment are important to reduce the complications such as hemodynamic instability , abdominal compartment syndrome , multiorgan dysfunction and even death . the treatment of such a hematoma includes transfusion with packed red blood cells and supporting therapy based on regularly monitoring of hemoglobin levels . rivaroxaban has a mean terminal half - life of 7 - 11 h so in bleeding events supporting therapies are likely to be effective for the majority of patients . several studies have shown that prothrombin complex concentrate may be useful in reversing the effects of rivaroxaban . other possible measures include the use of recombinant factor viia to reduce bleeding or the use of activated charcoal to reduce absorption in cases of overdose . several factors are reported which increase the risk of patients developing hemorrhage while receiving rivaroxaban , these include advanced age , hypertension , history of hepatic / renal disease , previous stroke , coagulopathy , concomitant use of antiplatelet agents and alcohol consumption . jaeger et al . have reported a 61-year - old female patient who developed a spontaneous spinal epidural hematoma after being treated by rivaroxaban . boland et al . also reported acute onset severe gastrointestinal tract hemorrhage in a postoperative patient taking rivaroxaban after total hip arthroplasty . in our patient , there was no other medication except rivaroxaban that could cause the hematoma . based on naranjo 's scale , a score of 7 showed that the rivaroxaban was the probable cause of the rsh . several case reports of muscle hematoma due to the antiplatelet and anticoagulant agents have been reported previously , but this is the first reported case of spontaneous rsh due to the rivaroxaban .

rivaroxaban is an oral anticoagulant agent that directly inhibits factor xa and interrupts both the intrinsic and extrinsic pathway of the coagulation cascade and is currently indicated for use in patients for atrial fibrillation and prophylaxis of deep venous thrombosis . the present case reports of spontaneous rectus sheath hematoma during rivaroxaban therapy for atrial fibrillation in a 75-year - old woman .

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a 60-year - old male patient , with a known history of arterial hypertension and type-2 diabetes mellitus , was referred to the outpatient echocardiography laboratory to undergo stress echocardiography because of recent episodes of chest pain occurring on mild exertion . he was on drug therapy with angiotensin - receptor blocker , thiazide diuretic , nondihydropyridine calcium antagonist , and dronedarone , which had been started because of recent episodes of paroxysmal atrial fibrillation and discontinued 2 days before the stress test . the patient received a standard protocol of high dipyridamole infusion in two doses ( 0.56 mg / kg and 0.28 mg / kg ) followed by atropine administration ( 1 mg in four 0.25 mg doses ) . at rest , no ischemic abnormalities were observed on electrocardiogram ( ecg ) and transthoracic echocardiography ( tte ) [ figure 1 and videos 13 ] . after completion of dipyridamole infusion , the patient complained a mild chest discomfort , without any significant ecg changes and any apparent wall - motion abnormalities on tte [ videos 46 ] . after atropine injection , a worsening of the anginal symptoms combined with a descending st - depression in v3 occurred ; despite the absence of relevant echocardiographic changes , two - dimensional ( 2d ) strain analysis showed lower longitudinal strain of the anterior interventricular septum from rest to peak dose [ figure 2 ] . afterward , as recommended , aminophylline was administered ; interestingly , a more pronounced st - depression and deep inverted t - waves in v2v4 appeared [ figure 3 ] . the patient was admitted to the cardiology department and underwent coronary angiography from the radial access , which revealed a long myocardial bridge ( mb ) of the left anterior descending ( lad ) artery with systolic milking [ figures 4 , 5 and videos 79 ] . the patient was , thereafter , treated with a beta - blocker and discharged without symptoms and ischemic abnormalities on rest ecg . electrocardiogram at rest two - dimensional strain analysis showing global longitudinal strain at rest ( upper panel ) and at peak dose of dipyridamole ( lower panel ) electrocardiogram after injection of atropine and aminophylline coronary angiography images in diastole ( left ) and systole ( right ) showing myocardial bridge of the left anterior descending artery with systolic milking angiographic images did not change after intracoronary nitroglycerine administration this case depicts a clinical scenario of positive dipyridamole stress test in a patient affected by mb of the lad . although mb has been classically deemed a benign coronary artery abnormality , it has been recently related to acute myocardial infarction and sudden cardiac death . moreover , mb has been also associated with endothelial dysfunction , early atherosclerosis , and coronary vasospasm . in particular , a worsening of systolic coronary narrowing of mb has been found when using vasodilator agents , such as nitroglycerine , which are usually not administered in these patients . in our case , dipyridamole provoked chest pain associated with minor ecg ischemic changes , such as an only one - lead ( v3 ) st - depression ; these abnormalities worsened after the administration of atropine and later , aminophylline , likely because of drug - induced positive inotropic and chronotropic effects . atropine might also have determined myocardial ischemia through a paradoxical coronary vasoconstriction induced by acetylcholine as observed in the presence of endothelial dysfunction and mb . this case has the following interesting implications : ( 1 ) the ability to detect mb also using a vasodilator stress test ( and not only dobutamine echocardiography or exercise test ) , particularly with the addition of atropine injection , ( 2 ) the utility of 2d strain analysis in confirming subtle regional wall - motion abnormalities , and ( 3 ) peculiar diagnostic features , on stress echocardiography , suggesting mb rather than obstructive coronary artery disease . in particular , the observed ecg and echocardiographic ischemic changes , although suggestive of a lad disease , appeared to be late occurring and less extended than usually observed in patients with stable hemodynamic lad obstruction , which should have determined more pronounced ecg and wall - motion abnormalities . indeed mb , differently from a fixed coronary obstruction , is a dynamic stenotic lesion , requiring a consistent increase in heart rate and myocardial contractility to provoke myocardial ischemia . hence , mb could have been clearly unmasked only whenever positive inotropic and chronotropic agents had been added to the vasodilator stress caused by dipyridamole . thus , the finding of worsening ischemic abnormalities after the administration of atropine ad aminophylline , during dipyridamole stress echocardiography , may represent a particular diagnostic feature of mb .

a 60-year - old male patient was submitted to dipyridamole - atropine stress echocardiography ( dse ) for chest pain during exertion . at rest , no electrocardiographic ( ecg ) and transthoracic echocardiographic ( tte ) abnormalities were observed . after dipyridamole infusion , the patient complained a mild chest discomfort , without ecg changes and tte wall - motion abnormalities . subsequently , worsening of the anginal symptoms combined with descending st - depression and t - negative waves occurred after atropine and unexpectedly , aminophylline administration . coronary angiography was performed showing a myocardial bridge ( mb ) of the left anterior descending artery . the occurrence , during dse , of worsening ischemic abnormalities after atropine and aminophylline administration may be a particular diagnostic feature of mb .

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institutional review board approval was obtained from the wake forest university , and informed consent forms were completed for all participants . items for four concepts identified in the literature were glucose control , well - being and side - effects , lifestyle burden , and treatment complexity and convenience and were evaluated in a series of five focus groups made up of five to eight patients drawn from an evaluation study of community diabetes clinics in north carolina ( 11 ) . participants were male and female and white and nonwhite with simple and complex medication regimens and a1c levels that ranged from well controlled to uncontrolled . the resulting 35-item prototype instrument was administered by mail to a convenience sample of 75 patients ( the exploratory sample ) , who were treated with diabetes medications at our study community - care site , to assess item reliability , mean and distribution , redundancy or uniqueness , skewness , and construct validity . also examined were item correlations with a1c level , the multidimensional diabetes questionnaire ( 12 ) lifestyle interference scale , the medical outcomes studies ( mos ) health worries scale score ( 13 ) , and global items assessing extent that blood glucose has been unacceptably high or low . an item performance score was constructed ( 0 , weak ; 1 , moderate ; or 2 , ideal performance ) to guide item retention . fifty - five ( 73% ) patients completed the survey , and nine items were removed based on skewness or redundancy ( r > 0.75 ) with other items . in the initial test sample , patients of a large family - medicine practice treated for diabetes with a recent a1c value within the last 3 months ( the evaluation sample ) were invited to complete the study survey packet including the revised 26-item instrument and validation instrument described above . medication complexity was assessed using a score of 0 or 1 ( no / yes ) for common diabetes medications and a score of 0 or 2 ( no / yes ) for insulin , a more demanding regimen . self - reported adherence to medications was by recall of skipped or missed doses over the last 10 days . packets were mailed to patients with instructions and a voucher for a 25 usd gift certificate . exploratory factor analysis ( efa ) of the dmsat items was conducted using sas ( version 8 ; sas , cary , nc ) to assess whether the common factor model was appropriate ( 14 ) based on kaiser 's sampling adequacy , scree plot , and model fit . discriminant validity of the dmsat was examined by comparing means across levels of a1c ( < 8% and 8% ) , treatment complexity ( low and high ) , self - reported adherence , and mos health worries . for the final test sample , another sample of patients from our community diabetes care clinics ( 11 ) and from an academic medical center was recruited to conduct and evaluate confirmatory factor analysis of the dmsat and confirm validity in the evaluation sample , 194 ( 63% ) of 307 eligible patients returned the survey packet ; of these , 140 reported current medication use . participants had a mean age of 63 years , and most had completed high school ( 77% ) and had been diagnosed with diabetes at least 5 years previously ( 61% ) . one - third ( 2939% ) were taking one , two , or three medications for diabetes , with 16% taking insulin ; 14% had a recent a1c > 8.0% , and 19% rated their adherence to their medication regimen in the last 10 days as less than complete . ten items displayed high inter - item correlations ( > 0.75 ) and were removed . initial factor analysis of the reduced 16-item questionnaire identified a four - factor structure consistent with our domains of lifestyle , medical control , convenience , and well - being and explained 75% of the total variance . as shown in table 1 , dmsat scales and total score discriminated ( p < 0.05 ) between high and low levels of treatment complexity , self - rated glucose control , mos health worries scale score , and clinical value for recent a1c ( < 8% vs. 8% ) in the expected direction . correlation of the dmsat scores with continuous a1c values was 0.24 ( p = 0.0049 ) . in the final , confirmatory sample , as shown in table 1 , dmsat scales and total scores discriminated between validity groups as in the previous sample and were highly correlated with the dtsq ( r = 0.68 ; p < 0.001 ) . unlike the dmsat , the dtsq total score did not discriminate between levels of treatment complexity and clinical a1c value . the dmsat is intended as a brief measure of diabetes medication treatment satisfaction and discriminates between important correlates of patient management . it performed as well as the dtsq in detecting self - rated glucose control and health worries but showed superior properties in correspondence with treatment complexity and a1c . note that appraisals of cost of medications or specific side effects that may be caused by diabetes or its treatment , such as diminished sexual functioning , bloating , or weight gain , are not separately assessed and may require assessment elsewhere . , we believe that the 16-item dmsat offers a comprehensive assessment of satisfaction with diabetes therapy and may aid in individualizing patient diabetes treatment .

objective to develop and test a patient questionnaire on treatment satisfaction with diabetes regimens.research design and methods survey items were developed from community clinic focus groups , pretested in patients with diabetes , and examined in two samples of treated patients.resultssixteen items performed well in assessing treatment experiences : ease and convenience , lifestyle burdens , well - being , and medical control . construct validity was supported by associations ( p < 0.05 ) with treatment complexity , self - rated glucose control , health worries , and a1c . internal consistency ranged from 0.89 to 0.95.conclusionsthe diabetes medication satisfaction tool offers a comprehensive assessment of patient acceptability , with diabetes therapy useful for individualizing therapeutic decision making .

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the incidence of infective endocarditis ( ie ) remained relatively stable from 1950 through 2000 at about 3.6 to 7.0 cases per 100,000 patient - years.1 in selected areas , the incidence may rise because of the concentration of populations at uniquely high risk of infection , specifically intravenous ( iv ) drug abusers . the risk of ie among iv drug abusers , 2 to 5% per patient - years , is several - fold greater than that for patients with rheumatic heart disease or prosthetic valves . ie is located on the tricuspid valve in 46 to 58% of patients with iv drug abuse . staphylococcus aureus causes more than 50% of ie occurring in iv drug abusers overall and 60 to 70% of infection involving the tricuspid valve . the clinical manifestation of ie in iv drug abusers depends on the valve involved and , to a lesser degree , on the infecting organism . the sensitivity of transthoracic echocardiography ( tte ) for the detection of vegetations in patients with native valve endocarditis ( nve ) is approximately 65% , whereas that of transesophageal echocardiography ( tee ) in these patients is 85 to 95%.2 tee is the preferred approach in patients in whom tte is technically suboptimal and is the procedure of choice for imaging the pulmonic valve.3 when initial tee is negative and the clinical suspicion of ie remains , repeating tee within 7 to 10 days is advocated.4 perivalvular abscess or intracardiac fistula formation occurs in 10 to 14% of patients with nve.6 persistent , otherwise unexplained , fever despite appropriate antimicrobial therapy in patients with ie suggests infection extending beyond the valve leaflet . tee is superior to tte for detecting invasive infection in patients with nve and prosthetic valve endocarditis ( pve ) . cardiac surgery should be considered to debride abscesses , allowing the eradication of uncontrolled infection , and to reconstruct cardiac structures , restoring homodynamic and alleviating congestive heart failure . a 21-year - old man , who was an iv drug abuser , presented with fever and dyspnea . tte showed highly mobile , large vegetation on the anterior leaflet of the tricuspid valve and no vegetation on the aortic valve , mitral valve , and pulmonic valve . the patient was admitted to the cardiology ward for antibiotic therapy and received vancomycin and gentamycin . the blood culture of the patient became positive for staphylococcus areus 3 times in 24-hour intervals . despite the antibiotic therapy for 10 days tee revealed severe aortic insufficiency , moderate tricuspid regurgitation , and an echo - lucent space between the tricuspid and aortic valves ( figures 1 ) . color doppler demonstrated a flow within the echo - lucent space and a connection between that and the left ventricle , suggesting a perivalvular abscess of the tricuspid valve opening in the left ventricle ( figure 2 ) . after consultation with a cardiac surgeon , the patient was transferred to the operating room , where he unfortunately expired due to severe bleeding and disseminated intravascular coagulation . ie is a serious complication of iv drug abuse , with a reported mortality of 5 to 10%.1 endocarditis in iv drug abusers commonly involves the tricuspid valve , and staphylococcus aureus is the most common causative organism . dyspnea , cough , and chest pain are the common complaints of iv drug users . this is likely related to the predominance of tricuspid valve endocarditis in this group and secondary embolic showering of the pulmonary vasculature . within a week after the initiation of effective antimicrobial therapy , almost 70% of patients with nve or pve are afebrile and 90% have defervesced by the end of the second week of treatment.5 persistence or recurrence of fever more than 10 days after the initiation of antibiotic therapy identifies patients with increased mortality rates and with complications of infection or therapy.5 patients with a prolonged or recurrent fever should be evaluated for intracardiac complications . perivalvular infection beyond the valve leaflet results in abscesses in the annulus or adjacent structures , intracardiac fistulas , and purulent pericarditis . periannular extension is common , occurring in 10 to 40% of all native valve ie and complicates aortic valve endocarditis more commonly than mitral or tricuspid valve endocarditis.3 intra - cardiac fistulas are rarely seen and they are estimated to account for < 1% of all cases of ie.6 fistulization of the paravalvular abscess has been found in 6 to 9% of all cases.7 perivalvular abscess and intracardiac fistula of the tricuspid valve is very rare . ie is a lethal disease if not treated aggressively with parental antibiotics , often in combination with surgery . cardiac surgery should be considered in patients with perivalvular abscess and intracardiac fistula to debride abscesses and to reconstruct cardiac structures , restoring homodynamic and alleviating congestive heart failure .

infective endocarditis is a serious complication of intravenous ( iv ) drug abuse , with a reported mortality of 5 to 10% . a 21-year - old man , who was an intravenous drug abuser , presented with fever and dyspnea . transthoracic echocardiography showed a highly mobile , large vegetation on the anterior leaflet of the tricuspid valve . despite antibiotic therapy for ten days , the patient remained febrile . transesophageal echocardiography revealed severe aortic regurgitation and an echo - lucent space between the tricuspid and aortic valves . color doppler demonstrated a flow within the echo - lucent space and a connection between that and the left ventricle , suggesting a perivalvular abscess of the tricuspid valve opening in the left ventricle . the patient was transferred to the operating room , where he unfortunately expired .

