Urinary bladder agenesis is a very rare congenital anomaly with very few cases reported in the literature.We report a one-month-old baby presenting with ambiguous genitalia and recurrent urinary tract infections. Her clinical course was complicated by renal impairment. Magnetic resonant imaging (MRI) revealed a diagnosis of bladder agenesis with bilateral ectopic insertion of the ureters into the vagina, associated with several other anomalies. The patient underwent bilateral high anterior ureterostomies in an hospital abroad at 5.5 months of age. She then developed ureteral necrosis that had to be corrected with left pyeloplasty and by placing a left nephrostomy tube for drainage. Eventually, the patient’s renal function declined, and she developed chronic kidney disease (CKD).The case with its imaging findings and pathogenesis as well as a review of the literature are presented.Urinary bladder agenesis is a rare congenital condition that can be associated with multiple anomalies. Early diagnosis and therapeutic intervention can prevent progression to chronic kidney disease. Reported associated anomalies include those of the urogenital, gastrointestinal, vascular and musculoskeletal systems [Anomalies of the urogenital system are commonly found on pre- and postnatal imaging studies. Urinary bladder agenesis is one of the rarest urinary tract anomalies, with a reported incidence of 1 in 600,000patients –5. Only  systems , 6. The  systems . We repoThe patient was the product of a term uncomplicated pregnancy and a spontaneous vaginal delivery, who was found to have ambiguous genitalia after birth in another center. The parents are nonconsanguineous and have 5 other healthy siblings. The patient was referred to the plastic surgery unit at our hospital at the age of 1 month for possible surgical correction. She also had low imperforate anus with stenosis that was dilated.Laboratory investigations ruled out underlying congenital adrenal hyperplasia and revealed a female karyotype of 46 XX. Cystoscopy revealed what appeared to be a short urethra and atresia of the lower vagina. An ascending urethrogram then followed, with the aim of assessing the location of the confluence between the vagina and urethra. The urethrogram failed to opacify the urethral canal; instead, it revealed a triangular-shaped cavity with a uterine cervix impression at its superior aspect Fig. . The plaThe patient then suffered from recurrent urinary tract infections (UTIs) indicated by several positive urine cultures, the first of which was associated with dehydration and acute kidney injury (AKI), requiring admission. On her initial ultrasound, the left kidney was normal, the right kidney was dysplastic and it was difficult to assess the urinary bladder. A micturating cystogram (MCUG) showed a urinary bladder-like structure, with a tubular outpouching from its superior aspect, thought to be a diverticulum or a refluxing ectopic ureter Fig. .Fig. 2a At 5 months of age, the baby developed progressive ascites. Ascitic taping and drainage showed high ascitic fluid creatinine level compared to serum creatinine level , denoting urinary ascites. CT cystogram performed to rule out bladder injury showed no leak of contrast. The structure opacified with contrast on the CT cystogram had a similar appearance to that observed on the MCUG. Nevertheless, the structure was lower and more posterior than was the normal full urinary bladder; hence,it was thought to possibly be the vagina Fig. . EvaluatSurgery was then planned to perform bilateral nephrostomy tubes for urine diversion and to obtaina better idea about anatomy. Before the surgery, the urologist was aware of the urogenital anomalies and the superficial course of the artery supplying the right lower limb seen on the MRI. However, the parents decided to take the baby to another center abroad for treatment and operation, where she had bilateral high anterior ureterostomies at 5.5 months of age.She presented 3 weeks later to our emergency department with AKI, ascites and left ureterostomy obstruction. At presentation, the ureterostomies were nonfunctional, and insertion of the nephrostomy tube was deemed impossible according to the interventional radiologist. Therefore, surgical exploration of the left side (the only functioning side) was performed, revealing complete ureteral necrosis and a 1-cm segment of renal pelvis remaining. This renal pelvis remnant was open to the peritoneal cavity, and there was intraperitoneal urine leakage. The left renal pelvis was then closed after leaving a nephrostomy tube in place for drainage. No intraoperative or postoperative bleeding was encountered. The nephrostomy tube was then complicated by frequent blockage, leakage and infection with difficult reinsertions. The patient received several types of antibiotics throughout her hospital stay for her recurrent UTIs, including trimethoprim/sulfamethoxazole, imipenem, piperacillin/tazobactam, fluconazole, caspofungin, ceftazidime, colistin, nitrofurantoin, ciprofloxacinand clindamycin.The patient’s renal function declined slowly, and at the age of 11 months, she had evidence of chronic kidney disease associated with high creatinine, hyperphosphatemia, metabolic acidosis and anemia.thlive-birth case of bladder agenesis reported in the literature.We present a case of bladder agenesis associated with several other congenital anomalies. To our knowledge, this is the 26th week of embryogenesis, leading to urogenital sinus injury [The underlying etiology remains indeterminate and was postulated to be due to an insult at the 5th to 7s injury , 7. Altes injury , 7.Age, presentation and outcome are variable and depend on the associated anomalies. Most cases present during infancy or childhood. Only one reported case presented as an adult, at the age of 22 years . There iOur case, as with most reported cases, is a female with a presentation of ambiguous genitalia and recurrent UTIs associated with several anomalies. Specifically, our case had urinary bladder agenesis, bilateral hydroureteronephrosis, ectopic insertion of the ureters into the vagina, dysplastic right kidney, uterus didelphys, low aortic bifurcation with superficial course of the right iliac artery posterior to the anterior abdominal wall, absent internal iliac vessels and a segmentation anomaly of the spine. There was a considerable delay in the patient’s diagnosis with bladder agenesis, as she was diagnosed at the age of 6 months. This delay was due to the rarity of the condition, requiring an advanced center for management. Despite this limitation, we had good radiological tests, a good pediatric nephrology team and an expert pediatric urologist.Reported cases of urinary bladder agenesis had various associated abnormalities. The most common associated anomalies were those of the urinary system, including renal dysplasia/hypoplasia, pelvic kidneys, crossed fused renal ectopia, horseshoe kidneys, ectopic insertion of the ureters and urethral agenesis , 13–15. Variations of the aorta and iliac arteries were described in cases with urogenital anomalies. These alterations can predispose major vessels to injury during surgery, leading to major complications such as hemorrhage or limb loss . TherefoOther described associations include imperforate anus, omphalocele and anomalies of the spine and musculoskeletal systems, including VACTERL association, spina bifida, vertebral segmentation anomalies, scoliosis and hip dysplasia , 21, 22.The diagnosis of bladder agenesis was made by MRI in most reported cases , 14, 16.Urinary bladder agenesis is a rare congenital condition that can be associated with multiple anomalies. It should be considered in patients with unexplained or atypical UTIs because early diagnosis and therapeutic intervention can prevent or delay the progression to chronic kidney disease. Early MRI is advised for definitive diagnosis.