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gastrointestinal perforations constitute one of the commonest surgical emergency encountered by surgeons [ 1 , 2 ] . management of these patients continues to be highly demanding despite the advances made in diagnosis and surgical therapy . the etiological spectrum of perforation peritonitis in india differs significantly from its western counter parts [ 35 ] . our study was carried out to highlight the spectrum of perforation peritonitis ( diffuse ) in a single unit at lok nayak hospital , a tertiary care hospital in delhi . the retrospective study was conducted at the department of surgery , maulana azad medical college and associated lok nayak hospital , delhi , from may 2010 to june 2013 . the study population included 400 patients of perforation peritonitis ( diffuse or localized ) presenting to the surgical emergency of lok nayak hospital , delhi , who underwent exploratory laparotomy . cases were studied with respect to clinical features at the time of presentation , comorbid conditions , radiological investigations , operative findings , and postoperative course . after establishing the clinical diagnosis of perforation peritonitis on performing exploratory laparotomy , the operative findings were noted and the source of peritonitis was found and managed accordingly . all patients were then treated in the postoperative ward initially under the cover of parenteral broad spectrum antibiotics and fluids ; orals were started on the appearance of bowel sounds . 98% patients presented with the history of abdominal pain , 62.5% with altered bowel habit , 41.5% with nausea and vomiting , and 28% with abdominal distention . 15% patients had positive history of nsaid intake for more than 6 months ( table 1 ) . in our study , the commonly associated comorbidity was chronic obstructive pulmonary disease followed by renal disease , diabetes , and hypertension . multiple air fluid levels on abdominal x - ray in erect position were present in 28% patients . electrolyte imbalances included hyponatremia in 21% , hypokalemia in 19% and elevated serum creatinine in 18% patients . most of the patients were operated within 24 hours of presentation under the cover of broad spectrum antibiotics after adequate resuscitation and correction of electrolyte imbalances . the commonest cause of perforation peritonitis in our study was gastroduodenal perforation due to acid peptic disease ( 45% ) followed by appendicitis ( 18.5% ) , typhoid fever ( 12% ) , tuberculosis ( 10% ) , and trauma ( 9% ) , ( table 2 ) . patients of peptic ulcer perforation usually had a short history of pain starting in epigastrium followed by generalized tenderness . 175 such were managed by an omental pedicle repair , in the other 4 cases a feeding jejunostomy was also done due to the large size of the perforation . 8% of these patients were managed by a limited resection with ileo - ascending anastomosis due to associated unhealthy caecum . patients of typhoid perforation had an initial history of high grade fever prior to abdominal complaints . of the 40 patients of tubercular perforation , 60% had previous history of tuberculosis and 50% of these patients took antitubercular therapy for < 6 months . in cases of traumatic perforation , the most common site was jejunum ( 49% ) followed by ileum ( 42% ) . the most commonly performed procedure was omental pedicle closure of peptic ulcer perforation ( 43.75% ) , followed by exteriorization of the gut in the form of ileostomy or colostomy ( 22.5% ) . appendectomy was the third most common procedure ( 17% ) , ( table 2 ) . the most common complication was wound infection followed by dyselectrolytaemia , abdominal collection , and respiratory complications . the morbidity rate was higher in the patients with intestinal perforation ( 58% ) than those with gastroduodenal perforation ( 32% ) . factors involved in death included septicemia due to fecal peritonitis , respiratory complications , pulmonary embolism , and late presentation . it is commonly seen in a younger age group in the tropical countries ( mean age in our study was 37.8 years ) as compared to the studies in the west [ 79 ] . more commonly the perforations involve the proximal part of the gastrointestinal tract ; [ 1013 ] this being in contrast to studies from the western countries , where perforations are common in the distal part [ 1416 ] . etiological factors also show a wide geographical variation . according to a study from india , infections formed the most common cause of perforation peritonitis , around 50% cases in this study were due to typhoid . in our study 22% of the cases were due to typhoid and tuberculosis . in contrast to this , noon et al . from texas in their study reported only 2.7% cases due to infections . also studies from the west have shown that around 1520% cases are due to malignancy [ 19 , 20 ] , this being in stark contrast to our study where malignancy was ascertained to be the cause of perforation peritonitis in only 3% of the cases . this shows that malignancy is not a common cause of perforation peritonitis in our setup as compared to our western counterparts . adequate preoperative resuscitation ( with fluids , etc . ) , correction of electrolyte imbalances followed by an early surgical intervention , to remove the source of infection and stop further contamination , is imperative for good outcomes minimizing morbidity and mortality . peptic ulcer perforation , perforating appendicitis , typhoid , and tubercular perforations are the major causes of gastrointestinal perforations . early surgical intervention under the cover of broad spectrum antibiotics preceded by adequate aggressive resuscitation and correction of electrolyte imbalances is imperative for good outcomes minimizing morbidity and mortality .

background . perforation peritonitis is the one of the commonest emergency encountered by surgeons . the aim of this paper is to provide an overview of the spectrum of perforation peritonitis managed in a single unit of a tertiary care hospital in delhi . methods . a retrospective study was carried out between may 2010 and june 2013 in a single unit of the department of surgery , lok nayak hospital , delhi . it included 400 patients of perforation peritonitis ( diffuse or localized ) who were studied retrospectively in terms of cause , site of perforation , surgical treatment , complications , and mortality . only those patients who underwent exploratory laparotomy for management of perforation peritonitis were included . results . the commonest cause of perforation peritonitis included 179 cases of peptic ulcer disease ( 150 duodenal ulcers and 29 gastric ulcers ) followed by appendicitis ( 74 cases ) , typhoid fever ( 48 cases ) , tuberculosis ( 40 cases ) , and trauma ( 31 ) . the overall mortality was 7% . conclusions . perforation peritonitis in india has a different spectrum as compared to the western countries . peptic ulcer perforation , perforating appendicitis , typhoid , and tubercular perforations are the major causes of gastrointestinal perforations . early surgical intervention under the cover of broad spectrum antibiotics preceded by adequate aggressive resuscitation and correction of electrolyte imbalances is imperative for good outcomes minimizing morbidity and mortality .

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. the baff / april axis plays an important role in the pathogenesis of various autoimmune diseases including sle ; inhibitors of this axis are therefore useful in treatment of these disordersresults from bliss-52 and bliss-76 trials have shown belimumab to be efficacious in treating patients with sleserious adverse effects noted with belimumab such as progressive multifocal leukoencephalopathy , depression , suicidal tendency , and malignancy are to be watched for . the baff / april axis plays an important role in the pathogenesis of various autoimmune diseases including sle ; inhibitors of this axis are therefore useful in treatment of these disorders results from bliss-52 and bliss-76 trials have shown belimumab to be efficacious in treating patients with sle serious adverse effects noted with belimumab such as progressive multifocal leukoencephalopathy , depression , suicidal tendency , and malignancy are to be watched for . . the baff / april axis plays an important role in the pathogenesis of various autoimmune diseases including sle ; inhibitors of this axis are therefore useful in treatment of these disordersresults from bliss-52 and bliss-76 trials have shown belimumab to be efficacious in treating patients with sleserious adverse effects noted with belimumab such as progressive multifocal leukoencephalopathy , depression , suicidal tendency , and malignancy are to be watched for . the baff / april axis plays an important role in the pathogenesis of various autoimmune diseases including sle ; inhibitors of this axis are therefore useful in treatment of these disorders results from bliss-52 and bliss-76 trials have shown belimumab to be efficacious in treating patients with sle serious adverse effects noted with belimumab such as progressive multifocal leukoencephalopathy , depression , suicidal tendency , and malignancy are to be watched for .

belimumab is the only approved biological agent for the treatment of systemic lupus erythematosus ( sle ) . it is a fully humanized igg1 monoclonal antibody directed against soluble b lymphocyte stimulator ( blys ) . it is indicated as an add - on therapy for the treatment of adult patients with active , autoantibody - positive sle , who are receiving standard therapy . belimumab is generally well - tolerated , common adverse effects include infections , infusion reactions , hypersensitivity , headache , nausea , and fatigue . psychiatric events including suicidal tendency , progressive multifocal leukoencephalopathy and malignancies too have been reported . apart from sle , the drug is also being tried for other autoimmune disorders .

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cementoblastoma in the current world health organization classification of odontogenic tumor , is in the category of tumors of mesenchyme and/or odontogenic ectomesenchyme with or without odontogenic epithelium . it generally occurs in young persons , comprises < 1 - 6.2% of all odontogenic tumor and is characterized as being attached to the roots , most frequently associated with first permanent molar . the majority of these tumor are radiopaque , but radiolucent tumor may occur in rare instances . histologically , it presents as a well - circumscribed tumor composed of cementum like tissue surrounded by a fibrous capsule . this was a case report of a 16-year - old male patient who reported to the department of oral medicine , gdc , ahmedabad with the chief complaint of swelling and mild pain in the right side of the jaw since 6 months , with a history of extraction of 46 because of pain before 6 months in a private dental clinic . on clinical examination , there was diffuse bony hard swelling present in the right body of mandible with normal overlying skin . intra - orally , there was diffuse bony hard swelling in 46 region with normal overlying mucosa and expanded buccal and lingual cortical plates [ figure 1 ] . adjacent teeth were immobile and undisplaced . radiological examination revealed a well - defined round radiopacity with radiolucent rim in the right body of mandible [ figure 2 ] . he had pre - extraction intraoral periapical radiography ( iopa ) radiograph of 46 region , which showed a well - defined radiopacity surrounded by radiolucent rim attached to roots of 46 [ figure 3 ] . the typical radiographic presentation of radiopacity attached to root with radiolucent rim suggested of benign cementoblastoma with differential diagnosis of hypercementosis , osteoblastoma , periapical cemental dysplasia and condensing osteitis . histopathologically it showed a well - circumscribed tumor composed of cementum like tissue surrounded by a fibrous capsule [ figure 4 ] . intra - oral view showing diffuse swelling in right lower quadrant with missing 46 and expansion of buccal and lingual cortical plates right lateral oblique of mandible showing well defined radiopacity surrounded by radiolucent zone in body of mandible with missing 46 pre - extraction radiograph showing radiopaque lesion attached to roots of 46 microphotograph showing sheet of cementum like tissue with intervening loose fibrovascular connective tissue stroma the male to female ratio of the prevalence has been reported to be 2.1:1 with a mean age of 20.7 years . benign cementoblastoma is also reported in the maxillary sinus and associated with deciduous and unerupted permanent tooth and multiple teeth . clinical sign and the radiographic appearance of benign cementoblastoma is well - defined radiopacity with radiolucent zone . though it is a benign tumor , but some cases reported in the literature exhibited signs of local aggressiveness and destruction , including bony expansion , erosion of cortical plates , displacement of adjacent teeth , maxillary sinus involvement , invasion of the pulp chamber and root canals and extension to and incorporation of adjacent teeth . expansion , pain and erosion or perforation of bony cortex were seen in a higher percentage of recurrent tumors , but were also seen in non - recurrent tumors . the differential diagnosis for a periapical radiopacity include cementoma , osteoblastoma , periapical cemental dysplasia , condensing osteitis and hypercementosis . histologically , this tumor presents sheets of cementum like tissue , which may contain a large number of reversal lines with active cementoblasts . cementoblastoma and osteoblastoma are essentially identical histologically and the only distinguishing feature is attachment of cementoblastoma to the root of a tooth . histologically , the cementoblast in cementoblastoma may be plump with pleomorphic and hyperchromatic nuclei , however , mitotic figures are not seen in cementoblastoma . in contrast to osteoblastoma , the cementoblastoma is an odontogenic tumor that recapitulates cementum deposition similar to that during root formation in the later stages of odontogenesis . furthermore , the cementoblastoma is continuous with the cemental layer of the apical third of the tooth root and remains separated from bone by a continuation of the periodontal ligament , all of which supports an odontogenic origin . whereas osteoblastoma arises in the medullary cavity of bones . the treatment of choice is complete removal of the lesion with extraction of associated tooth , followed by thorough curettage and peripheral ostectomy . cases have been also reported of endodontically preserving the tooth while surgical removal of benign cementoblastoma is done . we presented a rare case of benign cementoblastoma in mandible and it should be considered in differential diagnosis of bony swellings of mandible . when extraction is attempted in such cases leaving the lesion behind makes the clinical diagnosis difficult . though the patient had pre - extraction records , which helped in formulating the diagnosis .

we present a rare case of radiopaque lesion with radiolucent rim in the right body of mandible , with history of extraction of involved tooth , which made diagnosis confusing unless pre - extraction intraoral periapical radiography radiograph was recovered , finally diagnosed as benign cementoblastoma . it was surgically managed , with no recurrence of the lesion more than 2 years of follow - up . benign cementoblastoma is a rare , benign tumor of odontogenic ectomesenchymal origin , usually associated with roots of first mandibular molar .

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the anterior choroidal artery ( achoa ) supplies several important structures including the optic tract , geniculate body , posterior limb of internal capsule , globus pallidus , cerebral peduncle , uncus , amygdala , ventrolateral nucleus of thalamus , and choroid plexus . the vascular injury of achoa may result in severe neurological deficits such as hemiplegia , hemianesthesia , hemianopsia , decreased consciousness , and extrapyramidal symptoms . therefore , complete knowledge about vascular anatomy of achoa is very important before surgical or endovascular manipulations . although several detailed reports about anatomical variations of achoa have been reported , transposition of achoa as to posterior communicating artery ( pcoma ) origin was demonstrated only in 3 reports2 - 4 ) . we report a case with ruptured cerebral aneurysm arising from pcoma , which originated from the internal carotid artery ( ica ) distal to the origin of achoa . a 40-year - old woman presented with sudden onset of headache . computed tomography ( ct ) revealed subarachnoid hemorrhage ( sah ) which was slightly predominant in the left basal and sylvian cisterns . two small aneurysms arose from both distal supraclinoid ica , these were observed on ct angiograms ( fig . cerebral angiography demonstrated a small aneurysm arising from left fetal type of pcoma with suspicion of rupture and the other small aneurysm originating from right pcoma or perforating branch of ica . in this study , left achoa originated from ica proximal to the origin of fetal type of pcoma ( fig . achoa usually arises from the posterolateral wall of ica , a few mm distal to the origin of pcoma , and it provides blood supply to the important neural structures around crural and ambient cisterns . the achoa can have several variations in the course , size , number , branching pattern , and brain region supplied by this artery1 ) . among them , there have been several anomalies related to the origin of achoa including transposition of achoa and pcoma , origin from pcoma , origin from junction of ica and pcoma , and origin from middle cerebral artery4,5 ) . the clinical findings of transposed achoa cases including present one are summarized in table 1 . all of the transpositions of achoa occurred in the left side . among all the cases , our case showed sah due to ruptured aneurysm arising from fetal type of pcoma among multiple cerebral aneurysms founded and achoa originating from ica proximal to pcoma . typically , achoa runs posteromedially within the crural cistern and continues ambient cistern to enter the choroidal fissure . the proximal portion of this artery is cisternal segment and following portion becomes intraventricular segment1 ) . the transposed achoa in the previous cases including ours did not seem to affect the vascular territory or course despite of unusual vascular arrangements . the achoa can be exposed or manipulated in surgical approaches into the pathological lesions around temporal lobe or involving supraclinoid ica . although the achoa aneurysms accounting for 4% of all cerebral aneurysms are not so common , vascular injury of achoa with small caliber supplying the eloquent areas may be critical because of the possibility of significant morbidity and mortality . and , another caution is that achoa do not have significant collateral flows despite a potential anastomosis with posterior choroidal artery3 ) . we tried to preserve pcoma itself and all perforating branches arising from it because of the possibility to compromise the collateral flows of fetal type pcoma . it is notable that careful review of the preoperative angiograms helps to find unexpected vascular anomalies which may impose an additional surgical risk .

the anterior choroidal artery has several kinds of variations . among them , the transposition of anterior choroidal artery and posterior communicating artery origins has been extremely rare . we report a case with cerebral aneurysm arising from posterior communicating artery which origin was distal to the anterior choroidal artery and review the relevant literature .

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in recent years , video - assisted intubation devices have flooded theaters , and , to a lesser extent , are also being used in icus . but can such new technology really contribute to making tracheal intubation in the icu less life - threatening , or even more life - saving ? noppens and colleagues , in a study just published in critical care , evaluated the efficacy of the c - mac video laryngoscope for endotracheal intubation in an icu managed by anesthesiologists and compared the results to those obtained during a previous period when conventional direct laryngoscopy was used . in patients with at least one predictor for difficult intubation , poor glottic views ( cormack and lehane grades iii and iv ) were reduced from 38% to 19% , and intubation success on the first attempt increased from 56% with direct laryngoscopy to 79% when the c - mac video laryngoscope was used . even in all patients , the incidence of at least two intubation attempts though this was not statistically significant , halving this incidence may have substantial clinical impact . although the overall intubation success was 100% in either group , successful tube placement in the trachea on the first attempt is crucial in icu patients with poor tolerance to apnea . after implementation of a video laryngoscope , however , intubations were more often performed by less experienced physicians . even if one reason may be that younger physicians are more open to the newer technique , it must be guaranteed that physicians ' experience is not substituted solely by technical equipment . similarly , the use of endoscopic intubation was reduced substantially in the intervention phase ( 4% versus 13% ) , so experience with such devices may decrease , causing problems if video laryngoscopy is not possible or is contraindicated . video laryngoscopy for every patient ? sparing such devices for the really difficult cases ? to answer these questions it needs to be emphasized that all video - assisted and optical intubation devices have different view angles , thus producing different images with particular distortions , and even experts in ' old - fashioned ' airway management need a substantial level of training with a certain device before using it safely and successfully in critical situations and patients . video laryngoscopes , regardless of a particular brand or device , can not be used intuitively - they require expert skills and routines to be tamed and turned into life - saving tools . tube placement can turn out to be difficult and even impossible , despite the excellent glottic exposure video laryngoscopes most often provide . the reason is that the operator 's eye is no longer in a straight - optical and anatomical - line with the larynx , but at the tip of the laryngoscope blade . optical and anatomical axes divert , and tube placement is sometimes difficult or fails . becoming alert to these potential problems associated with video laryngoscopy and constantly improving one 's technical skills to overcome such pitfalls can only be achieved when such devices are used on a daily basis and in routine patients . apart from the primary aim of the study - evaluating video laryngoscopy - it needs to be highlighted that noppens and colleagues routinely used capnography to verify correct tube placement after intubation . capnography has become a mandatory requirement in any operating room , but its use in the icu seems still to be somewhat dowdy . as of 2010 , only 25% of icus in the uk and the republic of ireland used capnography to confirm correct tube placement after intubation . such refusal may produce catastrophic outcomes : data collected from all uk national health service hospitals over a period of one year in 2008/2009 revealed that 61% of adverse events during airway management in the icu resulted in death or permanent brain damage . in 74% of such fatal cases no capnography was used ! doubtless , video laryngoscopy is a helpful tool in the hands of experienced physicians , but should not be considered a bailout procedure for beginners in airway management . and as attractive and simple video laryngoscopy appears , we must always remind ourselves to maintain our skills in endoscopic intubation . both authors receive continuous material support for clinical studies from karl storz gmbh & co. kg , the manufacturer of the c - mac video laryngoscope .

the c - mac video laryngoscope substantially reduced poor glottic views and increased intubation success in icu patients with at least one predictor for difficult intubation . however , all video - assisted and optical intubation devices have different view angles , thus producing different images with particular distortion , and even experts in ' old - fashioned ' airway management need a substantial level of training with a certain device before using it safely and successfully in critical situations and patients . video laryngoscopes , regardless of a particular brand or device , can not be used intuitively - they require expert skills and routines to be turned into a life - saving airway management tool .