Neuroendocrine neoplasm of the gallbladder is an extremely uncommon diagnosis. We present a case of a benign gallbladder paraganglion that was initially incorrectly diagnosed as a neuroendocrine tumour (NET).A 27-year-old female with symptomatic gallstone disease underwent an uncomplicated laparoscopic cholecystectomy. Routine histopathologic examination suggested the presence of a small adventitial NET. However, histopathological revision was performed by our pathologist because of regional gallbladder carcinoma (GBC) treatment evaluation. The revision demonstrated the presence of a normal paraganglion, a preexistent structure that is only rarely encountered during routine histopathologic examination of the gallbladder.Neuroendocrine neoplasms of the gallbladder are extremely rare. Treatment varies from a simple cholecystectomy to extensive surgical resections. Chemotherapy is usually reserved for metastatic disease. In contrast, a gallbladder paraganglion is a benign entity not requiring additional treatment.A neuroendocrine neoplasm of the gallbladder may closely resemble a benign paraganglion. If a NET is suspected, the clinician should be aware of the histopathologic mimicry of a paraganglion prior to initiating additional treatments. Physicians may be confronted with a gallbladder neuroendocrine neoplasm as a coincidental histopathological finding following a standard cholecystectomy for benign gallstone disease. In general, an up to 2.8 % incidence of gallbladder malignancy is demonstrated following an elective cholecystectomy, and just 2 % of these 2.8 % are neuroendocrine neoplasms . We pres2In 2007, a previously healthy 27-year-old Caucasian female was referred to the surgical outpatient clinic for analysis of abdominal pain. The upper right quadrant pain had started a few months earlier and was intermittently present up to one hour following food ingestion. Jaundice, fever, vomiting, melena or altered bowel habits were absent. The family history for cancer or MEN-1 disease (Multiple Endocrine Neoplasia Type 1) was unknownPhysical examination only revealed mild pain following deep upper quadrant palpation without signs of peritonitis. As gallbladder disease was likely, ultrasonography of the abdomen was advised that demonstrated cholecystolithiasis. The morphology of the gallbladder was deemed normal as were all other abdominal organs. The patient was planned for a laparoscopic cholecystectomy that was performed uneventfully a few months later.A routine histopathologic examination showed a 0,3 mm NET in the adventitia of the gallbladder with clear resection margins . A subty3GBC is a rare form of cancer with a declining incidence in the Netherlands . NonetheThe diagnosis of a gallbladder NET is difficult due to its rarity. In addition, presenting symptoms are generally non-speciﬁc including right upper quadrant abdominal pain, jaundice and weight loss. Most lesions are found after routine histology following cholecystectomy for symptomatic cholelithiasis. Fewer than 1 % of all NETs present with a carcinoid syndrome or rarely with a variety of other paraneoplastic syndromes .These cases, patients are diagnosed in an advanced stage of the disease and survival varies depending on subtype. Radiological tests including ultrasonography, computed tomography scan (CT scan), magnetic resonance imaging and Positron emission tomography–computed tomography (PETCT) scans can reveal gallbladder masses indicative of gallbladder cancer. Ga-DOTATOC-PET scans ((68)Ga-DOTA(0)-Phe(1)-Tyr(3)-octreotide Positron Emission Tomography Computed Tomography) can distinguish between a neuroendocrine neoplasm and GBC, but are almost never performed preoperatively in case of suspicion of benign gallbladder disease. Therefore, it is almost impossible to clinically, radiologically, or intra-operatively distinguish neuroendocrine neoplasms from other subtypes of gallbladder carcinomas ,11. PathNeuroendocrine neoplasms of the gallbladder are classified into three main histological categories according to the current World Health Organization (WHO) Classification of Tumours of the Digestive System (2019): neuroendocrine tumor (NET), neuroendocrine carcinoma (NEC), and mixed neuroendocrien-nonneuroendocrine neoplasms (MINEN)  Fig. 2)Fig. 2). NET are well-differentiated tumors divided into NET1, NET2 and NET3. Grading is mainly based on the assessment of proliferation. NET-G1 is characterized by a mitotic count of less than 2 mitoses per 10 high power fields (hpf) and/or a Ki-67 proliferation index of less than 3 %. NET-G2 is characterized by a mitotic count of 2–20 mitoses per 10 hpf and/or a Ki-67 proliferation index between 3 % and 20 %. NET-G3 have more than 20 mitoses/2 mm2 and a Ki-67 proliferation index of more than 20 %, but they are morphologically not poorly differentiated unlike NEC. NEC are further subdivided into small cell and large cell carcinoma based on cytonuclear features like the amount of cytoplasm and the presence of prominent nucleoli .MINEN have a phenotype that is morphologically recognizable as neuroendocrine and also nonneuroendocrine epithelial (mostly adenocarcinoma) and is diagnosed only when both portions form more than 30 % of the tumor ,12.The first NET-G1 (previously known as carcinoid tumor) was described in 1888, while the first primary NET-G1 tumor of the gallbladder was reported by Joel in 1929 . The mosThe surgical treatment of NETs of the gallbladder varies widely and is similar to other types of gallbladder cancer, ranging from cholecystectomy to extensive surgical resections, including regional lymph node dissection with possible hepatic lobectomy. Due to the rarity of the NETs, distinctive surgical strategy is missing for NETs in comparison to other types of gallbladder cancer. The therapeutic value of chemotherapy remains unclear, but is likely the best form of palliation of metastatic NET. Other palliative treatments like octreotide, Peptide Receptor Radionuclide Therapy (PRRT) or everolimus are reserved for metastatic NEC . AdditioThe 5 year survival rate for NETs is versatile and mainly dependent on the subtype. Thus, NET grade I has a favorable prognosis compared to NET grade II or a NEC. Soga et al. found 5 year survival rates of 60.4 % and 21.3 % for NET grade I and NET grade II respectively ,18.Paraganglions of the gallbladder are in contrast a benign entity  and liteParaganglion should not be confused with another entity of the gallbladder know as a paraganglioma. These paraganglioma of the gallbladder arises from chief and sustentacular cells origination from the neural crest cells . ParaganIn short, NETs of the gallbladder are an extremely uncommon form of the already rare entity of GBC and dependent on the subtype can show aggressive behaviour and poor prognosis. Patients present usually with symptoms of benign gallstone disease. NETs are clinically indistinguishable from other types of gallbladder cancer. Radical surgery is recommended for localised disease and may include lymphadenectomy combined with hepatic resection. Conversely, a paraganglion of the gallbladder is a benign condition which does not require additional surgery following a cholecystectomy.4We encountered a case of an otherwise healthy patient, with a unexpected diagnosis of the gallbladder. Initial diagnosis was a NET-grade 1 of the gallbladder. However, revision of the pathology years later, showed a benign paraganglion. With this case-report we would like to draw attention of clinicians to the histopathologic mimicry of neuroendocrine neoplasms and paraganglion tissue.No funding to disclose.Ethical approval is not needed for this case report.Informed consent was obtained for publication of this case report.Conception and design of study: BJGA Corten, WKG Leclercq.Acquisition of data: BJGA Corten, W van den Broek, PH van Zwam.Drafting the manuscript: BJGA Corten, WKG Leclecq, MW Dercksen, PH van Zwam.Revising the manuscript critically for important intellectual content: BJGA Corten, WKG Leclercq, MW Dercksen, W van den Broek, PH van Zwam, C Dejong, GD Slooter.No registration of research studies.BJGA Corten, GD Slooter.Not commissioned, externally peer-reviewed.The authors declare that they have no competing interests regarding the publication of this manuscript.


Hamartomas are considered a mixture of nonneoplastic tissue, which may be indigenous to a different location in the body. As such, they may be epithelial, mesenchymal, or mixed. In the sinonasal region, the following hamartomatous lesions are considered to lie on a spectrum and include respiratory epithelial adenomatoid hamartoma (REAH), chondro-osseous respiratory epithelial adenomatoid hamartoma (COREAH), and nasal chondromesenchymal hamartoma (NCMH). To our knowledge, less than 50 cases of sinonasal hamartomas have been reported in the English literature so far with NCMH being very rare and primarily a tumor in infancy, with only 2 cases reported in individuals older than 16 years of age. We report a highly unusual case of a NCMH in the right maxillary sinus of a 70-year-old female. A 70-year-old female presented with a two-year history of slowly growing, nonpainful maxillary sinus mass. She has a history of chronic maxillary sinusitis corresponding to presentation of the mass, with the first episode reported in 2014. Computed tomography (CT) imaging revealed an erosive right maxillary sinus mass (2.5 x 2.1 cm) with bony destruction.Surgical excision of the right maxillary sinus mass revealed a fragmented, white, vaguely nodular, and whorled lesion. Histological examination revealed fragments of respiratory-type epithelium with focal cystic invagination and associated squamous metaplasia . The undNasal chondromesenchymal hamartomas are most commonly seen in the nasal cavity of children less than 3 months old, with less common involvement of the paranasal sinuses . As per Our case would lend support to extending the age range for NCMH and considering it in the differential diagnosis of all sinonasal region tumors, irrespective of age, and location in the head and neck region. Despite primarily being a benign lesion, these tumors can present with areas of necrosis and local destruction, including bony invasion. The tumors can be aggressive appearing on imaging, extending into bony structures, including the cranium and/or the orbital cavity, which should not lead away from the diagnosis of this benign lesion. Detailed CT or preferably MRI prior to surgical excision should be performed.NCMH has been associated with development of pleuropulmonary blastoma (PPB) during infancy. A recent  report hWhether the presentation of a NCMH at a later age predisposes to malignant transformation due to the long-standing nature of the lesion is up for debate. It could represent a somatic DICER1 mutation rather than a germline mutation, causing the hamartoma to form later in age. Longer follow-up results from the adult cases and routine genetic testing in all NCMH will help provide an answer to these questions.We report an unusual case of NCMH eroding the right maxillary sinus of a 70-year-old female. Although, NCMH is a rare entity with predilection for pediatric age groups, it is important to consider NCMH in the differential diagnosis of nasal/sinonasal masses in adult patients in order to avoid diagnostic errors.