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septic shock is consistently the most common causative factor identified for acute kidney injury ( aki ) in critical illness , and has been associated with nearly 50% of cases internationally . despite advances in our understanding of the pathophysiology of septic aki , treatment aimed at reversing or preventing septic aki remains primarily based on supportive haemodynamic management . in the previous issue of critical care , legrand and colleagues examine the association between haemodynamic targets of resuscitation ( cardiac output , mean or diastolic blood pressure , central venous pressure ( cvp ) and central venous oxygen saturation ) and development or persistence of aki in a single centre study of patients with septic shock admitted to a surgical icu . the authors found that only higher cvp and low diastolic blood pressure were associated with increased risk of development of new aki , or persistence of renal dysfunction present at icu admission . in these patients importantly , the association between cvp and aki remained when potentially confounding effects of positive fluid balance and higher positive end - expiratory pressure were accounted for in a multivariable analysis . so , a 5 mmhg increase in cvp predicted 2.7-fold odds of new or persistent aki . furthermore , when the association between different levels of mean cvp and aki was examined in a non - parametric logistic regression , there was a trend for higher cvp to be associated with worse renal outcome for all levels of cvp from 4 mmhg upward , so that a cvp of 15 mmhg was associated with an approximately 80% risk of new or persistent aki , compared to approximately 30% at a cvp of 6 mmhg . these findings are important because current surviving sepsis campaign guidelines recommend fluid resuscitation of patients with sepsis - induced tissue hypoperfusion to target a cvp of 8 to 12 mmhg ( or 12 to 15 mmhg in mechanically ventilated patients ) within 6 hours of presentation . in patients with sepsis - induced hypotension the rationale for cvp targeted fluid resuscitation is to ensure ' adequate cardiac preload and hence maintain cardiac output and organ perfusion . however , absolute levels or changes in cvp poorly predict cardiovascular response as sepsis - induced hypotension is multi - factorial , related to changes in myocardial performance , vascular tone , regional blood flow distribution , venous reservoir capacity and capillary permeability . in contrast , elevated cvp will cause a direct and predictable increase of renal venous pressure that , experimentally , has been associated with elevated renal interstitial and intra - tubular pressure . resultant renal venous congestion can reduce renal blood flow ( lower trans - renal pressure gradient ) and decrease the pressure gradient for ultrafiltration ( higher intra - tubular pressure ) . in septic shock , . aggressive fluid resuscitation beyond reversal of hypovolaemia to arbitrary cvp targets could result in increased venous congestion without substantial benefit to forward renal perfusion , thus predisposing to renal dysfunction . the association between elevated cvp and renal dysfunction has been made previously in the setting of chronic cardiac failure . now legrand and colleagues have provided evidence that such an association is observed in septic shock and its treatment . their findings are also in accord with secondary analysis of the vasopressin in septic shock trial , which reported that a more positive fluid balance and elevated cvp were associated with increased mortality in patients with septic shock even when accounting for differences in baseline illness severity and demographics . although these observational findings are hypothesis - generating rather than confirmatory , they add weight to the literature suggesting the adverse effects of fluid overload in critical illness and aki in particular , effects that may be mediated in part by venous congestion causing impaired organ function . it is now important to determine whether uncritical pursuit of cvp targets such as those advocated by the surviving sepsis campaign might , in some patients , add insult to sepsis - induced organ injury . prospective data are required to evaluate the true contribution of raised venous pressure to renal and other organ dysfunction and to determine better endpoints for fluid resuscitation in sepsis . rajkumar rajendram bsc mbbs mrcp frca fficm , specialty registrar in anaesthesia , royal free hospital , london . john r prowle ma msc md mrcp fficm , consultant in renal medicine and intensive care , the royal london hospital , london .

in critical illness , septic shock is a contributing factor in nearly half of all cases of acute kidney injury ( aki ) . traditional approaches to prevention of organ dysfunction in early sepsis have focused on prevention of hypoperfusion by optimisation of systemic haemodynamics , primarily by fluid resuscitation . fluid administration to a target central venous pressure ( cvp ) of 8 to 12 mmhg ( 12 to 15 mmhg in mechanically ventilated patients ) is currently recommended for the early resuscitation of septic shock . however , in the previous issue of critical care , legrand and colleagues report that higher cvp in the first 24 hours of icu admission with septic shock was associated with increased risk for development or persistence of aki over the next 5 days . this study highlights a potential association between venous congestion and the development of septic aki , suggesting that cvp - targeted fluid resuscitation in septic shock might not be beneficial for renal function .

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one hundred forty - seven clinical isolates of m. tuberculosis were obtained from the molecular mycobacteriology laboratory , department of microbiology , faculty of medicine , siriraj hospital , mahidol university , thailand , and the t-2 project from 1997 to 2001 ( table ) . these strains were isolated from 74 cerebrospinal fluid ( csf ) samples and 73 sputum samples from 147 different patients . dna from these isolates was isolated by an enzymatic method and submitted for genotyping by performing the is6110 restriction fragment length polymorphism with the standard method ( 12 ) and for sequencing the pks15/1 region ( 8) . * csf , cerebrospinal fluid . using the genotyping results , we categorized m. tuberculosis isolates into beijing , single - banded , few - banded ( 25 bands ) , nonthaburi , and heterogeneous with > 5 bands ( table and figure 1 ) , as recently reported ( 13,14 ) . all m. tuberculosis genotypes were sequenced around the junction of pks15 and pks1 ( corresponding to the m. tuberculosis h37rv sequence ) to determine whether they contained an intact pks15/1 or separated pks15 and pks1 . unexpectedly , the results showed that the 7-bp insertion of pks15 that causes a frameshift mutation resulting in an intact pks15/1 was found in most strains of all genotypes , except the heterogeneous group with > 5 bands ( table and figure 2 ) . r indicates the m. tuberculosis mt 14323 strain used as the positive control for is6110 typing . sequence alignment of region corresponding to the 3 portion of pks15 and 5 portion of pks1 in various mycobacterium tuberculosis genotypes . b ) m. tuberculosis strains isolated from sputum . letters in brackets refer to is6110 restriction fragment length polymorphism patterns : bj , beijing ; sb , single banded ; fb , 25 bands ; nb , nonthaburi ; h , heterogeneous . the 7-bp insertion is shown in boldface , and the start codon of the pks1 gene is underlined . the intact pks15/1 has been shown to be responsible for the production of phenolic glycolipids and is seemingly found in m. tuberculosis w - beijing family , but it was not found in m. tuberculosis cdc1551 and h37rv ( 8) . previous studies suggested that pgls produced by the m. tuberculosis w - beijing family were associated with the hypervirulent phenotype by inhibiting the innate immune response ( 6,7 ) . the intact pks15/1 has also been shown to be nonpolymorphic in the w - beijing family ; it was found in all 102 w - beijing strains tested ( 15 ) . from this observation , we hypothesized that if the ability to produce pgls is among the factors that make this family more virulent than others , the intact pks15/1 should be absent in strains other than the w - beijing family . our results showed that the 7-bp insertion of the pks15/1 was not only present in the w - beijing family but also in other m. tuberculosis genotypes . although almost all beijing strains contain the intact pks15/1 ( 97% ) , 38.5%100% of strains of other genotypes also contain it . these strains could , therefore , produce pgls and cause both pulmonary and disseminated diseases as the w - beijing strains do . our results showed no significant difference in the percentage of m. tuberculosis isolates with an intact pks15/1 gene between csf isolates ( 65 [ 87.8% ] of 74 ) and sputum isolates ( 62 [ 84.9% ] of 73 ) . the hypothesis that the hypervirulence of the w - beijing family is solely attributable to pks15/1 is still inconclusive . this family may have only recently been transmitted globally and may have had more chances to cause infections and disease than other families . although pgls are involved in the hypervirulence of the pgl - producing strains , they are not a unique characteristic of the w - beijing family . if w - beijing strains are more virulent than others , other virulence determinants besides pgls must be responsible for the hypervirulent phenotype .

to determine whether intact pks15/1 is unique to the w - beijing family , we investigated 147 mycobacterium tuberculosis strains with different is6110 genotypes . intact pks15/1 was found in 87.8% of cerebrospinal fluid and 84.9% of sputum isolates . it was found not only in w - beijing strains ( 97% ) but also in other genotypes ( 38.5%100% ) .

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dr . coyne - beasley receives research funding from merck & co. none of these funds were used in the conduction or completion of the research contained in this manuscript . this article was supported by grants from the national institutes of health 1r01ai113305 - 01 ( cates pi ) , and by the north carolina translational and clinical sciences institute , through support from the national center for advancing translational sciences ( ncats ) , national institutes of health , grant award number 1ul1tr001111 . the content is solely the responsibility of the authors and does not necessarily represent the official views of the nih .

a significant barrier to the delivery of hpv vaccine is reluctance by both healthcare providers and parents to vaccinate at age 11 or 12 , which may be considered a young age . this barrier has been called vaccine hesitancy in recent research . in this commentary , we suggest using social marketing strategies to promote hpv vaccination at the recommended preteen ages . we emphasize a critical public health message of a sexually transmitted infection ( sti ) as preventable and vaccination against hpv as a way to protect against its consequences . the message tackles the issue of vaccine hesitancy head on , by saying that most people are at risk for hpv and there is a way to prevent hpv 's serious consequences of cancer . our approach to this conversation in the clinical setting is also to engage the preteen in a dialog with the parent and provider . we expect our emphasis on the risk of sti infection will not only lead to increased hpv vaccination at preteen ages but also lay important groundwork for clinical adoption of other sti vaccines in development ( hiv , hsv , chlamydia , and gonorrhea ) as well as begin conversations to promote sexual health .

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restoration of structure and function using autologous free fat grafts has always been a challenge . the advent and refinement of liposuction and lipoinjection techniques , abundant donor - tissue availability , and relative ease of harvesting have made autologous fat an attractive material for use as soft - tissue filler . fat is harvested using either ( 1 ) syringe aspiration or ( 2 ) liposuction aspiration,(1 ) performed with a liposuction pump ( byron medical , tucson , ariz . ) . a standard coleman aspirator cannula ( byron medical ) with a curettage tip ( 500 mm mercury).(2 ) syringe aspiration is tedious if the amount of fat required is substantial . when the liposuction aspiration using a liposuction pump is performed the whole suction system , and the container needs to be kept sterile . ( figure 1 ) we have devised a novel method of lipoaspiration to overcome these problems . the above picture helps us to compare the three modalities of lipoaspiration and also helps us to note the advantage of our technique materials required 20 - 50 cc disposable syringe , a piece of 2 2 cm polypropylene mesh , lipoaspiration suction pump ( figure 2a ) . ( a ) materials required for the procedure , ( b ) connected syringe mesh system , ( c ) close - up view of the mesh in liposuction aspiration , a suction pump is used . in our technique , a piece of polypropylene mesh is kept at one end of the lipoaspiration syringe , which is then connected to the suction pump . ( figure 2b & 2c ) as one syringe fills , it is replaced by another one until the required amount of fat is obtained . these syringes are then centrifuged and the emulsified fat thus obtained is used for grafting ( video 1 ) . by using a polypropylene mesh in our technique , we are able to separate the transfusate from the harvested fat during harvesting itself in our experience , the fat graft obtained by this method is found to be concentrated and has fewer contaminants . this method also has the added advantage of having a short processing time , from harvesting to grafting . fat grafting remains unpredictable , and various anecdotal harvesting and preparation techniques have been advocated . our method is a more economical and an easier way to extract sterile emulsified fat . as it is a simple method , it can be practiced even in day care centers . hence , we recommend our technique as a reliable and routine method for extracting sterile emulsified fat in an economical way .

background : successful restoration of structure and function using autologous free fat grafts has remained elusive . review of literature shows that various harvesting and preparation techniques have been suggested . the goal of these techniques is to obtain greater adipocyte cell survival and consequently more reliable clinical results.materials and methods : in our technique , a piece of mesh is kept at one end of the lipoaspiration syringe , which is then connected to the suction pump . as one syringe fills , it is replaced by another one until the required amount of fat is obtained.results:by using a polypropylene mesh in our technique , we can separate the transfusate from the harvested fat graft during harvesting itself . the fat graft thus obtained is dense and concentrated , with fewer impurities.conclusion:hence , we recommend our technique as a reliable method for extracting sterile emulsified fat in an economical way .

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turner syndrome ( ts ) is caused by partial or complete monosomy of the x chromosome . only a few cases of ts with ischemic stroke have been reported [ 2 , 3 , 4 , 5 ] . in previously reported cases , various arteriopathies affecting the cerebral arteries such as fibromuscular dysplasia , hypoplasia , moyamoya syndrome , and , we report a case of ts with multiple embolic infarcts caused by a cryptogenic embolism . a 28-year - old woman with ts was referred to our hospital because of abnormal findings on brain magnetic resonance imaging ( mri ) . she was diagnosed with ts [ karyotype : 45 , x/46 , x , + mar ] at the age of 16 years due to amenorrhea . she denied alcohol , tobacco , or drug use and had no family history of neurologic disorders . the patient was afebrile with a blood pressure of 180/130 mm hg and a heart rate of 89 beats / min . she was obese with a body mass index of 27.3 kg / m ( 160 cm height , 70 kg weight ) . she visited hospital due to sudden - onset severe headache with high blood pressure ( 240/140 mm hg ) . she underwent brain computed tomography and mri at the referring hospital for differential diagnosis of thunderclap headache including hypertensive intracranial hemorrhage , subarachnoid hemorrhage , arterial dissection , or reversible cerebral vasoconstriction syndrome . brain computed tomography was unremarkable , but diffusion - weighted imaging showed restricted diffusion in multiple lesions involving multiple vascular territories ( fig 1a ) . cerebral magnetic resonance angiography ( mra ) ( fig 1b ) and carotid sonography demonstrated no extracranial or intracranial arterial stenosis . laboratory tests revealed diabetes mellitus ( fasting plasma glucose 219 mg / dl , hemoglobin a1c 11.0% ) and hyperlipidemia ( low - density lipoprotein 189 mg / dl ) . transthoracic echocardiography revealed normal valvular structure and function with a normal left ventricular ejection fraction . patent foramen ovale , atrial septal aneurysm , or any aortic pathologies including aortic atheroma were not detected in transesophageal echocardiography . the results of the hypercoagulability panel , vasculitis panel , and cerebrospinal fluid examination were all negative . aspirin was administered , and medications for hypertension , hyperlipidemia , and diabetes were also started . within few hours after blood pressure control , the patient was discharged from the hospital without any symptoms of stroke , and recurrence was not observed in the 2 years after discharge . cerebrovascular arteriopathies such as fibromuscular dysplasia , congenital hypoplasia , moyamoya syndrome , and premature atherosclerosis have been shown to be causes of ischemic stroke in previous case reports . in this case furthermore , our patient showed multiple lesions in different vascular territories that were strongly suggestive of embolic stroke . atrial fibrillation ( af ) could also be considered a potential source of embolic stroke in ts , as p - wave dispersion , a potential substrate for af , is increased in ts . the classical risk factors for developing af include hypertension , diabetes mellitus , and valvular disease , and these are more common in ts than in the general population . however , we could not completely exclude the possibility of paroxysmal af , as holter monitoring has a low yield for af detection ( 35% ) . other possible cardio - aortic sources of embolism include intracardiac shunts , thrombi , calcifications / vegetations in the mitral valve , or aortic atheroma plaque . we sought to identify possible cardio - aortic sources on transthoracic echocardiography , transesophageal echocardiography , and transcranial doppler shunt test ; however , all tests were negative . some case reports have described deep venous thrombosis and portal vein thrombosis in ts [ 11 , 12 ] . these factors include fibrinogen , d - dimer , factor viii , von willebrand factor , and proteins c and s . we also considered the possibility of hypercoagulability as a cause of stroke in this case ; however , levels of d - dimer , fibrinogen , protein c and s were normal . the embolic source of stroke in our patient remains unclear ; however , this is the first report of a ts patient with an embolic stroke pattern . our case suggests another possible mechanism of stroke in ts that is different from previous case reports . ischemic stroke in ts could be due to embolism as well as various cerebral arteriopathies . further investigation is needed to more fully understand the various mechanisms of ischemic stroke in ts . ischemic stroke in ts could be due to embolism as well as various cerebral arteriopathies , as documented in previous reports . further studies are needed to determine the extent of the underlying mechanisms of ischemic stroke in ts . this case report has been approved by the institutional review board of the inha university hospital .

only a few cases of turner syndrome ( ts ) with ischemic stroke have been reported . various arteriopathies of the cerebral arteries , including fibromuscular dysplasia , congenital hypoplasia , moyamoya syndrome , and premature atherosclerosis have been assumed to be the cause of ischemic stroke in ts . there has been no case report of a ts patient presenting with an embolic stroke pattern without any cerebral arteriopathy . a 28-year - old woman with ts was referred to our hospital because of abnormal brain magnetic resonance imaging ( mri ) findings . she underwent brain mri at the referring hospital because she experienced sudden - onset diffuse headache . diffusion - weighted imaging revealed multiple acute embolic infarcts in different vascular territories . intracranial and extracranial arterial disease was not detected on cerebral magnetic resonance angiography and carotid sonography . embolic source workups , including transthoracic and transesophageal echocardiography , holter monitoring , and transcranial doppler shunt study , were all negative . hypercoagulability and vasculitis panels were also negative . our patient was diagnosed with cryptogenic embolic stroke . this is the first report of a ts patient with an embolic stroke pattern . our case shows that ischemic stroke in ts could be due to embolism as well as the various cerebral arteriopathies documented in previous reports .