An 85-year-old male referred to the Gastroenterology (GI) clinic with three-month history of failure to thrive and three-week history of nausea, vomiting, and melanotic stools. Ulcerative mass obstructing gastric outlet was found on endoscopy and on follow-up CT abdomen a homogeneous submucosal mass in the gastric antrum was identified. Radiological diagnosis of giant gastric lipoma was established and patient was evaluated for surgery and, however, was rendered unfit for surgery due to his comorbid conditions. Patient was taken for endoscopic resection of the mass. On endoscopy, only partial resection was achieved due to the size of the mass, but endoloops were deployed at the stalk at the end of the procedure in hope of limiting blood supply to the lesion. On six-week follow-up endoscopy, patient's mass had completely disappeared with limited scar tissue at the site. Symptomatic gastric lipomas are rare neoplasms of GI tract. Bleeding due to mucosal erosions on top on the lipomas and mass effect leading to gastric outlet obstruction are some of the common presentations in such cases. Due to its rarity, there is no generalized consensus on the management. Surgical resection of the mass is the common approach in such cases, but in cases where debility or other comorbidities preclude surgery, endoscopic resection and debulking can be attempted. Here we present the case of a giant gastric lipoma where surgical resection was not feasible due to patient's comorbidities. Endoscopic resection was attempted; however due to the size and location of the lesion, only partial resection was achieved. At the end of the procedure, using a needle knife, base of the lesion was cut to create deep grooves at the stalk to prevent slipping of endoloops and an endoloop tightly encircling the stalk was left in place. On six-week follow-up, there was complete disappearance of the mass with minimal scarring of the site. Endoscopic loop placement to limit blood supply can be attempted in the management of large gastric lipoma to prevent major surgeries and its associated morbidity.An 85-year-old male was referred to GI clinic by his primary care physician for evaluation of anemia, weight loss, and positive stool occult blood. On obtaining a detailed history he admitted to having early satiety for the past three months and nonbilious vomiting and colicky epigastric abdominal pain for the past three weeks. His physical examination was pertinent for pallor and his abdomen was soft and nontender with no apparent swelling or hepatosplenomegaly. An esophagogastroduodenoscopy  was perfAfter the failure of initial attempts to do a complete resection, partial piecemeal resection was made with the aim of debulking the lesion to relieve symptomatic gastric outlet obstruction. To limit bleeding, endoloops were deployed at the base of the lesion prior to the start of resection. Because of the location and size of the lesion, only 2 cm by 2 cm of the mass was resected. At the end of the procedure, one of the endoloops remained deployed tightly at the base protecting the margins and limiting blood supply . The basRepeated EGD  at four-Lipomas of GI tract are rare in occurrence 1 in 600). These are the third common benign tumors in GIT after adenomas and hyperplastic polyps. These are intramucosal mesenchymal tumors of mature adipocytes and are often incidental findings on endoscopy or colonoscopy [ in 600. In radiology, a well-defined and homogeneous mass in the stomach wall is suggestive of gastric lipoma. On endoscopic ultrasound, the lesion shows up as a homogeneous hypoechoic lesion. Biopsy is rarely needed for a diagnosis and endoscopic signs such as Cushing sign and tenting sign combined with EUS or CT scan are usually diagnostic.In addition, because of the submucosal location, routine biopsies are never deep enough to secure tissue to make an accurate diagnosis .Treatment for gastric lipoma is controversial. Because of its rarity, a standardized approach in treating large symptomatic lesions has not yet been established . GeneralOur patient, because of his cardiovascular risk factors, was not a candidate for surgery. The technique of limiting blood supply to the lesions using an endoloop resulted in complete resolution of the lesion. This technique which has already been used for other benign gastric lesions such as colonic polyps can be used effectively for cases of gastric lipoma as well. Such endoscopic approaches can be a good alternative to surgery and can prevent significant morbidity and mortality. Gastric lipomas are benign mesenchymal tumors of adipocytes commonly seen in the gastric antrum. They are diagnosed by the homogeneous appearance, well-defined borders, and submucosal location in imaging studies. In the rare situations where they cause symptoms, they can cause symptoms of upper gastrointestinal bleed or gastric outlet obstruction. Symptomatic gastric lipoma is a rare occurrence. Because of the rarity, there is no consensus on the treatment in such cases. Majority of cases are managed through surgical resection which can cause significant morbidity. In a systematic review of 32 cases of large gastric lipoma published by Cappell et al.  the vast


To evaluate clinical presentations, diagnosis and management of paediatric patients presenting with pituitary apoplexy.A retrospective case series describing a cohort of paediatric patients presenting with this condition from 2010–2016 to a tertiary referral children’s hospital in the United Kingdom.Pituitary apoplexy is a rare condition that seems to have a higher relative incidence in children than adults. Our series suggests that pituitary apoplexy in paediatric patients with adenomas appears more common than previously described. All our patients required surgery, either as an acute or delayed procedure, for visual compromise. Two patients had commenced growth hormone (GH) for GH deficiency two weeks prior to the onset of pituitary apoplexy.With only a limited number of published case reports surrounding this topic our case series contributes to help further understand and manage this condition. Pituitary adenomas in children are rare with a prevalence of one per million and almost never malignant . They acPituitary tumour apoplexy is a rare endocrine emergency, resulting from ischaemia and necrosis of a pituitary tumour. There are few case reports –6 and noWe therefore