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pulmonary arteriovenous malformation ( pavm ) , first described in 1897 by churton , is characterized by abnormal communications between pulmonary veins and arteries , and is known to disturb the filtering action of pulmonary capillaries , causing thromboembolic event in systemic circulation . most of the pavms have no symptoms , and are detected as abnormal shadow in the chest . conclusive diagnosis is generally made by means of pulmonary arteriography or three - dimensional computed tomography ( 3d - ct ) angiography . while hypervascular lesions can mimic pavm , we observed a rare case of a lung granuloma mimicking pavm , and performed video - assisted thoracic surgery ( vats ) . a 76-year - old woman , who was otherwise symptom - free , was admitted to our hospital because an abnormal shadow was detected in the left lung field on her chest x - ray . chest computed tomography ( ct ) revealed a 20 mm 14 mm nodule with well - defined margins and smooth contours in the left upper segment ( fig . an enhanced solitary lung nodule , connected with linear structures suggestive of feeding artery and drainage vein , was revealed by contrast - enhanced 3d - ct ( fig . arterial blood gas analysis showed hypoxemia , with arterial oxygen pressure ( pao2 ) 56 mmhg in room air . first , the patient was placed in the right lateral decubitus position , and the left lung was deflated . intraoperatively , elastic hard nodule was palpable in the left upper segment and bruit was not convincing . the nodule was nontortuous in shape , covered with the visceral pleura , and neither the feeding artery nor the drainage vein was detected . histopathological findings revealed multiple foci of coagulative necrosis surrounded by epithelioid cell granuloma containing langhans - type multinucleated giant cells , involving the medium - sized blood vessels in the pulmonary parenchyma . the lung granuloma was difficult to be preoperatively distinguished from pavm , because hypervascular lesion such , as inflammatory changes can present as strongly enhanced nodules after injection of contrast material . the patient had an uneventful postoperative course and was discharged 14 days after the operation . causes of acquired or secondary pavm include chest trauma , thoracic surgery , hepatic cirrhosis , infections ( actinomycosis , schistomiasis ) , metastatic carcinoma and systemic disease . twenty eight percent of cases are considered to have no symptoms . it was reported that when a single isolated pulmonary arteriovenous malformation is 2 cm or smaller , no symptoms appear . generally , when pavm is 2 cm or more , symptoms such as breathing difficulty , cyanosis , hypoxemia , finger clubbing and polycythemia occur , and the rate of occurrence of severe complications , such as rupture of the malformation , hemoptysis , cerebral infarction and cerebral abscess , is approximately 30% . the patient in the present case was asymptomatic and had no history of chest trauma , surgery , infection or systemic disease . abnormal vascular structures , such as pavm are not convincing in histopathological findings . coincidentally , the inflammatory granuloma of the lung looked almost identical to pavm because of the nontortuous shape with linear structures suggestive of feeding artery and drainage vein . the resection of lung granuloma is highly successful for an isolated malformation , and mortality rate is low in this surgery . embolization is an appropriate treatment modality in multiple pavm for which surgery is not suitable . complications of embolization include pleuritic chest pain , pulmonary infection , air embolism , migration of coils and paradoxical embolism . although pulmonary angiography or contrast - enhanced 3d - ct has been the standard diagnostic tool for pavm , it has limitations in detecting pavms presenting as small nodules and enhanced nodules . in our case , since the enhanced lung nodule size was 22 mm in diameter , it mimicked pavm . in this patient , it was difficult to distinguish the lung granuloma from pavm , preoperatively . though there is a report which shows lung cancer mimicking pavm , to our knowledge , this is the first report of a lung granuloma mimicking pavm . in conclusion , clinicians should pay particular attention to enhanced nodules to rule out a variety of disorders , including neoplasm , infection , inflammatory and vascular abnormality when pavm was suspected . for the purpose of not only diagnosis but also for safety in treatment of pavm , the surgical approach seems appropriate . written informed consent was obtained from the patient for publication of this case report and accompanying images . a copy of the written consent is available for review by the editor - in - chief of this journal on request . yoshinobu ichiki : study design , data collections , data analysis , writing ; junji kawasaki : data collections ; takayuki hamatsu : data collections ; taketoshi suehiro : data collections ; makiko koike : data collections ; fumihiro tanaka : study design , data collections , data analysis ; keizo sugimachi : data collections .

introductionwhile hypervascular lesions in the lung are known to mimic pulmonary arteriovenous malformation ( pavm ) , here we report a rare case of lung granuloma mimicking pavm , on which video - assisted thoracic surgery ( vats ) was performed.presentation of casea 76-year - old woman without any symptom was admitted to our hospital because of abnormal shadow in the left lung field on chest x - ray . a 20 mm 14 mm nodule with well - defined margins and smooth contours in the left upper segment was detected in her chest computed tomography ( ct ) . contrast - enhanced three - dimensional ct ( 3d - ct ) revealed an enhanced solitary lung nodule , which was connected with linear structures suggestive of feeding artery and drainage vein . thus , we made a preoperative diagnosis of pavm by performing partial pulmonary resection by vats . intraoperatively , elastic hard nodule was palpable in the left upper segment and bruit was not convincing . histopathological findings revealed multiple foci of coagulative necrosis surrounded by epithelioid cell granuloma containing langhans - type multinucleated giant cells , involving the medium - sized blood vessels in the pulmonary parenchyma . abnormal vascular structures , such as pavm were not convincing . based on these findings , a diagnosis of left lung granuloma was made.discussionpreoperatively , it was difficult to distinguish the left lung granuloma from pavm , because hypervascular lesion , such as inflammatory changes can present as strongly enhanced nodules after injection of contrast material.conclusionsurgical approach seems appropriate , not only for the purpose of diagnosis , but also for the safety in treatment of a pavm .

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c57bl/6j male mice ( 25 months of age ) were housed individually on a 12 hr/12 hr light / dark schedule with lights on at 7 a.m. ( zt0 ) and handled for 6 days . mice were sleep - deprived ( sd ) in their home cages for 5 hours by gentle handling beginning at zt5 or left undisturbed ( non - sleep - deprived mice , nsd ) . for contextual fear conditioning experiments , animals were placed in a novel chamber for 3 minutes , and received a 2-second , 1.5 ma footshock after 2.5 minutes . mice received intra - peritoneal injections of rolipram ( rol ; 1 mg / kg ) or vehicle ( 2% dmso in 0.9% saline ) immediately and 2.5 hours post - training . testing of contextual memory was performed 24 hours after training in the trained context and 48 hours after training in a novel chamber . 1-train ltp was induced by a single 100 hz , 1-second duration train of stimuli . 4-train ltp consisted of 4 trains applied with a 5-minute inter - train interval ; for massed 4-train ltp a 5-second inter - train interval was used . theta - burst stimulation ( tbs ) consisted of 40-ms duration , 100 hz bursts delivered at 5 hz for 3 seconds ( 15 bursts of 4 pulses per burst , for a total of 60 pulses ) . chemical ltp was induced by treatment of slices for 15 minutes with 5 m forskolin ( fsk ) in 0.1% ethanol , or a combination of 50 m forskolin and 30 m 3-isobutyl-1-methylxanthine ( ibmx , in water ) . rolipram ( 0.1 m in 0.1% dmso ) was applied for 60 minutes , beginning 30 minutes before tetanization . camp assays on ca1 regions of hippocampal slices 10 minutes after treatment for 15 minutes with forskolin ( 50 m ) , forskolin + ibmx ( 30 m ) , or vehicle ( 0.1% etoh ) were performed by radioimmunoassay according to kit instructions . camp - specific pde activity assays29 and western blots for pde4a530 were performed as previously described . full methods and any associated references are available in the online version of the paper at www.nature.com/nature .

millions of people regularly obtain insufficient sleep1 . given the impact of sleep deprivation on our lives , understanding the cellular and molecular pathways affected by sleep deprivation is clearly of social and clinical importance . one of the major effects of sleep deprivation on the brain is to produce memory deficits in learning paradigms that are dependent on the hippocampus25 . in this study , we have identified a molecular mechanism by which brief sleep deprivation alters hippocampal function . sleep deprivation selectively impaired camp / pka - dependent forms of synaptic plasticity6 in the hippocampus , reduced camp signaling , and increased activity and protein levels of phosphodiesterase-4 ( pde4 ) , an enzyme that degrades camp . treatment with pde inhibitors rescued the sleep deprivation - induced deficits in camp signaling , synaptic plasticity , and hippocampus - dependent memory . these findings demonstrate that brief sleep deprivation disrupts hippocampal function by interfering with camp signaling through increased pde4 activity . thus drugs that enhance camp signaling may provide a novel therapeutic approach to counteract the cognitive effects of sleep deprivation .

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the treatment of patients with multiple trauma requires a different approach to that of patients with regular trauma because they are threatened not only by the injuries themselves , but also by the metabolic disruptions that follow.1 delay in surgery , blunt trauma , extensive soft tissue damage , and combined orthopedic and vascular injuries have been associated with an increased risk of amputation , while associated nerve and bone injuries with extensive soft tissue damage are risk factors for a poor outcome.2 acute renal failure is the main cause of death in patients with war wounds and trauma of the extremities . it would be helpful to minimize mortality in these patients by managing shock in a timely manner and taking the decision to amputate appropriately and promptly.3 severe head injury is known to be a major determinant of mortality in patients with multiple injuries , but other injuries also contribute to the clinical outcome.4,5 different mechanisms of injury , such as motor vehicle crashes , falls , or pedestrians being struck by a motor vehicle , impart varying degrees of force and energy transfer that may impact outcomes ; this was found to predict mortality and functional impairment independently at hospital discharge.6 acute lower extremity compartment syndrome is a devastating complication that often presents silently in critically injured patients.7 patients who underwent delayed fasciotomy had twice the rate of major amputation and a three - fold higher mortality rate.8 the aim of this study was to evaluate if injuries of the extremities are associated with a higher one - month mortality rate than other types of associated trauma . this prospective , observational , cohort study was carried out in the regional emergency center of hospital de base after prior approval by the ethics research committee of the so jos do rio preto medical school . the emergency department follows a systematic pathway to provide initial assistance to accident victims using the atls ( advanced trauma life support ) protocol . all live accident victims treated in the emergency department from july 2004 to june 2005 were included . patients who were dead on arrival and not submitted to any type of inhospital resuscitation procedure were not included in the study . accident victims were allocated to two groups , ie , those with severe injuries to the extremities or pelvis ( abbreviated injury scale [ ais ] 34 ) and those without injuries or with minor injuries to the extremities ( ais 02 ) . the fisher s exact test and relative risk were used for statistical analysis , and an alpha error of 5% ( p 0.05 ) was considered statistically significant . a total of 3489 patients were evaluated in this study ; 3244 ( 92.98% ) did not have severe trauma of the extremities ( ais 02 ) , 34 ( 1.05% ) of whom died . severe injuries of the extremities ( ais 34 ) occurred in 245 ( 7.02% ) of the patients , with 13 ( 5.31% ) dying ( fisher s exact test : p = 0.001 , relative risk 5.063 , 95% confidence interval [ ci ] : 2.7079.467 , table 1 ) . table 2 shows the age , type of injury sustained , and time until death after trauma for the patients who died . of the 245 patients with ais 34 , 71 ( 28.98% ) were women and 174 ( 71.02% ) were men , with the mean age of the men being 40.1 20.5 years and of the women 60.2 23.6 years . this study assessed whether severe injuries of the extremities affect the overall mortality rate in accident victims . the death rate was found to be higher for patients with ais 34 than in those without injuries or with minor injuries to the limbs . thus , these data serve as a warning in respect to increased risk of death in orthopedic patients compared with general trauma patients . when the severity of specific injuries in accident victims is reported in the literature , head trauma is cited as one of the main causes of death.5,6 however , there are few data in the literature about the association between death and injuries to the extremities . one study reported that the mortality rate in accident victims with extremity injuries was higher in pedestrians struck by motor vehicles ( 20% ) , and for those with head injuries , it was higher for motor cycle crash victims ( 16%).6 the first phase of management for these patients aims to control bleeding , by surgical intervention if necessary , and to prevent further wound contamination . the second phase consists of resuscitation in the intensive care unit , and the third phase aims at definitive repair of the injuries sustained.9 pelvic injuries represent a thorny and stubborn therapeutic challenge . rapid diagnosis and effective treatment ( damage control ) of these injuries play a key role in the patient s survival , inasmuch as the mortality of multiply injured patients with pelvic ring disruption remains high ( 20%35%).10 the preclinical management of patients with multiple trauma influences the prognosis regarding mortality and morbidity . diagnostic overview , protection of vital functions in the special circumstance of shock , immobilization of the spine , and treatment of isolated injuries are an essential part of preclinical management.11 the type of trauma is known to influence the mortality rate.12 in this study , all the patients received specialized pre - hospital assistance , and were treated in a regional trauma reference center . in spite of all the care given at the scene of the accident through to discharge from hospital , trauma of the extremities was a significant cause of death . accident victims with injuries of the extremities are at higher risk for death than those with other types of trauma .

background : the aim of this study was to evaluate one - month hospital mortality in victims with injuries of the extremities.methods:all accident victims admitted to the hospital de base in so jos do rio preto , brazil , during the period from july 2004 to june 2005 , were evaluated in an observational study . patients were classified using the abbreviated injury scale ( ais ) . patients with severe injuries of the extremities ( ais 34 ) were compared with those without injuries or with minor extremity injuries ( ais 02).results : a total of 3489 accident victims were evaluated ; 3244 ( 92.98% ) did not suffer injuries or had minor injuries of the extremities ( ais 02 ) and 245 ( 7.02% ) had severe injuries ( ais 34 ) . of the 245 patients with ais 34 extremity injuries , 13 ( 5.31% ) patients died , and of those without severe injuries to the extremities , 34 ( 1.05% ) died ( fisher s exact test p = 0.0000 , relative risk 5.063 , 95% confidence interval [ ci ] : 2.7079.467).conclusion : patients with injuries of the extremities are at greater risk of death than accident victims with other types of trauma .

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antiphospholipid syndrome ( aps ) is an autoimmune disease characterized by thrombosis and morbidity , specifically in pregnancy , due to antiphospholipid antibodies . about half of the cases of aps occur as a primary disorder , while the rest arise in association with other autoimmune diseases , such as systemic lupus erythematosus ( sle ) . some diseases , such as pulmonary thrombosis and pulmonary hypertension , are known to be complicated by aps ; however , aps with pleural effusion is extremely rare . here , we present a case of aps complicated by unilateral pleural effusion that responded well to oral corticosteroid therapy . a 75-year - old japanese man was admitted to our hospital for spreading erythema on his trunk and extremities , as well as dyspnea . one year prior to admission , he visited us with a 1-year history of erythema and purpura on his legs , accompanied by intermittent fever . results of laboratory examinations for antiphospholipid antibodies , lupus anticoagulant ( using the phospholipid neutralization test ) , and anticardiolipin antibody had been positive 12 weeks apart . in addition , he showed positive antinuclear antibody ( 1:80 , homogeneous pattern ) , but was negative for anti - dsdna antibody , anti - sm antibody , anti - rnp antibody , anti - ss - a antibody , anti - ss - b antibody , antitopoisomerase i antibody , and anticentromere antibody . mpo - anca , pr3-anca , and cryoglobulin were negative . given the diagnosis of aps , we initiated combination therapy with aspirin ( 100 mg daily ) and warfarin ( target international normalized ratio , 2.03.0 ) , but the skin lesions continued to gradually worsen . violaceous erythema , purpura , and pigmentation were widely noted on his trunk and extremities ( fig . 1 ) ; they were associated with low platelets ( 93,000/l ) and elevated partial thromboplastin time ( 48.4 s ) . a biopsy specimen revealed marked thrombosis in the dermal and subcutaneous small vessels ( fig . 2 ) . there were interface changes of the dermo - epidermal junction and mild inflammatory infiltrates in the perivascular area of the dermis , but mucin deposition and thickening of the basal layer of the epidermis were not apparent . in addition , a chest x - ray and computed tomography demonstrated a large pleural effusion in the left lung ( fig . 3 ) , without evidence of large vessel thrombus . electrocardiogram and echocardiogram were normal . despite serial thoracenteses , effusion recurred . bacterial and fungal cultures , as well as cytology analyses for malignant cells , were all negative . after excluding infectious diseases , malignancies , pulmonary thrombosis , and heart failure , we added oral prednisolone ( 30 mg daily ) to his prior anticoagulant regimen . the skin lesions and the pleural effusion improved rapidly , eventually disappearing without complication ( fig . 4 ) . on follow - up clinical examinations , no symptoms related to sle or other collagen diseases were noted . common causes of pleural effusion include malignancies , infectious diseases , pulmonary embolism , collagen vascular disease , and heart failure . aps - related pleural effusion has rarely been reported , and those cases that have been reported appeared to be complications of accompanying pulmonary embolism , sle , or catastrophic aps [ 4 , 5 , 6 ] . pleuritis , which can induce pleural effusion , is the most common pleuropulmonary manifestation of sle . in the present case , after excluding these differential diagnoses , aps was determined to be the direct cause of the pleural effusion . however , a strong possibility still exists that the pleural effusion may be associated with occult collagen vascular disease , particularly sle or lupus - like disease ( lld ) heretofore undiagnosed . a long - term follow - up study in 128 patients with primary aps demonstrated that 11 patients ( 8% ) developed sle , while 6 ( 5% ) developed lld during a median follow - up period of 8.2 years ( range , 114 years ) . the results of this study suggest that the pleural effusion may be attributed to a coexisting condition like lld , although our patient has not fulfilled american college of rheumatology diagnostic sle criteria to date . this may be supported by the fact that oral corticosteroid therapy was a remarkably effective treatment of the pleural effusion that had previously been unsuccessfully treated by anticoagulant therapy and repeated drainage . corticosteroids and immunosuppressants continue to be the treatment of choice for severe sle complications , including pleural effusion . furthermore , the clinical manifestations of primary aps and aps associated with sle are similar , which makes it more difficult to differentiate these diseases . as pleural effusion can be life - threatening corticosteroids might be an effective choice of treatment for intractable pleural effusion in aps patients .

antiphospholipid syndrome ( aps ) with pleural effusion is extremely rare . a 75-year - old man was admitted to our hospital for spreading erythema on his trunk and extremities , as well as dyspnea . one year before admission , he had visited us with a 1-year history of erythema and purpura on his legs and occasional fever . given the diagnosis of aps , we initiated a combination therapy of aspirin and warfarin , but the skin lesions had gradually worsened . a biopsy specimen revealed marked thrombosis in the dermal and subcutaneous small vessels . in addition , chest x - ray and computed tomography demonstrated a large pleural effusion in the left lung . he underwent repeated drainage of the pleural effusion but the effusion recurred . we added oral prednisolone 30 mg daily to his prior anticoagulant therapy . the skin lesions and pleural effusion rapidly improved and disappeared without any complication . corticosteroids might be a choice of treatment for intractable pleural effusion in aps patients .