present a series of paediatric patients presenting with pituitary tumour apoplexy to help better understand this rare condition.We present a case series of five paediatric patients,  all of whom presented as an emergency to a tertiary referral children’s hospital since 2010, with pituitary tumour apoplexy. All the patients were noted to have prolactinomas. Table A 16 year old girl presented with severe headache and visual loss, upon further questioning she also had primary amenorrhoea and galactorrhoea. An MRI [Fig. A 14 year old boy was admitted with headaches and weight gain. At presentation he had only perception of light in his left eye and hand movements in the right eye. His MRI showed a pituitary tumour extending suprasellarly to compress the chiasm with features of apoplexy. The initial prolactin was 10,626 miU/L. Given his very poor visual function, he underwent emergency ETSS. Post-operatively his vision improved but he remains with significant visual impairment. Histology confirmed a prolactinoma with a moderate Ki67 (5%) and some mitotic activity. He remains on cabergoline although his subsequent MR imaging shows minimal residual disease.A 15 year old boy was admitted with severe headache, reduced visual acuity and a sixth nerve palsy. Following an MRI he was diagnosed with a large pituitary macroadenoma with features of apoplexy. His prolactin was raised at 47,173 miU/L and he was commenced on cabergoline. His acuity had some improvement, however his sixth nerve palsy remained. Given his on-going visual symptoms, he underwent ETSS 6 weeks later. Post-operatively, his vision returned to normal with no ophthalmoplegia. Histology confirmed a prolactinoma with a low Ki67 and no up-regulation of p53.A 15 year old boy initially presented with visual deterioration, galactorrhoea and weight gain to another neurosurgical unit. MRI showed a large pituitary tumour. He had a prolactin of 67,000 miU/L and was managed conservatively with cabergoline. His follow-up showed evidence of tumour reduction and prolactin reduced to ~ 10,000 miU/L. He presented to our unit almost a year later with sudden onset severe frontal headache and significant bi-temporal hemianopia. His MRI showed an enlarged apoplectic pituitary tumour with considerable chiasmal compression. Given the severity of his visual deterioration he underwent emergency ETSS. Post operatively his vision improved but recovery was complicated by transient diabetes insipidus, which settled over a few days. Histology confirmed pituitary apoplexy within a prolactinoma. No mitotic activity was seen.A 13 year old girl presented with a 6 month history of headache and right temporal hemianopia. Her family were also concerned that this previously very studious girl was struggling at school. Her MRI showed a suprasellar apoplectic tumour with a prolactin of 30,824 miU/L. She was started on a dopamine agonist and monitored as an outpatient. During this time her prolactin returned to normal and she was monitored with regular visual field testing and monitoring of her prolactin. After a year she presented again with deteriorating vision. MRI showed an enlarging apoplectic tumour despite a prolactin of 63 miU/L. She therefore underwent ETSS to prevent any further visual loss. Post-operatively, both her vision and her academic performance have returned to normal. Histology confirmed a prolactinoma with apoplexy. The Ki67 and p53 labelling were not elevated.All the children were admitted to our specialist neurosurgical ward within a tertiary children’s hospital and reviewed by a paediatric endocrinologist and a paediatric neurosurgeon. A full endocrine profile was performed as shown in Table All the patients had an urgent MRI scan to help evaluate their condition and in all cases this confirmed the diagnosis of pituitary tumour apoplexy.Figure Blood products of various ages within the mass are shown in child 4 and 5, where there is clear layering in the lesion Figs. , 3, 4.In all cases the patients had pituitary surgery to preserve visual function. Surgery was performed via an endoscopic trans-sphenoidal approach using the “two nostril, four hand” technique by a paediatric neurosurgeon and otorhinolaryngologist. Following surgery, a MRI scan was performed within 24 h as a baseline—Fig. The clinical syndrome of pituitary tumour apoplexy is a medical emergency and requires life saving rapid replacement with hydrocortisone . The decIn our adult practice, pituitary tumour apoplexy is more commonly seen within non-functioning adenomas. In this paediatric series, however, all the children presented with prolactinomas.It is now widely recognised that a conservative approach using dopamine agonists (DA) should be used first line in the vast majority of adult prolactinomas presenting with apoplexy, due to a similar efficacy of surgery to conservative management, in terms of visual outcome , 10. In In those who do not respond satisfactorily to conservative management or present with severe visual compromise, surgery may be required. Visual defects should still improve even when delay between onset of symptoms and surgery is long . It has In our series only one of the five children was already known to have a pituitary macroadenoma (Child 4), the other four children all presented initially with an apoplectic event. One child (Child 5) presented twice with apoplexy, with only the second event requiring surgical intervention.Pituitary tumour apoplexy in the adult population can also be managed conservatively with surgery being used as a second line treatment for visual compromise. During a similar period, 61 adults presented with apoplexy, 37 (61%) acutely requiring admission and 24 as an out-patient. Only 12 (32%) of the patients admitted to our adult specialist centre needed surgical intervention and in all but one case the underlying tumour was a non-functioning adenoma.Despite dopamine therapy, our entire paediatric cohort required surgery, either as an emergency or delayed procedure for visual compromise. Histologically, all the children had prolactin-secreting tumours. Proliferation markers showed a low Ki67 in all except Child 2 (moderate ~ 5%) and no up-regulation of p53. Despite the benign histological features the clinical course in our series suggests a more aggressive condition in comparison to adults.There is no published consensus on how frequently the prolactin level