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in this issue of critical care , laporta and coworkers review a multidisciplinary working group 's analysis of a case study on the causes of and solutions for staff turnover in an intensive care unit ( icu ) setting . this issue is of profound significance to health care leaders in western countries because the workforce is shrinking as a result of impending baby boomer retirements and , as the population ages , the demand for intensive care services will grow considerably . these demographic factors are further compounded by the fact that the complexity of care provided in the icu demands professionals who are highly trained and skilled . in this environment , turnover can be costly to the organization because of the significant expenses associated with recruiting and training workers . there are many well documented reasons for staff turnover in the intensive care setting that are highlighted by laporta and coworkers as core reasons . these core reasons include job dissatisfaction due to inflexible scheduling practises , insufficient opportunity for professional development , as well as a lack of collaborative decision making around clinical and practice issues . the authors discuss that data on icu turnover comes from nursing literature and that this research may be applicable to other health care professionals . however , it is important not to assume that reasons for turnover are the same among different groups of health care providers and that staff turnover is something to be avoided at all costs . for example , misra - hebert and coworkers state that one contributor to physician turnover is conflict between the physician 's and organization 's philosophy and goals . physician turnover in this case may be beneficial both to the physician and organization if the two parties can not reconcile their differences and the conflict impacts on the ability of both parties to move forward . there are other important reasons for turnover that should be considered by icu leaders , and these include burnout and generational diversity . burnout is a prevalent phenomenon in icus , and the nursing literature suggests that issues such as moral distress when engaging in futile care contributes to burnout . in the medical literature causes of physician burnout include volume of work , increased expectations of the public , lack of sleep and the possibility of being sued . the consequence of burnout is that there is a negative impact on quality of care and staff morale , which can ultimately cause turnover . for example , gunderson indicates that physicians who are dissatisfied may engage in inappropriate prescribing patterns . neuhauser , furthermore , discusses how environments with rigid systems and attitudes among the leadership will decrease staff morale because staff desire flexible policies and autonomy in decision - making . the generational diversity found in the icu environment can also be a source of turnover of staff . it is well documented that generation x ( born in 19651980 ) and the millennial generation ( born in 19802000 ) have a strong desire for more balanced work life than veterans ( born in 19251945 ) and baby boomers ( born in 19461964 ) . research conducted by lorin and coworkers on internal medicine residents of the millenial generation showed that although 41% considered a fellowship in critical care , only 3.4% chose this training because of lack of leisure time and stress levels among faculty and fellows . clearly , it is important for leaders to be attuned to these generational differences when developing recruitment and retention plans and redesigning the workplace environment . the review from laporta and coworkers also highlights the importance of icu leadership working with frontline staff to create a vision and strategy that addresses the core reasons for turnover . it is essential that this vision be aligned with the vision , mission and values , and strategic plan of the health care organization . furthermore , the team should assess whether their hospital is highly reputable , has high patient satisfaction , and sufficient resources and equipment to provide care . all of these components are signs of a positive work environment , and leadership can build on these attributes to recruit and retain staff . the other key factor in this process is the use of a team work approach . team work training in the areas of conflict resolution , learning styles and giving feedback will help the staff to work together to create and achieve an inspiring vision . although the financial and human resource investments required to engage in this process are considerable , there is substantial evidence in the literature that highly functioning , satisfied teams lead to more efficient patient care and better outcomes . staff turnover is a critical issue that icu leaders need to understand and address in their unit settings . attention to this issue with a systematic , evidence - based approach that focuses on team work and collaboration will not only improve retention but will also make the icu a highly competitive and desirable place to work .

this commentary discusses laporta and coworkers analysis of a case study on the causes of and solutions for staff turnover in an intensive care setting . staff turnover is a significant issue for health care leaders due to the shrinking workforce in western countries and an increased demand for intensive care services as the population ages . the commentary considers reasons for turnover such as burnout and generational diversity , and highlights the importance of a team work approach to address the issue of turnover .

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implant - supported overdenture prostheses can be divided into bar overdentures and single attachment overdentures . single attachment elements for overdentures include single retentive anchors , single magnet anchors , and individually cast telescopic copings.1 among these , telescopic copings have the benefit of implant splinting found in bar overdentures and the retrievability of single attachment overdentures . however , this method is typically fabricated using gold materials , so it is not an economical treatment option . also , if an inner crown will be worn , it is difficult to maintain appropriate retentive forces . in this case report , a telescopic implant - supported overdenture prosthesis was made using a new material , polyaryletherketone ( paek ) based polymer ( pekkton ivory , cendres + mtaux sa , biel / bienne , switzerland ) . it shares benefits of typical telescopic coping , in additional to being highly economical , wear resistant , and light in weight compared to conventional implant overdenture prostheses . 1 ) presented to the department of prosthodontics at chonnam national university dental hospital . after clinical and radiographic examinations after maxillary teeth extractions and use of provisional maxillary complete denture for six months , six small diameter implant fixtures ( 3.0 10.0 mm usii , osstem implant co. ltd . , seoul , korea ) the definitive prosthesis was planned as a telescopic overdenture using paek based polymer . after a making definitive impression by polyvinylsiloxane ( honigum , dmg , hamburg , germany ) , a polymer telescopic abutment and an outer overdenture frame were fabricated with consideration of the patient 's vertical dimension ( fig . telescopic abutment and framework design were laid out by cad software ( exocad dental cad , exocad gmbh , darmstadt , germany ) . the milling machine ( s1 , vhf camfacture ag , ammerbuch , germany ) made the final framework and abutment according to the design . polymer abutment and titanium link were sandblasted by 110 um grit aluminum oxide , and bonded with primer ( sr link , ivoclar vivadent , schaan , liechtenstein ) and bonding agent ( multilink n , ivoclar vivadent , schaan , leichtenstein ) . after that , the definitive prosthesis was made by autopolymerized pour - type resin ( press lt , retec , rosbach , germany ) ( fig . 4 ) ; the design and weight of the prosthesis were adjusted to achieve acceptable esthetics and phonetics . after 6 months , there were no problems with alveolar bone around the implant fixtures and retention of the overdenture prosthesis . however , no treatment modality meets all criteria for successful treatment , and conventional overdenture material can sometimes be limited by economic , functional , and technical considerations . now , many new prosthetic materials are available to overcome these limitations , and as in this case , a new polymer can be used to make telescopic crowns and frameworks to obtain satisfactory results . paek based polymer , pekkton ivory , as used in this case , is a member of the high performance semi - crystalline thermoplastic resin group , recognized for its keto and ether group ratio . paek has good dimensional stability at high temperature , high chemical and mechanical resistance against wear , and high tensile , fatigue and flexural strength , making it an attractive material with expanded uses in medicine and dentistry.3 however , peek ( polyetheretherketone ) , a conventional paek - based polymer , can not be used as a permanent material due to its relatively weak physical properties . a new material , pekkton , is mainly composed of pekk ( polyetherketoneketone ) ; its molecular structure has an added ketone to the structure of peek with and has a wide range of uses due to its amorphous and crystalline structure . pekk reveals up to 80% greater compressive strengths than peek , so this polymer may be used in permanent prostheses according to the manufacturer.4 thus , this new polymer can be considered to have greater strength than peek and have greater esthetics titanium , higher resin bond strength than zirconia , and a lighter weight ( 1.4 g / cm ) than metal . therefore , this material is found to be mechanically suitable for fpd frameworks , milled overdenture bars , clasps , telescopic crowns , and other applications . despite the reportedly good bond strength , bonding between the titanium link and the telescopic abutment is still very sensitive , with the risk of fracture of the thin abutment wall due to connecting titanium link ; hence more studies are needed on such cases . she reported satisfaction with its strength and esthetics , and no negative symptoms . due to the lack of evidence on the long term retentive capabilities of this material , due to its functional and economic advantages , paek based polymer is a good alternative material to conventional materials and methods in the fabrication of implant overdenture .

although many prosthetic materials exist for fabrication of implant - supported telescopic overdentures , available materials have not been thoroughly evaluated from a functional standpoint . this case report describes the use of polyaryletherketone ( paek ) based polymer for an implant - supported telescopic overdenture , a seldom used material in dentistry . this material is lighter than traditional materials , can accommodate changes in retentive forces , and is an easily retrievable by cad / cam fabrication . this case highlights the possibility of using new polymer materials for implant - supported telescopic overdentures .

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xanthogranulomatous pyelonephritis ( xgp ) is a rare , distinct and aggressive form of chronic infectious pyelonephritis . it accounts for lesser than 1% of chronic pyelonephritis . though common in fifth to sixth decade complications can occur in the form of psoas abscess , nephro cutaneous fistula , enterocolonic fistula , paranephric abscess and sepsis . it is essential to suspect and diagnose this condition early to prevent the morbidity and mortality . owing to its rarity and clinical curiosity a 75-year - old man presented with difficulty in micturition since 15 days , fever , abdominal and flank pain and burning micturition since 7 days . x - ray , ultrasonography ( usg ) and computed tomography ( ct ) scan abdomen findings include pyonephrosis and cortical atrophy in the right kidney . intravenous urography ( ivu ) revealed non - excretory right kidney . left kidney showed normal excretion resected specimen was yellowish lobulated renal mass measuring 13 8 6 cm with ureter [ figure 1a ] . cut section showed dilated pelvis , calyces and cortical atrophy due to extensive destruction of renal parenchyma , which were covered with thick purulent material [ figure 1b ] . histopathology revealed atrophic and dilated renal tubules showing thyroidisation and sclerosed glomeruli and interstitial fibrosis [ figure 2 ] . many areas showed histiocytes with abundant foamy cytoplasm , lymphoplasmacytic inflammatory cells with foci of polymorph nuclear leukocytes [ figure 3 and inset ] ziehl nelsen stain was negative for acid fast bacilli . ( a ) yellowish lobulated renal mass with ureter ; ( b ) dilated pelvis , calyces and cortical atrophy covered with thick purulent material atrophic and dilated renal tubules showing thyroidisation , sclerosed glomeruli and fibrosis . inset show giant cells and cholesterol clefts ( h and e , 400 ) foamy histiocytes , inflammatory cells . xgp is a severe chronic renal inflammatory condition leading to focal or diffuse kidney destruction . it may be due to defect in degradation of bacteria in the macrophages especially when associated with infection and obstruction by stones . three forms of xgp are recognised : diffuse is characterized by diffuse involvement of kidney , segmental by segmental involvement and focal - is located within the cortex . it is often misdiagnosed pre - operatively as pyelonephritis , tuberculosis , perinephric abscess and renal cell carcinoma ( rcc ) . symptoms include flank or abdominal pain , fever , palpable mass , gross hematuria , pyuria , dysuria and weight loss . in our case , symptoms were attributed to bph . other organisms include staphylococcus aureus , group b streptococcus , candida , klebsiella and bacteroides . elevation of serum creatinine and bun in our case can be attributed to impaired renal function . non - functioning , ct scan is the main stay of diagnostic imaging for xgp . xgp has been shown to be associated with transitional cell carcinoma of renal pelvis and rcc . they have even been reported in renal allograph . in diffuse or advanced stage xgp , nephrectomy is the treatment option . focal or segmental xgp if diagnosed early pre - operatively can be treated with antibiotics . pre- and post - operative broad spectrum antibiotics and symptomatic management are also key factors for successful management and better prognosis . we presented this unusual case of elderly male to stress the importance of through evaluation of renal function who gave a history of recurrent urinary tract obstruction and infection . chronic renal infection and obstruction are two common etiological factors for xgp . in all patients of prostatic enlargement , renal function must be assessed for the extent of damage . in non - functioning kidney

xanthogranulomatous pyelonephritis ( xgp ) is rare and aggressive form of chronic infectious pyelonephritis . no single clinical or radiological feature is diagnostic of xgp . a 75-year - old man with prostatic enlargement presented with difficulty and burning micturition fever , abdominal and flank pain . x - ray , ultrasonography and computed tomography scan diagnosis was right kidney pyonephrosis . intravenous urography revealed non - excretory right kidney . right nephrectomy was done . histological diagnosis of xgp was made . in all patients of prostatic enlargement , renal function must be assessed for the extent of damage . surgery is the treatment choice in most cases . pre- and post - operative antibiotics are key factors for successful management and better prognosis .

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the incidence of bladder forming a part of an inguinal hernia is 14% . with correct knowledge of anatomy and careful dissection , injuries to the bladder during hernia repair we hereby report a case where the patient first presented with a scrotal abscess and vesicocutaneous fistula after surgical repair of sliding hernia . a 65-year - old morbidly obese man underwent mesh hernioplasty for large right inguinal hernia . after removing the urethral catheter , he developed gradually increasing right scrotal swelling with fever . thereafter , he developed continuous urine leakage from the site of incision and drainage , figure 1 . we carried out a cystogram via the urethral catheter that revealed a fistulous communication between bladder and scrotal skin , figure 1 . , cystoscopy revealed normal anterior and posterior urethra , non - obstructing prostatic lobes and a defect in the anterior bladder wall with no evidence of mesh erosion . almost the whole of the bladder was lying in the right scrotum and densely adherent to the right testis and cord structures and mesh . there was a fistulous opening at the dome of the bladder wall well away from the mesh . our main concerns were inguinal hernia repair and creation of extraperitoneal space to reposition the bladder in the normal position , which was not possible without performing right high inguinal orchiectomy . hence , we performed right high inguinal orchiectomy and removal of mesh and extraperitoneal space was made to reposition the urinary bladder to its normal position . fistula opening was repaired in two layers and the bladder was put on continuous drainage via 20 french urethral catheter , figure 2 . post - operatively at 2 weeks , there was no urinary leak on cystogram and the urethral catheter was removed and normal voiding was restored . scars of previous surgery with vesicocutaneous fistula and cystogram showing contrast in the left hemiscrotum the entire urinary bladder lying in the scrotum , with the bladder re - positioned into the normal position levine coined the term scrotal cystocoele in 1951 for inguinoscrotal herniation of the bladder . urinary bladder herniations are usually diagnosed at the time of inguinal herniorraphy and are commonly repaired through the same incision . they are sometimes found incidentally during the evaluation of a patient with lower urinary tract symptoms and associated inguinal hernias . two - stage micturition is the classical symptom , with the second stage facilitated by some form of external pressure on the bladder . the para - peritoneal type is the most common type and the extra - peritoneal type is the least common . because imaging all patients with large hernias may not be cost - effective , imaging studies are performed only when bladder herniation is suspected . the diagnostic triad of lateral displacement of the distal one - third of the ureter , small asymmetric bladder and incomplete visualization of the bladder base on an intravenous urogram has been described by reardon and lowman . iatrogenic injury to the bladder during hernia repair can be due to multiple factors , such as an inexperienced surgeon in the early part of the learning curve or an obese patient with large hernial sac with unrecognized bladder component . in our patient , there could have been an injury to the bladder that was not recognized at the time of hernia repair , which led to subsequent scrotal abscess formation resulting in a vesicocutaneous fistula . if unrecognized , these usually present immediately after catheter removal , but presentation can sometimes be delayed in case the fistula is very small and there is no infravesical obstruction . management includes immediate repair in case it is recognized intraoperatively . in case of unrecognized injury and with delayed presentation , the first step is to put a wide caliber per urethral catheter followed by thorough evaluation with urine culture examination and cystogram . a small fistula can be healed with only continuous bladder drainage with per urethral catheter or preferably suprapubic cystostomy , provided lower tract infravesical obstruction has been ruled out . a larger fistula needs open surgical management . careful dissection is needed in the extraperitoneal space while separating the sac from cord structures as the bladder forms a part of the posterior wall of the sac . after completing bladder dissection , the fistula is repaired in two to three layers and an adequate space is created in the extraperitoneal plane to reposition the bladder . sometimes , large hernias could be treated by resection of the herniated bladder as described by thomas and gomella . in our case , the whole bladder was lying in the scrotum and extensive adhesions were present between the cord structures and the bladder . high inguinal orchiectomy was performed to create space for the bladder and for proper closure of the inguinal canal . urinary bladder rarely forms a part of an inguinal hernia and , with the correct knowledge of anatomy and careful dissection injuries to the bladder during hernia repair , surgery can be prevented . surgically creation of an extraperitoneal space for bladder repositioning is of paramount importance , sometimes needing inguinal orchiectomy in the elderly .

sliding inguinal hernias are usually direct inguinal hernias containing various abdominal viscera . the incidence of bladder forming a part of an inguinal hernia , called as scrotal cystocele , is 14% . the risk of bladder injury is as high as 12% when repairing this type of hernia . this case report emphasizes this aspect in a 65-year - old man who presented with urinary leak through the scrotal wound following right inguinal hernia repair .