should be checked. There is an argument for annual prolactin levels and repeat imaging only if there is a marked increase in prolactin (> 250 µg/L) or clinical signs of tumour expansion .Although elevated prolactin levels can be secondary to the ‘Stalk effect’ where an expanding mass can interrupt dopamine delivery from the hypothalamus to the pituitary, resulting in loss of inhibition of prolactin release , levels Only two children (40%) had normal pituitary function pre and post operatively (Child 1 and 3), the other patients all showed some degree of anterior pituitary dysfunction. Child 2 was found to have panhypopituitarism pre-operatively and remained on GH, thyroxine, testosterone and hydrocortisone post-op. Child 4 was found to be deficient in GH, thyroxine and testosterone. Child 5 required GH, thyroxine and hydrocortisone on discharge. This need for hormonal replacement is similar to the adult population.From an imaging perspective, our case series correlates with the literature related to pituitary tumour apoplexy in the adult population. Typically, non-haemorrhagic, non-cystic adenomas are isointense to grey matter on T1 and T2 weighted imaging . As pituWe identified no specific imaging characteristics in the paediatric population that differ from that of adults. All five of our cases demonstrated macroadenomas of 2 cm or greater, several of which had the classic ‘snowman’ appearance as the lesion extends cranially. Child 4 also showed extensive right parasellar extension as well as suprasellar extension, with displacement of the right internal carotid artery. All five lesions caused compression of the optic chiasm.Two of our patients (Child 4 and Child 5) were started on growth hormone 2 weeks prior to the onset of apoplexy. Although this is not enough evidence for causation, with so little known about precipitating factors to apoplexy, it does merit focus for the future. We believe our data represents the first identification of this potential relationship between starting GH and the onset of apoplexy.The low incidence of pituitary tumour apoplexy in children mean there is little published literature surrounding this topic.Our case series suggests that paediatric pituitary tumour apoplexy is more likely to be seen in adolescents with prolactinomas over 2 cm in size.We believe our case series suggests that paediatric pituitary tumour apoplexy has a more aggressive natural history in comparison to adults and that early surgery may be more appropriate in this cohort of patients.


While LAGB has become uncommon in the bariatric surgery practice, band removal with or without revision surgery is still common. Retained postoperative foreign body, of which surgical sponges are the most common, is a rare condition. We report a rare case of retained gastric band port and the attached tube.A 31-year-old Caucasian female presented to the outpatient clinic, 5 years after her last surgery, complaining of a left upper quadrant abdominal mass over the last 2 years. She had a history of 2 weight loss operations. She had no significant family history nor smoking. CT of the abdomen and pelvis revealed a retained foreign body. On exploration, the port with 10 cm of the connected tube was found and removed through a small incision without laparotomy. The patient made an uneventful recovery.A bariatric surgeon should be involved in the evaluation of any patient who complains of abdominal pain and/or palpable mass if she/he has a previous weight loss procedure because the bariatric surgeon is fully aware of the possible complications of the bariatric surgeries. While LAGB has become uncommon in the bariatric surgery practice, band removal with or without revision surgery is still common. Surgeons performing band removal should be aware of the possible pitfalls and complications of this procedure .Pouch enlargement, band slippage, band erosion, port-site infections and port breakage represent the complications most commonly associated with LAGB . ProceduA 31-year-old Caucasian female presented to the outpatient clinic five years after her last surgery complaining of a left upper quadrant abdominal mass which is painful on movement, the mass size increased gradually over the last 2 years. She had no fever, diarrhea/constipation or nausea/vomiting.She had a history of LAGB 10 years ago. Five years later she had a revision surgery due to weight loss failure, the gastric band was removed and laparoscopic Roux-en-Y gastric Bypass was done in the same procedure. Her past medical history included hypothyroidism 13 years ago medically treated by a daily dose of L-thyroxine. She had no significant family history nor smoking.On examination, the patient was afebrile. The abdomen was soft and non distended, the surgical scars were healed. A 4 × 4 cm, symmetric mass with normal overlying skin was found in the left upper quadrant. This mass was spherical, superficial, tender, firm, mobile and didn’t disappear by compression. There was no bruit or lymphadenopathy.Laboratory findings including complete blood count, liver function tests, and renal function tests were within normal. The differential diagnosis was port site hernia or retained foreign body.Further investigations included CT of the abdomen and pelvis with oral contrast revealed subcutaneous spherical foreign body (probably the port) with the connected tube extending 10 cm into the abdominal cavity (Fig. Gastric band removal is a common procedure . HoweverCattanach et Teague  reportedStarnes et al. reported a case of retained gastric band tube . Felder Gastric band removal and laparoscopic Roux-en-Y gastric bypass can be a prolonged procedure when there are adhesions. Moreover, the surgical team becomes exhausted at the end of the procedure which may lead to such mistakes. That was the case in our patient where the procedure time was 210 min, the retained foreign body was the port with the connected tube discovered 5 years after gastric band removal and laparoscopic Roux-en-Y gastric bypass.The main complaint of the patient was a left upper quadrant abdominal mass increased in size over the last 2 years. CT of the abdomen and pelvis could detect a retained foreign body and that was the only necessary diagnostic investigation.On review, the removal of the gastric band port wasn’t mentioned in the patient’s medical record so we would suggest a checklist to be a part of the surgical notes of gastric band removal procedures.The possibility of a retained foreign body should be in the differential diagnosis of any postoperative patient who presents with pain, infection, or palpable mass .From a medico-legal standpoint, the surgical team should be fully aware of the consequences of the retained foreign bodies. Therefore, a double check should be done to avoid such cases. Fortunately, the patient didn’t claim against the responsible surgeon for retained gastric band port but that doesn’t apply to every patient.Detailed history and full clinical examination in addition to a high index of suspicion are mandatory to evaluate patients who have retained foreign body. Hospitals also should develop strategies for LAGB removal procedures to avoid medico-legal issues. Finally, a bariatric surgeon should be involved in the evaluation of any patient who complains of abdominal pain and/or palpable mass if she/he has a previous weight loss procedure because the bariatric surgeon is fully aware of the possible complications of the bariatric surgeries.


Abdominal hemangiolymphangioma (HLA) in neonates is a rare condition that demands surgical intervention after a complete preoperative diagnostic approach. The differential diagnosis and the establishment of a therapeutic algorithm is a challenge, both for the neonatologists and the pediatric surgeons, because there is no consensus in the management of HLAs in infancy according to the literature. We report a rare case of abdominal HLA in a female newborn that was admitted to our pediatric surgery department with a prenatal diagnosis of an abdominal cystic tumor. After a thorough preoperative diagnostic approach, the neonate underwent an explorative laparotomy and lesion excision with simultaneous splenectomy due to the operative findings. The neonate had an uncomplicated postoperative period and is free of recurrence a year after. Only the pathology examination can reveal the HLA diagnosis. When a total surgical excision is evitable, a close follow-up follows an uncomplicated postoperative hospitalization. Abdominal tumors in neonates are a challenging issue for pediatric surgeons because of the crucial need for appropriate surgical approach. Infantile lymphangioma and hemangioma (hemangiolymphangioma-HLA) is a rare mixed vascular tumor with both endothelial and lymphatic elements, which belongs to the family of mixed vascular malformations , 2. ApprAn otherwise healthy female neonate, the first child of a healthy couple, born with caesarian section at 38 + 4 weeks of gestation with a birth weight of 3200 g, was admitted to our department on the 3rd day of life for evaluation and management of a prenatally diagnosed cystic abdominal mass.Medical history began at 22 weeks of gestation, when a level-2 ultrasound raised the suspicion of a possible small intestine obstruction. A week later, an embryonic MRI revealed a 25 × 30 × 44 mm cavitary cystic mass with internal hemorrhagic elements. The mass was located in the subdiaphragmatic area, anteriorly but separate from the left kidney, the spleen, and the stomach. Echographic reevaluation at 29 weeks of gestation identified a slight enlargement of the mass (27 × 35 × 59 mm) that persisted during a second embryonic MRI 2 weeks later see .After birth, a postnatal abdominal ultrasound performed at day 1 of life confirmed the diagnosis and the family was referred to us for further consultation and treatment.β-hCG, AFP levels, and urine VMA levels. A new abdominal MRI was performed at day 9 of life and depicted an enlargement of the lesion (37 × 32 × 70 mm), while bone scan and 123I-MIBG scan were normal . At the moment, 1 year after the operation, the child remains healthy and asymptomatic. Follow-up included regular abdominal ultrasounds that did not reveal any recurrence.Abdominal infantile hemangiolymphangioma is an extremely rare condition that calls for a detailed surgical approach because of the risk of a fatal massive bleeding .When concern of an intraabdominal cystic tumor is raised during prenatal ultrasound, an embryonic MRI is proposed as the next step of the diagnostic algorithm. Postnatal abdominal ultrasound and possibly abdominal MRI can confirm the diagnosis.The differential diagnosis of a newborn with a cavitary abdominal cystic tumor includes cystic lymphangioma, hemangioma, ovarian cyst, cystic teratoma, cystic lesions of the kidney, and cystic neuroblastoma , 9. The In our case, abdominal HLA was suspected preoperatively and confirmed during histological analysis of the resected specimen that showed both hemangiomatous and lymphangiomatous components, while neither mitotic activity nor necrosis was present.Etiology and pathogenesis of HLA are not elucidated in the literature. The coexistence of these two pathological entities may be related to abnormal development of the lymphatic structures during the embryonic period .There is no consensus in the management of HLAs in infancy. Main therapeutic approaches include the “wait-and-watch” approach because of the high probability of tumor remission until the age of 18 to 24 months and the surgical approach with complete excision of the lesion. Remission-helping drug components, sclerotherapy, and embolism are described in the literature as alternative pathways –15.When surgical excision is of low risk as in our case, and due to the possibility of mass bleeding, infection, and respiratory distress due to enlargement and space occupation, we prefer the surgical approach in similar cases. We emphasize that each medical case is unique and demands a unique therapeutic approach which should be individualized depending on the size of the lesion, anatomic localization, and possible complications.Αbdominal HLA in infancy is an extremely rare condition that demands an adequate surgical intervention because of the high risk of fatal complications. Only the histolopathological examination can reassure the disease. If the prenatal ultrasound raises the concern of an intra-abdominal cystic tumor, an embryonic MRI is proposed as the next step of the diagnostic algorithm. There is no consensus in the management of HLAs in infancy. In our pediatric surgical department, we choose the surgical approach in similar cases as analyzed above. When a total surgical excision is evitable, a close follow-up follows an uncomplicated postoperative hospitalization.