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though limited in efficacy in many cases , the control methods available today represent a major progress when compared to the lack of any means for the control of these plants one or two decades ago . crops can be protected by resistance , by selective fungicides , by biocontrol agents , and by cultural methods that did not existed before . the current focus in applied breeding is leveraging biotechnological tools to develop more and better markers to allow marker assisted selection with the hope that this will speed up the delivery of improved cultivars to the farmer . to date , however , progress in marker development and delivery of useful markers has been slow in legumes . we are now also facing an accelerated progress in the genomic and biotechnological research , which should soon provide important understanding of some crucial developmental mechanisms in both the parasites and their host plants and will provide candidate genes for resistance to ascochyta blight . the application of ngs technologies will provide a new research framework and molecular tools to be applied in resistance to ascochyta blight in legumes . the authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest .

legume cultivation is strongly hampered by the occurrence of ascochyta blights . strategies of control have been developed but only marginal successes achieved . breeding for disease resistance is regarded the most cost efficient method of control . significant genetic variation for disease resistance exists in most legume crops with numerous germplasm lines maintained , providing an excellent resource for plant breeders . fast and reliable screening methods have been adjusted to fulfill breeding program needs . however , the complex inheritance controlled quantitatively by multiple genes , has been difficult to manipulate . successful application of biotechnology to ascochyta blight resistance breeding in legume crops will facilitate a good biological knowledge both of the crops pathogen interaction and of the mechanisms underlying resistance . the current focus in applied breeding is leveraging biotechnological tools to develop more and better markers to speed up the delivery of improved cultivars to the farmer . to date , however , progress in marker development and delivery of useful markers has been slow in most legumes . the limited saturation of the genomic regions bearing putative qtls in legume crops makes difficult to identify the most tightly linked markers and to determine the accurate position of qtls . the application of next generation sequencing technologies will contribute to the development of new markers and the identification of candidate genes for ascochyta blight resistance .

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proteus syndrome ( ps ) is a rare and sporadic disorder that causes postnatal overgrowth of tissues in a mosaic pattern . the complications of ps include , progressive skeletal deformities , invasive lipomas , benign and malignant tumors , and deep venous thrombosis with pulmonary embolism . we report a rare case of ps that presented with hypertrophy of index and middle finger without any other abnormalities or complications . incidentally we noticed that he had enlarged index and middle fingers of both hands and thumb of right hand [ figure 1 ] . on probing patient revealed that it was present since childhood with onset around the age of 5 years and gradual progression over years to the present size . no similar tissue growth in other parts of the body and there was no one in the family with similar features . on examination hypertrophy of index and middle finger of both the hand ( a , b , c ) and thumb of the right hand ( c ) laboratory investigations revealed normal renal and liver function tests . his x - ray of hands showed hyperostosis of involved fingers [ figure 2 ] . x - ray hands showing hyperostosis of both index and middle fingure ( a , b ) and thumb of right hand ( b ) proteus who had the ability to change his shape and was proposed by wiedemann , et al . in 1983 . happle , et al . in 1987 hypothesized that the syndrome might be due to somatic alteration of a gene leading to mosaic effects that would be lethal if the mutation were carried in nonmosaic fashion . the dysregulated tissue growth in mosaic pattern results in various phenotypic presentations and hence the clinical manifestations of ps are highly variable . the tissue overgrowth is usually absent or mild at birth and progressive in nature but usually appears to plateau after adolescence . the disproportionate overgrowth of tissue is usually asymmetrical and involves the arms , legs , hands , feet , and digits . characteristic manifestations include hyperostoses , often near epiphyses with associated impaired mobility and cerebriform connective tissue nevus seen most commonly on plantar surface . other findings are lipomas , epidermal nevi and capillary vascular malformations [ table 1 ] . criteria for the diagnosis of proteus syndrome there is no specific molecular marker , or laboratory test , for the diagnosis of ps . the diagnosis is mainly based on history , clinical examination and imaging studies . because of its variable presentation , ps may be confused with other conditions . the two disorders most commonly confused with ps are klippel - trenaunay syndrome and hemihyperplasia / lipomatosis syndrome . the important points in ps that help in the differential diagnosis are : differential diagnosis of proteus syndrome sporadic and progressive nature of tissue overgrowththe absent or mild tissue growth at birthabsence of bone tumor , enchondromasabsence of specific gene mutations differentiates from neurofibromatosisabsence of familial inheritance ( postzygotic somatic mutation of genes ) sporadic and progressive nature of tissue overgrowth the absent or mild tissue growth at birth absence of bone tumor , enchondromas absence of specific gene mutations differentiates from neurofibromatosis absence of familial inheritance ( postzygotic somatic mutation of genes ) there are no effective treatment modalities for ps . the patients should be followed up regularly for development of complications and their management . the management is also challenging because of progressive nature of tissue growth . both benign and malignant two relatively common tumors include cystadenomas of the ovary and monomorphic adenomas of the parotid gland . presented with macrodactyly of index and middle finger of both the hands and thumb of right hand ( one criteria of category b ) and he met all the three general criteria . his skeletal survey was normal except for hyperostosis of index and middle finger and there were no associated complications except for the limitation of his affected finger movements . although the patient did not satisfy the proposed criteria [ table 1 ] , a literature search revealed that out of the 205 cases reported 90 satisfied the criteria highlighting the variability in clinical presentation in cases of ps . our patient was managed as a case of inferior wall mi and is presently on anti - ischemic medications with no new complications . in conclusion , ps is a very rare and highly variable , progressive tissue overgrowth disorder . patients should be kept under regular follow - up for the development of complications and their management .

proteus syndrome ( ps ) is a rare hamartomatous disorder characterized by various cutaneous and subcutaneous lesions , including vascular malformations , lipomas , hyperpigmentation , and several types of nevi . partial gigantism with limb or digital overgrowth is pathognomonic of ps . we report a rare case of ps in a 50-year - old man who presented with inferior wall myocardial infarction and was incidentally detected to have hypertrophy of index and middle fingers of both the hands .

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craniopharyngioma is an uncommon tumor of the nervous system ; it is well - known to recur even several years after surgery . we are here with reporting a case of craniopharyngioma which recurred at a site removed from the original site 5 years after surgery and radiotherapy . a 4-year - old girl was admitted for progressive deterioration of vision of 2 months duration . in addition there was no history of endocrinopathy , fits or any symptom of raised intracranial pressure . on examination , visual acuity was questionable perception of light on the right side and counting fingers at 3 m distance on the left . imaging revealed a solid and cystic craniopharyngioma in the sellar - suprasellar region [ figure 1a and b ] . she underwent a right frontotemporal craniotomy and transsylvian exploration and almost total excision of the tumor . postoperative mr showed a tiny residual tumor adherent to the pons [ figure 2a and b ] . ( a and b ) showing the preoperative images ( before first surgery ) ( a and b ) images after first surgery showing no residual tumour in the primary site but a small fragment adherent to the pons she was given a course of radiotherapy for this residue 54 gy in 30 fractions . follow - up imaging at the end of 2 years did not reveal any residual tumor [ figure 3 ] . two years after surgery and radiotherapy no recurrence imaging was being done periodically to check for recurrence . the 5-year surveillance imaging showed a recurrence in the right sylvian fissure along the route taken during the first surgery . there was no evidence of tumor in the sellar - suprasellar area [ figure 4a and b ] . she underwent reexploration by the same route , and a tiny fragment densely adherent to the middle cerebral artery was left behind . ( a and b ) showing remote recurrence five years after first surgery and radiotherapy . although craniopharyngiomas are benign tumors they are known to recur even after years and even after the administration of radiotherapy , recurrence rates ranging from 25% to 70% . recurrences at a site removed from the original site are very rare < 25 cases have been reported . these ectopic recurrences are not to be misinterpreted as ectopic primary occurrences since craniopharyngioma can occur anywhere along the obliterated rathke 's pouch . these ectopic recurrences may occur along the surgical pathway or at a site , not along the surgical pathway . the cells of the tumor may get implanted and may subsequently metamorphose into a fresh neoplasm . these tumor cells may in turn give rise to the regrowth of the tumor . the usual time to recurrence is around 4 years . but why this has to be a peculiarity of craniopharyngiomas can not be explained . another way the tumor may get seeded at a distant site the evidence for this is strong since tumor cells have been observed in the csf . although most recurrences are along the surgical corridor an instance where the recurrence has occurred in the spine has been recorded . when the transsphenoidal route is used , the csf spaces are not violated this may explain the absence of recurrences after transsphenoidal route . it is not surprising to observe that the histologic examination of the primary and recurrent lesions are the same . recurrences rates are said to be low after total surgical excision . but recurrences even decades after a quiescent period are well - known . it can be assumed that radiotherapy would have sterilized the surgical corridor and ectopic recurrences will not occur . but this was not the case in our patient , and in the few that have been reported . total excision is not an assurance that recurrences at ectopic or primary site will not occur . certain measures have been proposed to minimize these ectopic recurrences , protecting the operative field with patties to prevent seeding , emptying the cyst prior to removal of tumor , thorough irrigation of the field before dural closure . probably , a higher mib-1 index and expression of p53 may predispose to these recurrences . but eternal vigilance and regular imaging are mandatory to detect recurrences . another point worthy of note is why this phenomenon is not seen with respect to other benign tumors like meningiomas or even malignant tumors .

the aim was to present a rare case of recurrent craniopharyngioma remote from the primary site of origin . a young girl was operated for sellar region craniopharyngioma . for a small residual tumor , she underwent radiotherapy . follow - up imaging did not reveal any residual tumor or recurrence . surveillance magnetic resonance imaging after 5 years revealed a recurrence in the right sylvian fissure . this tumor was totally excised . recurrence of craniopharyngioma is well - known , but recurrence at a site remote from the original site after radiotherapy is extremely rare . one such case is being presented .

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mucocele of the appendix ( collection of mucus within the appendiceal lumen ) is a rare lesion , found in only 0.2% to 0.3% of 43,000 appendectomies reviewed . currently , the assessment of pelvic masses relies heavily on usg as the primary diagnostic tool . in such cases , clinical findings and other investigative modalities are warranted to aid the diagnostic process . in spite of extensive preoperative investigations , the diagnosis may still remain elusive and may only be made at the time of surgery . some regard this lesion as benign , a result of obstruction of the proximal lumen by fibrosis ; others believe it to be a neoplasm of the appendix . is the method of choice in the management of simple mucocele and for cystadenoma with an intact base . several studies ( mostly case reports ) on laparoscopic resection of mucocele have been reported . a 60-year - old female presented with pain in lower part of abdomen and palpable tender lump in the right ileac fossa . ultrasound of the abdomen reports a cystic mass of size 12 15 cm with thin internal septations in the right adnexa . the pneumoperitoneum was created with veress needle using carbon dioxide and the pressure was kept at 11 mmhg . a 0 telescope was introduced through the umbilical port for the complete examination of the abdomen . diagnostic laparoscopy revealed approximately 14 15 cm large bluish mucocele of the appendix with omental adhesions . two 5-mm ports were placed in the supra pubic area below the pubic hair line as the working port . the mucocele of the appendix was isolated after separating the mesoappendix from it with the help of bipolar cautery . following this , mucocele of the appendix [ figure 1 ] was retrieved out in a plastic bag through the umbilical port . hemostasis was obtained and a suction drain left in situ which was removed when non - productive . cut section showed appendix was filled with mucin - like material [ figure 2 ] . she was started orally after 4 hours of operation and solid food on the next day . appendicular lump from the distal portion of appendix after removal the appendicular lump filled with mucinus material mucocele of the appendix is a descriptive term for an appendix distended by mucus , secondary to mucinous cystadenoma ( 63% ) , mucosal hyperplasia ( 25% ) , mucinous cystadenocarcinoma ( 11% ) , and retention cyst . clinical presentation may include right lower quadrant pain , change in bowel habits , per rectal bleeding , or a palpable mass . approximately 23 - 50% of patients are asymptomatic , with the lesions being discovered incidentally during surgery , radiological evaluations , or endoscopic procedures . the preoperative clinical diagnosis of appendiceal mucoceles can therefore be difficult because of this lack of clinical symptomotology . the initial detection of the lesion may be facilitated by radiological , sonographic , or endoscopic means . on barium enema , the lesion may be seen as a sharply outlined sub - mucosal or extrinsic mass indenting the cecum and laterally displacing it . purely cystic lesions with anechoic fluid , hypoechoic masses with fine internal echoes as well as complex hyperechoic masses can be seen depending on the contents . ct of the abdomen usually shows a cystic well - encapslated mass sometimes with mural calcification , in the expected location of the appendix . it may be causing extrinsic pressure on the cecal wall without any surrounding inflammatory reaction . colonoscopic findings include the volcano sign , the appendiceal orifice seen in the center of a firm mound covered by normal mucosa or a yellowish , lipoma - like submucosal mass . in our case , usg was unable to provide a preoperative diagnosis . in our case , the decision for excision of the appendiceal mucocele was made as a result of diagnostic laparoscopy and a need to rule out malignancy . therefore mucocele of the appendix can mimic an adnexal mass and prove to be a diagnostic challenge . in a woman presenting with right iliac fossa mass and with clinical features not indicative of gynecological pathology , an appendiceal origin should be considered in the differential diagnosis . surgery is the treatment of choice and should be done early as tumor can not be ruled out as the causative factor for the mucocele . pre - operative diagnosis is important to avoid unintended rupture and the development of pseudomyxoma peritonei during surgery . however , laparoscopic dissection , grasping of the appendix specimen , pneumoperitoneum , or transport of the specimen through the abdominal wall might contribute to peritoneal dissemination of a tumor , if present . these setbacks can be avoided by taking precautions like using bowel holding graspers ( non - traumatic ) to handle the mucocele and using a non - permeable bag to deliver the specimen out of the port . mucocele of the appendix can mimic an adnexal mass and prove to be a diagnostic challenge . laparoscopic resection of mucocele of the appendix is feasible in spite of the danger of malignancy , provided necessary precautions are taken .

mucocele of the appendix is an aseptic dilatation secondary to obstruction . the preoperative clinical diagnosis of appendiceal mucoceles can therefore be difficult because of this lack of clinical symptomotology . surgical excision is the treatment of choice in benign mucocele . we report a case presenting to the surgeons where initial clinical findings and investigations suggested a cyst in the right adnexa . diagnostic laparoscopy revealed mucocele of the appendix and laparoscopic appendicectomy was done .

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reactivation of latent tuberculosis ( tb ) is a serious hindrance to continuation of therapy . we present here a case of pleural tb in a patient on infliximab for ankylosing spondylitis . a 36-year - old male presented to our hospital in 2006 with low back ache inflammatory type with symmetric joint pains involving large joints such as shoulder joints , hip joints , knee joints , and small joints such as metacarpophalangeal joints , elbow joints , and metatarsophalangeal joints . there was associated early morning stiffness for over an hour . on examination , synovitis was present in peripheral joints with restriction of movement in all joints . modified schober 's test was positive , and the chest expansion ( <3 cm ) was restricted . on evaluation , he had anemia and elevated erythrocyte sedimentation rate ( esr ) ( 46 mm / h ) and c - reactive protein ( crp ) ( 18 mg / l ) . imaging revealed kyphoscoliosis of thoracic spine , syndesmophytes at multiple levels giving the appearance of bamboo spine [ figure 1 ] . diffuse ossification of interspinous and paraspinal ligaments with fusion of thoracic and lower cervical vertebra was present . radiograph showing bamboo spine in view of hla - b27 , more than 2 spa features and typical radiological features , ankylosing spondylitis was considered . he was started on nonsteroidal anti - inflammatory drugs ( nsaids ) and dose escalated for symptomatic relief . in view of persisting symptoms and elevated esr and crp in spite of optimal nsaids , following three doses of infliximab and 10 months after initiation of therapy , patient came with complaints of fever and cough for 1 week . on examination , breath sounds were reduced over the right side , and there was dullness on percussion . diagnostic thoracocentesis showed lymphocytic ( total leukocyte count - 4800 cells / cumm , lymphocytes - 90% ) exudative type of effusion with high adenosine deaminase ( ada ) ( 114 iu / l ) . according to light 's criteria ( effusion protein - 6 g / dl , serum protein - 3.37 g / dl , effusion lactate dehydrogenase ( ldh ) - 575 ankylosing spondylitis is a chronic , systemic , inflammatory disease that affects primarily the sacroiliac joints and spine . it is a spondyloarthropathy with a prevalence of 0.1%0.4% globally . data from india are sparse . its more commonly seen among males under 30 years of age . diagnosis is made after thorough clinical examination and radiography . infliximab is one such biologic which acts by inhibiting a pro - inflammatory cytokine tnf- and reducing inflammation . target - related adverse effects with tnf inhibitors are infections , opportunistic infections , malignancies , demyelinating conditions , hematologic abnormalities , congestive heart failure , autoantibodies ( antinuclear antibody and anti - double - stranded dna ) , hepatotoxicity , dermatologic reactions , and lupus - like syndromes , whereas the agent - related adverse effects are administration reactions and immunogenicity . tnf- is a cytokine that plays an important role in the mediation of inflammation and immune regulation . they are required for inflammatory response against intracellular organisms . in experimental models , fungal and bacterial infections pneumocystis carinii and histoplasma capsulatum are some of the fungal pathogens , whereas the bacterial agents are listeria monocytogene , mycobacterium tuberculosis , and mycobacterium avium . upper and lower respiratory tract infections are the most commonly seen ones . there was also an increased risk of serious infections compared with controls ( 3.6% vs. 1.7% ) . registries of rheumatoid arthritis patients have shown that the relative risk for infection ( 3.34.1 ) as well as serious infection ( 2.72.8 ) was significantly higher among patients receiving tnf inhibitors . to conclude , severity of disease , use of other medications such as corticosteroids , and the presence of comorbidities also contribute to infections in addition to tnf inhibitors alone . opportunistic infections following tnf inhibitor therapy include disseminated m. tuberculosis . in a study of seventy cases of tb following infliximab therapy , thirty were pulmonary tb and of forty were extrapulmonary disease , only two were cases of pleural tb . around a quarter of the cases were disseminated disease . the majority of cases of tb were observed within a median period of 12 weeks after initiation of therapy and is likely due to reactivation of latent tb . a study by grover et al . in india has shown a high incidence of tb ( 21% ) following biologic therapy . it was also seen that among those who received low doses of infliximab ( 3 mg / kg body weight ) did not develop tb . another study by malaviya et al . has found a lower incidence ( 9.4% ) of tb among those on tnf inhibitors . the rate of development of active tb among rheumatoid arthritis patients on anti - tnf therapy has dropped by 83% with the help of screening . suppressing the action of tnf- can help in relieving the symptoms of ankylosing spondylitis by reducing the inflammatory process , but at the same time , it weakens immune response to microbes such as tubercle bacilli . hence , meticulous screening and close monitoring of patients on infliximab for any symptoms and signs of tb are important as there is a risk even though the screening tests have come out be negative .

we present a case of pleural tuberculosis ( tb ) in a patient on infliximab for ankylosing spondylitis . a 36-year - old male presented to our hospital with low back ache of inflammatory type along with multiple symmetric inflammatory type of joint pain . further clinical examination , laboratory and radiological investigations were suggestive of ankylosing spondylitis . he was initially treated with nonsteroidal anti - inflammatory drugs but citing poor response it was decided to initiate biologic therapy using infliximab ( antitumor necrosis factor - alpha ) . mantoux test and chest radiograph were done before the therapy to rule out tb . following three doses of infliximab , patient came with complaints of fever and cough for 1 week . on investigation , it was found to be a case of pulmonary tb . this shows the importance of close monitoring of patient for tb among patients on infliximab even though the screening test has come out to be negative .

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myoepithelial carcinoma ( mc ) is a rare tumor with an incidence of 0.2% of all salivary gland tumors . most of the reported cases of mc arise in the parotid gland ( 4875% ) , followed by minor salivary glands , and the submandibular gland . the first case was described by higashiyama et al . , in 1998 . since then , only seven cases have been reported in literature . a mentally retarded 13-year - old girl , with a history of congenital hypothyroidism and cystic lymphangioma in the left dorsal region , operated in 2004 , had consulted for cough and dyspnea in september 2010 . physical examination showed a decrease in vesicular breath sounds at the basal areas of chest bilaterally , without fever . pulmonary computed tomography ( ct ) showed bilateral pleural masses measuring 6.3 cm and 7.4 cm at the right and left bases , respectively , with lymph node metastases [ figure 2 ] . ( a ) bilateral pulmonary opacities ( b ) disappearance of pulmonary opacities pulmonary computed tomography ( ct ) : bilateral pulmonary masses a histopathological study of the left pleural biopsy revealed a monomorphic proliferation of round cells with clear cytoplasm and a weak , mitotic hyperchromatic oval nucleus [ figure 3 ] . there were some cohesive layers , separated by bands of sclerosis ; the stroma was sparse with foci of necrosis . the immunohistochemistry ( ihc ) study was focally positive for vimentin , cd99 , and ps100 . the abdominal ultrasound , bone scan , and metaiodobenzylguanidine ( mibg ) scintigraphy were normal . the histopathological and the ihc review at the institut bergoni in france concluded the diagnosis of myoepithelial carcinoma of the soft tissues with intermediate malignancy . ihc was positive for pancytokeratin ( ae1/ae3 ) and ps100 , and negative for ema , cd34 , desmin , and cd99 . the progression was marked by the disappearance of the pulmonary opacities [ figure 1 ] . they include mucoepidermoid carcinoma , adenoid cystic carcinoma , acinic cell carcinoma , oncocytoma , epithelial it arises from the submucosal bronchial glands of the lower respiratory tract . in the world health organization classification , published in 2004 , mc was cited as being synonymous with epithelial myoepithelial carcinoma . as mc and epithelial myoepithelial carcinoma of the salivary gland are distinguished by the presence or absence of ductal cells , their pulmonary counterparts must also be differentiated . myoepithelial tumors are low - grade lesions without recurrences or metastasis described after resection , whereas , the rate of metastasis in mc is high , as seen in our case . our patient represents the first pediatric case , described in the literature , having primitive pulmonary mc . the tumor was peripheral and bilateral , measuring 6 cm and 7 cm , which was in agreement with the literature . in fact , the size of the mc ranged from 15 mm to 130 mm ( mean 50.7 mm ) [ table 1 ] . characteristics of pulmonary mc in our case , the histopathological study oriented to the diagnosis of a primitive neuroectodermal tumor or neuroblastoma . the pathological review , with further ihc analysis in france , had concluded the diagnosis of mc . the following markers were found to be useful in myoepithelial carcinoma : cytokeratins ( ae1/ae3 ) and vimentin ( reported to be positive in neoplastic myoepithelial cells and negative in normal myoepithelial cells ) . other variable markers , such as , ps100 , calponin , smooth muscle actin ( sma ) , muscle - specific actin ( msa ) , smooth muscle myosin , and p63 protein , could be positive . however , neoplastic transformation of myoepithelial cells could result in a loss or a modification of their smooth muscle phenotype . in our case , although the eight patients reported in literature were treated with optimal surgery , metastases were reported in seven of them ( 87.5% ) ( contralateral lung , forearm , liver , and brain ) . it was significant that a patient who had not developed metastasis , had the lowest tumor mitotic rate of 5/10 hpfs . this fact reasoned that the mitotic rate could be an important prognostic factor of the clinical outcome and survival in primary myoepithelial carcinoma of the lung . given the rarity of these tumors , recommendations regarding chemotherapy or radiation , either pre- or postoperatively , are difficult to formulate . the originality of our case is the disappearance of the pulmonary opacity spontaneously , without any treatment . our case represents , to the best of our knowledge , the first pediatric case having primitive pulmonary mc . the histopathological study familiarizes the diagnosis , but a further ihc study is needed to confirm the diagnosis and to eliminate other etiologies . surgery represents the main treatment for the operable forms . to the best of our knowledge , we have reported the first case , with spontaneous regression of this tumor , without any treatment .

primary myoepithelial carcinoma ( mc ) of the lung is exceedingly rare . we report here , to the best of our knowledge , the first pediatric case having primitive pulmonary mc . the originality of our case was the disappearance of the pulmonary opacity spontaneously , without any treatment . the difficulties in our case were the diagnosis of this rare entity and its subsequent treatment . in fact , given the rarity of these tumors , recommendations regarding chemotherapy or radiation , were difficult to formulate .

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amisulpride came into the indian market a few years back with hypes and hopes in the management of schizophrenia . its broad spectrum effectiveness with lower chances of extrapyramidal symptoms ( eps ) and metabolic syndrome did help psychiatrists to treat schizophrenia and related disorders more effectively . although this antipsychotic does not block serotonin receptors at all , it is a high - affinity and highly selective d3/d2 receptor antagonist with atypical properties . its selective affinity for dopamine receptors in the limbic structures , but not in the striatum , leads to a low risk of extrapyramidal side effects . all available reports suggest that chance of eps is very less with amisulpride at doses < 400 mg / day . however , there are sporadic reports of drug - induced eps including dystonia and akathisia even in patients receiving low doses of amisulpride . here a 30-year - old male with schizophrenia for the past 10 years now presented with predominantly negative symptoms . he was on olanzapine 15 mg / day for more than 6 months without much improvement . hence , amisulpride was instituted with a starting dose of 50 mg / day with a gradual increment up to 300 mg / day within 14 days . the patient came after 14 days to the casualty with features of parkinsonian syndrome such as slowed gait , mild rigidity , salivation , and bradykinesia . he was hospitalized , amisulpride was immediately stopped , and trihexyphenidyl 4 mg / day was given to manage the side effect . his eps gradually subsided and for negative symptoms , clozapine was introduced at a small dose of 25 mg / day and gradually increased to 200 mg / day over a period of 10 days . at the time of discharge , on the 14 day , he was free from parkinsonian symptoms . subsequent follow - up showed no parkinsonian symptoms and he had modest improvement in negative symptoms . a 48-year - old male with schizophrenia for the last 20 years was treated with various antipsychotics without much improvement . since the last 6 months , he was on olanzapine 15 mg / day . as there was no significant improvement , his olanzapine dose was gradually tapered and stopped over a period of 14 days and was started on amisulpride 100 mg / day and was increased to 200 mg / day over a period of 3 weeks . the patient returned on the 24 day with severe parkinsonian symptoms . in this patient also , there was no prior history of parkinsonism . we managed him with injection promethazine 25 mg intramuscular bid first 3 days along with trihexyphenidyl 2 mg bid after stopping amisulpride . after 7 days , parkinsonian symptoms improved considerably and clozapine was introduced at a dose of 25 mg / day which was subsequently increased to 100 mg / day on the 10 day and the patient was discharged . since the discovery that clozapine induces fewer eps and is more effective for negative symptoms than conventional antipsychotics for the treatment of schizophrenia , psychopharmacological research has focused on the development of drugs that block central 5-ht2 receptors more than d2 receptors . combined 5-ht2/d2 receptor antagonism is the most current explanation for the so - called atypical profile of some antipsychotics . amisulpride at low doses binds selectively to dopamine d2 , d3 autoreceptors , thereby enhancing dopaminergic transmission and thus might be effective for negative symptoms . it has no affinity for d1 , d4 , and d5 receptor subtypes . at higher doses , it blocks postsynaptic receptors , thus inhibiting dopaminergic hyperactivity . at the same time , amisulpride has greater specificity for the limbic system and thus has low incidence of eps . amisulpride binds more loosely than dopamine to the dopamine d2 receptor and is rapidly dissociated from the dopamine d2 receptor . low - dose therapy with amisulpride is associated with a significantly lower blockade of striatal dopamine d2 receptors than is seen during high - dose treatment . however , a significant striatal d2 blockade was demonstrated at therapeutically effective dose ranges , and a good relationship between the degree of striatal dopamine d2 receptor occupancy and the amisulpride plasma concentration or the administered dose was shown . in general , asians are slow metabolizers . low body weight and slow metabolism may increase the plasma concentration of drugs causing side effects . reported a low postsynaptic d2 occupancy in the striatum at low doses of amisulpride ( 50100 mg / day ) . it has also been suggested that extrastriatal binding could mediate the effect on negative symptoms . the probable causes of eps with low doses of amisulpride could be that it blocks postsynaptic d2 receptors significantly in striatum without much effect in the mesolimbic pathway . , the lower incidence of eps which is claimed by western researchers as well as pharmaceutical companies should be studied well in the indian context . we should at least keep this side effect in our minds while starting or increasing the doses .

amisulpride , recently introduced atypical antipsychotic , is well - known for its broad spectrum effectiveness and lower profile for extrapyramidal side effects ( eps ) . its selective affinity for dopamine receptors in the limbic structures , but not in the striatum , leads to a low risk of extrapyramidal side effects . here , we report two cases of eps associated with lower dose of amisulpride . the proposed mechanism for its causation is also discussed . authors invite more studies , specifically from the indian context to find out the incidence of eps and other associated side effects .

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thirty patients with zygomatic complex fractures were treated with one point fixation [ figures 13 ] . preoperative peripheral nerve stimulation x - ray preoperative computed tomography scan under general anesthesia , nasoendotracheal intubation was done . the incision can be made from anterior to posterior or from medial to lateral and should extend through mucosa , submucosa , and any buccinators muscle fibers [ figure 4 ] . rowe 's zygomatic elevator was then inserted behind the infra temporal surface of the zygoma , and bone was reduced into its correct anatomical position using superior , lateral and anterior force . an audible click and fullness of the cheek together with palpation for normal contour of the zygomatic bone and orbital rim gave an idea about the adequacy of the reduction . one hand over the side of the face was used to assist in the reduction . a four hole plate with a gap was fixed with 4 mm 2.5 mm screws on the zygomatic buttress [ figure 5 ] . immediate post operative immediate peripheral nerve stimulation x - ray six months postoperative and peripheral nerve stimulation x - ray for all the patients , immediate postoperative and 6 months postoperative peripheral nerve stimulation x - rays were taken , and the x - rays review successful reduction . none of the patients complained of any paresthesia , bony movements or pain in the frontozygomatic or zygomatic buttress region . since intraoral approach was used , all the patients had an aesthetic facial profile without any unsightly scars . the integrity of the zygoma bone is critical in maintaining normal facial width and prominence of the cheek . the zygomatic bone is a major contributor to the orbit and plays an important role in protecting the eyes . zygomatic bone alone is rarely involved in fractures ; usually its articulating surfaces which are maxilla , temporal , frontal and sphenoid bones are also involved . the fractured fragments of a tripod or tetrapod zygomatic complex fracture near these suture lines needs to be restabilized by open reduction followed by fixation . studies suggest that two point gives a considerable stabilization , and three point fixation gives the maximum stabilization . however other studies suggest that one point fixation for zymatic complex fractures gives an excellent results considering the esthetics and stabilization for simple tripod fractures without any comminution of the zygomatic bone or the lateral orbital wall one point fixation with a single mini plate in the frontozygomatic area through the lateral eyebrow incision have been suggested by many authors . i n these cases it was found that when a tripod fracture without any comminution or mild or no displacement can be stabilized very well with a single point fixation in the frontozygomatic area without any complications of diplopia or enopthalmos . however , zygoma provides the attachment point for muscles of mastication and facial animation , but amongst these , it is the masseter that provides the most significant intrinsic deforming force on the zygomatic body and arch . the integrity of zygomatic buttress is necessary for withstanding the contraction force of the masseter muscle . in 2002 fujioka et al . in vivo studies successfully proved that one point fixation at the zygomaticomaxillary complex gives three point alignment and sufficient rigidity when the fractures are not comminuted . in 2011 kim et al . found out that lateral eyebrow incision for mini plate fixation at the frontozygomatic area led to unaesthetic scar and few patients underwent plate removal through a second surgical re - entry through the existing scar of the lateral eyebrow incision which further enhanced the unsightly scars and compromised facial esthetics . since the skin over the lateral eyebrow region is thin there are more chances of palapation of the mini plates after fixation , and it may lead to pain . as early as in 1994 tarabichi et al . proved that in vitro studies are misleading regarding the mini plate fixation along the orbital margins and successfully applied transsinus reduction through anterior comminuted sinus wall . in 2012 kim et al . successfully reduced the zygomatic complex fractured fragments through intraoral approach and gained sufficient rigidity and excellent esthetics with one point fixation at the zygomatic buttress region . we also found that one point fixation with a single mini plate at the zygomatic buttress through intraoral incision provided excellent stability and esthetics in the selected cases of simple zygomatic complex fractures without any comminution of the zygoma or the lateral orbital rim without or with minimal displacement and none of our patient complained of pain or palpation or bony movements in the postoperative study period of 6 months rather they were happy to get operated without any unaesthetic facial scars .

for decades , facial beauty and esthetics have been one of the most important quests of the human race . the lateral prominence and convexity of the zygomatic bone makes it the most important bone for providing the aesthetic facial look and sets up the facial width but at the same time this prominence and convexity makes this bone more vulnerable to injury . zygomatic complex fractures or tripod fractures are the second most common fractures after nasal fractures among facial injuries . several studies have been undertaken regarding the reduction and fixation of zygomatic fractures with mini plates and screws . in 2002 fujioka et al in vivo studies successfully proved that one point fixation at the zygomaticomaxillary complex gives three point alignment and sufficient rigidity when the fractures are not comminuted . in this article , 30 cases have been reviewed with one point fixation of zygomatic complex tripod fractures at the zygomatic buttress through keen 's intraoral approach along with advantages and disadvantages .

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in march 2013 , cases infected with a novel reassortant avian - origin influenza a ( h7n9 ) virus emerged in china and had high mortality . that month , a patient with h7n9 influenza was admitted to our hospital , and daily lung ultrasound was performed . a 54-year - old woman , who ran a convenience store beside a poultry market , complained of cough and high fever for 4 days . her temperature was 38.6c , and she had a heart rate of 113 beats per minute and a respiratory rate of 26 breaths per minute . her white blood cell count was 2.7 10/l , and neutrophil , lymphocyte , and monocyte levels were 72.4% , 22% , and 5.2% , respectively . her partial pressure of oxygen in arterial blood was 72 mm hg , and her fraction of inspiratory oxygen ( fio2 ) was 40% . avian - origin influenza a ( h7n9 ) virus was confirmed from the pharyngeal swabs by real - time reverse transcriptase - polymerase chain reaction . treatment with oseltamivir ( 150-mg capsule taken by mouth twice a day ) was initiated , and she was admitted to an isolated room in the infectious diseases department . bothell , wa , usa ) with c60 convex probe ( 2 to 5 mhz ) was performed , and the lung ultrasound score ( lus ) was recorded , and both effectively reflected the progression of pneumonia ( figure 1 ) . dynamic changes of chest computed tomography ( ct ) , radiography , and lung ultrasound in a patient with h7n9 influenza . ( a - c ) ct showed pneumonia in the left upper lung , with partial consolidation on admission . ( d - f ) lung ultrasound corresponding to ct in ( a - c ) showed multiple abutting b2 lines , and some regions presented a tissue pattern ( arrow ) . ( g - i ) ct on day 6 after admission showed that the pneumonia was partially absorbed . ( j - l ) lung ultrasound corresponding to ct in ( g - i ) showed that the number of b lines was obviously decreased , and the consolidation disappeared . ( m - o ) ct on day 9 after admission showed that the size of the lesion was obviously reduced . ( p - r ) lung ultrasound corresponding to ct in ( m - o ) indicated that only the a line and few b lines were visible . ( s - u ) chest radiography on days 1 , 3 , and 6 after admission showed no obvious change of the pneumonia . ( v ) dynamic changes of lung ultrasound score ( lus ) ( total of 48 for normal lung ) . the onset of h7n9 influenza in this case was manifested by hyperpyrexia and flu - like symptoms and progressed to lobar pneumonia 4 days later . chest radiograph is the routine tool for assessment of pneumonia , but its sensitivity and accuracy were not so good . ct is regarded as the gold standard , but its application is limited in cases with this new emerging virus since strict protection should be followed to avoid person - to - person transmission . ultrasound has many advantages , including convenience , rapidness , non - invasiveness , availability for repeated examination , and absence of radiation . the right lung presented an a line with few isolated b lines , whereas the left lung presented multiple abutting b lines and consolidation . when the patient improved , both b lines and the area of consolidation were decreased and the lus was synchronously increased . this case highlights that ultrasound can be an adjutant to chest radiography and ct in caring for patients with h7n9 influenza . written informed consent was obtained from the patient for publication of this letter and accompanying images .

h7n9 influenza is a new emerging infection and has high mortality . both chest radiography and computed tomography ( ct ) had some limitations in assessing such patients . we performed daily lung ultrasound in a patient with h7n9 influenza . lung ultrasound and lung ultrasound score showed high consistency with ct and the progression of pneumonia . ultrasound can be adjutant to chest radiography and ct in caring for patients with h7n9 influenza .

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a 23-year - old engineering graduate presented with primary palmoplantar hyperhidrosis , for which he was advised an alternate day schedule of tap water iontophoresis . on his next visit , he presented with a very simple iontophoresis device that he devised on his own . the device was constructed with a rechargeable 12 volt battery , two aluminum trays and copper wires , and connecting clamps [ figure 1 ] . hence , using his engineering background he constructed this simple device based on basic mechanism behind iontophoresis . he followed an alternate day schedule of 20 min utes immersion for initial 4 weeks , followed by once a week for next 8 weeks . he achieved an excellent reduction in palmoplantar sweating without any adverse effect , within 3 months of starting iontophoresis . a simple user - made iontophoresis device iontophoresis is defined as passing of an ionized substance through intact skin by application of direct current ( dc ) . tap water iontophoresis is a reliable and effective method for the treatment of palmar and plantar hyperhidrosis , when practiced with appropriate technique and timing . many dermatologists consider simple tap water iontophoresis to be first line therapy for primary focal palmar and plantar hyperhidrosis . the mechanism of production of anhidrosis is not completely understood ; however , obstruction of sweat duct has been suggested as a possible cause . few brands of iontophoresis devices are commercially available ; however , they are expensive and are not readily available . commercially available construction of an iontophoresis device has been described by levit , in which output of the 115 volt isolation transformer is rectified by the full wave selenium rectifier and then filtered by the choke and capacitors , and the potentiometer acts as a voltage divider . levit had suggested that such devices can be constructed at home , and circuit diagram could be found in his original report , but we feel that the procedure for assembling such a device will be difficult for a layperson . for tap water iontophoresis , patients are instructed to apply petroleum jelly with a cotton swab to cover any cuts over the treated area before the session . the trays should be filled with tap water , then , the affected areas should be immersed in the tap water . a monday - wednesday - friday schedule should be followed until the condition improves ; subsequently , the treatment should be tapered once a week for 8 weeks and then once a month for maintenance . additionally , the patient should wear rubber or plastic footwear and should keep himself from directly touching the floor . burning and pin pricking sensations are very common and erythema and vesiculation are transient ; topical corticosteroids cream can be applied for persistent erythema and vesiculation . pregnant women , people with pacemaker or metal implants , cardiac conditions , or epilepsy are contradictions for the use of iontophoresis machine . once a home device is obtained and the patient has received adequate education and training , the maintenance cost and effort are minimal for the patient and health care provider alike . iontophoresis machines basically produce a voltage sufficient to drive a dc of 15 - 20 ma through the hands of patients . an ampere - meter could be used to measure the output current of such user - made devices and can upgrade the voltage of the battery , provided the output is low in terms of current . simple user - made devices such as this one would make the process of iontophoresis very easy , safe , and cost - effective . there are no conflicts of interest .

iontophoresis is defined as passing of an ionized substance through intact skin by application of direct electric current . tap water iontophoresis is reliable and effective method for treatment of palmar and plantar hyperhydrosis when practiced with appropriate technique and timing . one of the major setback for using iontophoresis is that the apparatus is expensive and is not readily available . a simple user - made iontophoresis device have been described here , which could be easily constructed and used at home .

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a 61-year male presented to the emergency department with a history of road traffic accident . he arrived hemodynamically stable with a blood pressure of 126/76 mmhg and a heart rate of 78 beats per minute . on plain radiograph ( fig . 1a ) anteroposterior and two judet 45 oblique view1 ) and computed tomography ( ct ) scan of pelvis ( fig . 1b ) , the findings revealed both column fracture of acetabulum without hip dislocation , but no presence of femoral head fracture or onfh . buttress plating through ilioinguinal approach was performed using a reconstruction plate , which was supplemented by a compact hand plate . the patient was transfused 8 units of whole blood , 3 units of fresh frozen plasma and 8 units of packed red blood cells . intra - operative hb was 9.7 gm% ; the average mean arterial pressure was 91.82 mmhg during the operative procedure . post - operatively the patient was transfused 2 units of whole blood and 1 unit of fresh frozen plasma . the hb postoperatively was 10.3 gm% , the patient was shifted to intensive care unit for a day , later was transferred to the ward . the post - operative x - ray ( fig . 2a ) and ct scan revealed an acceptable reduction of the fracture fragments and a concentric hip . sitting up was performed on the first postoperative day ; the patient subsequently began formal physical therapy and active range of motion exercises . partial toe touch weight bearing ( 20 to 30 lb ; 9 to 13.6 kg ) with a walker was maintained for 6 - 8 weeks . progression to full weight bearing was started on the basis of the follow - up radiographs . the patient 's 4-month postoperative x - ray revealed a radiolucent lesion in the superolateral part of femoral head , crescent sign , and sclerosis . 2b ) showed collapse and sclerosis , findings consistent with onfh . a ct scan ( fig . on was diagnosed only when the radiographic findings provided a clear differentiation from wear of the femoral head2 ) . the joint pain increased due to the onfh , we performed a total hip replacement ( fig . 3b ) 12 months after the index surgery . late complications of acetabulum fractures include heterotopic ossification and onfh , which are present in less than 10% of the population3 ) . the incidence of onfh is known to be high in transverse and posterior wall fractures associated with posterior dislocation6 ) . on also occurs in conjunction with approximately 3% of anterior hip dislocations and in more than 13% of posterior hip dislocations . in a recent meta analysis of 3,670 surgically treated displaced acetabular fractures the incidence of onfh showed an overall incidence of 5.6%3 ) , suggesting that it is grossly overestimated and that most of the observed changes in the head of the femur are probably due to osteoarthritis5 ) . onfh is caused by inadequate blood supply to the affected segment of the subchondral bone . when posterior surgical approaches have been used , on rates as high as 42% within the first year after surgery have been reported7 ) . many systemic conditions are associated with on , but 25% of all cases are described as idiopathic and can contributes as a cause9 ) . trauma is one of the most common causes of on , interruption of the blood supply to the affected segment of the bone being the cause of ischemia . in this case the exact cause of onfh eludes us , especially in the absence of any patient related predisposing risk factors , except presence of fracture without hip dislocation and subsequent intervention by an ilio - inguinal approach . a probable theory of etiology could be the intra - operative hypovolaemia , low mean arterial pressure , causing compromised flow to the femoral head being so as to act as the final blow . alteration of the blood supply to vital organs during hypovolaemia is well established . with mean arterial pressure usually in the range of 50 to 60 mmhg , the flow to the femoral head is potentially compromised10 ) so as to act in an accumulative stress theory , as suggested by kenzora and glimcher9 ) . it is questionable as to whether this alone would be enough to explain the development of on .

osteonecrosis in isolated fractures of the acetabulum without dislocation of hip seems to be a known complication , but to our knowledge it has not been reported adequately . the causative nature of post - traumatic femoral head osteonecrosis has not been studied critically . the pathophysiology of osteonecrosis in this case also eludes us . striking evidence points towards the intra - operative blood loss and low mean arterial pressure possibly leading to hypo - perfusion of femoral head leading to osteonecrosis . fractures of the acetabulum pose a difficult problem for the patient and the surgeon because of possible complications . thus any surgeon involved in surgery for fractures of the acetabulum should be aware of the possibility of this potential complication . here is a 61-year male , who sustained a complex fracture of the acetabulum without hip dislocation , subsequently was treated surgically with internal fixation using an anterior approach , 10 months after surgery patient developed osteonecrosis of the femoral head .

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caustic esophageal injury in infants is a devastating insult to the gastrointestinal tract and will often require major reconstructive surgery to replace the damaged esophagus . esophageal replacement with colonic interposition has been utilized since dale and sherman performed the first retrosternal colonic interposition in 1955 . up to 80% of patients with colonic interposition endoscopic dilation is relatively safe and effective for the initial treatment of anastomotic strictures , but surgical management is indicated in refractory cases . when surgery is required , graft revision utilizing both a thoracotomy and laparotomy is common . we report a case of cologastric stricture treated with resection and reconstruction of the anastomosis solely through an abdominal approach , which can offer less morbidity and mortality . a 31-year - old male developed a caustic esophageal injury after ingestion of an alkaline solution when he was 2 years old . he required emergent esophagectomy , proximal gastrectomy and reconstruction with a colonic interposition graft from the cervical esophagus to the stomach . the patient also had a pyloric stricture , for which a gastrojejunostomy was performed . over the next three decades , he required frequent hospital admissions for abdominal pain and dysphagia , and had multiple endoscopic dilations performed for a severe cologastric anastomotic stricture ( fig . the patient had severe malnourishment , with a body mass index of 14 kg / m . he had a jejunostomy feeding tube for nutritional support , which had been removed due to abdominal pain a few months prior to presentation . endoscopic evaluation revealed the severe cologastric stricture , severe inflammation just proximal to the cologastric anastomosis , and significant ulcerative disease at his gastrojejunal anastomosis . the patient underwent resection of the cologastric anastomosis and the gastrojejunal anastomosis with ulcerated stomach , neo - cologastric anastomosis and neo - gastrojejunal anastomosis via a transabdominal approach without a thoracotomy . intraoperative endoscopy was utilized during the case to ensure that the cologastric anastomotic stricture was entirely resected . figure 2:preoperative cologastric and pyloric strictures ( a ) and post - operative changes including new cologastric , gastrojejunal , duodenojejunal anastomoses ( b ) . preoperative cologastric and pyloric strictures ( a ) and post - operative changes including new cologastric , gastrojejunal , duodenojejunal anastomoses ( b ) . the patient tolerated the procedure well and was transferred to the intensive care unit . on post - operative day 2 , he was transferred to the floor . his post - operative course was remarkable for development of an intra - abdominal fluid collection , which required percutaneous drainage , but was otherwise uncomplicated . severe esophageal damage may require resection with creation of a neo - esophagus . because these operations occur in children , complications in regards to the colonic interposition graft classically , a transthoracic approach has been used to resect the entire colonic graft . in this situation , a repeat colon graft may be needed , which carries high morbidity and mortality . in particular , the majority of the proximal colon graft , which was functional , was able to be spared . given our patient 's preoperative nutritional status , he would be at a predisposed risk for wound healing complications . via transabdominal approach , to the best of our knowledge , this is the first written report of using a completely transabdominal approach for revision of the colonic graft . our patient had previously been refused surgery by multiple surgeons . if this relatively less complex surgical management was used , perhaps he would have had definitive treatment much earlier . our patient did well with our transabdominal approach given the chronicity of his symptoms . in light of his clinical dilemma of continued non - operative versus operative intervention , his symptoms were relieved immediately . this surgical approach should be considered in patients with history of colonic interposition requiring reoperation for complications of the colonic graft .

a 31-year - old gentleman who had undergone an emergent esophagectomy and reconstruction with a colon interposition graft , presented with a long - standing cologastric stricture . he had undergone multiple attempts at endoscopic dilation over multiple decades with little symptomatic relief . he underwent a resection and reconstruction of the anastomosis entirely through an abdominal approach . he did well from surgery and experienced complete symptomatic relief immediately . complications of colon interposition grafts can occasionally be treated using an abdominal incision only .

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a 60-year - old male patient was referred to an otorhinolaryngology clinic due to a lump on the left side of his jaw , which had grown in 2 months . ultrasound sonography test examination revealed a cystic mass that was 2417 mm in size with smooth contours . multiple echogenic and reactive lymph nodes with partially visible hila were visualized in the neighboring upper jugular chain , with the largest being 1610 mm in size . following a neck magnetic resonance imaging and a preliminary diagnosis of wt , left superficial parotidectomy materials were sent for pathologic examination in two pieces , which were 53.22 cm and 4.531.2 cm in size . cross section analysis showed an off white - yellowish , well - contoured nodular tumor with a bleeding center of 42.52.2 cm . microscopic examination indicated that the tumor had epithelial components with basaloid and oncocytic columns of cells neighboring lymphoid components ( fig . in addition to the lymphoid follicles with distinct germinal centers , infiltration of large neoplastic cells with bizarre and extremely atypical morphology was seen in the lymphoid component ( figs . 2 , 3 ) . 4b ) , leukocyte common antigen , igg , cd138 , mum1 , and focal positivity for kappa . staining for lambda , igm , iga , cd3 , cd5 , cd10 , cd15 , cd56 , epithelial membrane antigen , bcl2 , bcl6 , cyclind1 , s100 , pancytokeratin , cytokeratin 20 , human melanoma black 45 , actin , and desmin were negative . latent epstein - barr virus ( ebv ) was shown to be negative in tumor cells by using ebv - encoded rna chromogenic in situ hybridization . due to these findings , the patient was diagnosed with " wt and cd30 positive diffuse large b - cell lymphoma in the parotid gland . " following the lymphoma diagnosis , a full body screen was performed . in addition to these findings , the left suprarenal gland showed two nodular mass lesions , which were assessed as likely adenomas ; however , this preliminary diagnosis was not confirmed by histopathology . the patient was stage 3a and received six courses of rituximab , cyclophosphamide , doxorubicin , vincristine , and prednisone ( r - chop ) therapy . during 6-month follow - up , the patient was free of disease . wt is the second most common type of salivary gland tumor . in 10 - 15% of cases , it is bilateral , and it accounts for 70% of all bilateral salivary gland tumors.2 the male / female ratio is 1.6/1 , and it typically develops in the 6th and 7th decades . smoking increases the risk of developing wt.5 microscopically the tumors are typically composed of proliferative epithelial components accompanied by lymphoid stroma with lymphoid follicles that have distinct germinal centers . histogenesis of the lymphoid stroma in wt has been a topic of discussion for many years . lymphoid stroma can arise as a cell response to epithelial neoplasms or as a normal lymph node due to residue held by the epithelial neoplasm.5,6 the most widely accepted hypothesis suggests that wt is a neoplasm that develops in the heterotopic salivary gland ductus within or around the parotid lymph nodes.7 transformation to carcinoma in wt is a well - known phenomenon ; however , the development of lymphomas from wts is very rare.4,8 although some cases contain a normal residual lymphoid component , in others cases the lymphoid component contains entirely neoplastic lymphoid cells.4 in the present case , non - neoplastic lymphoid tissue was also present in the neighboring areas . the pathogenesis of malignant transformation of wt remains unclear ; however , exposure to radiation is of particular interest , as the relationship between previous radiotherapy and lymphomas arising from wts has been determined by some authors.4,5,9 chronic immune sialadenitis is thought to play an important role , independent of the presence of sjgren syndrome symptoms.4,7,10 in this case , there was no history of radiotherapy or sialadenitis , but a history of smoking may have provoked the development of wt . saxena et al.1 state that because the lymphoid stroma of wt is part of the systemic lymphoid tissue , in patients with lymphomatous spread of wt , disseminated disease is present during the staging either at the time of the diagnosis or after . in the present case , with screening techniques , lymphadenopathies of a pathologic size were found in the inguinal and iliac regions . some researchers suggested that although the relationship between wt and lymphoma could be coincidental , it might also be of a pathogenic nature . according to the latter statement , a single agent can affect different tissues or one tumor could trigger the formation of another . from this point of view , the epithelial component is a continuous antigenic stimulator for the lymphoid component , which provides the stimulus for the development of lymphoma.1,6,8 according to this theory , the frequently observed reactive follicular hyperplasia in wt may be histological evidence of chronic antigen stimulation.1 it has been suggested that the lymphomas seen with wt are typically non - hodgkin lymphomas ; however , there are a few cases reporting hodgkin 's lymphomas.11,12 the majority of non - hodgkin 's lymphomas in wt are follicular lymphomas . dlbcl , small lymphocytic lymphoma , extranodal marginal zone lymphoma of mucosa associated lymphoid tissue , and mantle cell lymphoma have also been reported.4,6,8,9 a small number of t - cell lymphomas such as peripheric t - cell lymphoma and t - cell lymphoblastic lymphoma have also been described in wt.4,8,13 in summary , malignant lymphomas in wt are very rare . the presented case is a diffuse large b - cell lymphoma expressing cd30 positivity . to the best of our knowledge this is the first case in literature describing dlbcl with expression of cd30 in wt .

warthin 's tumor is the second most common type of salivary gland tumor . microscopically , warthin 's tumor displays a proliferative epithelial component and lymphoid stroma . carcinomas arising from the epithelial component are well known , but malignant transformations of the lymphoid stroma are rare . when they do occur , they are most commonly b - cell type non - hodgkin lymphomas . a 60-year - old male patient underwent surgical resection of a parotid mass . after superficial parotidectomy , microscopic examination indicated that the tumor was of epithelial components with basaloid and oncocytic columns of cells neighboring lymphoid components . in addition to the lymphoid follicles with distinct germinal centers , there were large , bizarre and extremely atypical neoplastic cells seen in the lymphoid component . large neoplastic cells were diffusely cd20 and cd30 positive . the patient was diagnosed with " warthin 's tumor and diffuse large b - cell lymphoma with expression of cd30 . " the histopathologic and clinical features are discussed along with a review of the literature .

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