Geographic stomatitis is an uncommon inflammatory condition of unknown etiology. It is characterized by reddish areas surrounded by white borders affecting any location in the oral cavity and presenting a migratory and cyclic pattern. The most common affected sites include buccal mucosa, labial mucosa and mucobuccal fold. Some patients can complain of pain or burning sensation. There are few reports in the literature about this entity and its relationship with other oral and cutaneous conditions such as fissured tongue, Reiter’s syndrome, atopy and psoriasis has been suggested but it is still controversial. In the present study we describe three cases of geographic stomatitis associated with fissured tongue. Lesions involved the buccal mucosa, labial mucosa, soft palate and mucobuccal fold and all cases were diagnosed based on their clinical features. All patients were oriented about the innocuous behavior of the condition and were advised to avoid exposure of the lesions to irritation factors. The three presented cases highlighted the importance of a detailed oral mucosal examination by clinicians and provided further information about the natural history and clinical presentation of geographic stomatitis. Key words:Geographic stomatitis, geographic mucositis, geographic tongue. Geographic stomatitis (GS) (or Migratory stomatitis) is an uncommon oral condition that was first described and recognized as “Eritema migrans” in 1955 . GS is c-Case 1A 33 year-old male, with previous diagnosis of cutaneous lichen planus was referred to evaluation of oral painless erythematous areas detected during dermatological routine examination. Intraoral examination showed multiple reddish patches circumscribed by a discrete whitish border in the soft palate and well-defined reddish patches limited by yellowish-white borders in the dorsum of the tongue and fissured tongue Fig. A,B. The -Case 2A 14 year-old female was referred for evaluation of an intraoral burning sensation lasting few months. Medical history revealed no past/present local or systemic mucocutaneous disorder. Intraoral examination showed fissured tongue associated with extensive reddish well-defined patches surrounded by a whitish irregular border affecting the tongue and also the lower labial mucosa, muccobuccal fold and buccal mucosa Fig.  A,B,C. C-Case 3A 70 year-old male was referred for evaluation of a burning sensation in the mouth lasting a few months. Medical history revealed mild arterial hypertension controlled with losartan, moderate alcohol consumption and no past/previous history of any mucocutaneous disorder. Clinical oral examination revealed fissured tongue and reddish patches surrounded by whitish irregular borders in the tongue, and buccal and labial mucosa Fig. A,B,C. ClAlthough GS can affect all anatomical locations in the oral cavity, the gingiva, hard palate and floor of mouth seems to be the less affected sites ,7. When Diagnosis of GS relies on clinical signs and symptoms and natural history of the condition, as shown by the presented cases ,6. HowevThe exact etiology of GS and geographic tongue is still unknown, but the latter has been associated with some oral and cutaneous conditions, such as fissured tongue (as seen in the present cases), Reiter’s syndrome, atopy and psoriasis ,4,7,8. SAccording with Hume , geograpIn conclusion, it is important that oral clinicians and medical professionals are aware of oral mucosal conditions that can be potentially associated with mucocutaneous diseases, both for early diagnosis and prompt referral of the patient to an Oral Medicine service when necessary. GS is a rare condition that can be under diagnosed or misdiagnosed in daily practice. Additionally it is possible that, similarly to geographic tongue, GS can be associated with systemic and mucocutaneous disorders, such as psoriasis. The present report suggested that the co-existence of GS and geographic tongue with systemic and mucocutaneous disorders should be always reported to provide information on their possible association. A modified classification based on Hume´s classification and including the above mentioned parameters was also suggested.


Osteochondromas commonly affect the proximal humerus, pelvis, and knee but are rarely seen on flat bones. Herein, we present the case of a 15-year-old female patient with osteochondroma located at the dorsal aspect of the scapula. The patient was admitted to the Orthopedics and Traumatology Department with the complaint of a mass on the left upper back for five years. The patient complained of the inability to sleep in the supine position, pain with shoulder motion, and cosmetic discomfort for two years. X-rays of the left shoulder revealed a bony mass arising from the dorsal aspect of the left scapula. The patient underwent an operation, and a specimen was sent for histopathologic examination. The histopathologic investigation confirmed the diagnosis of non-malignant transformation osteochondroma. While osteochondroma is not common in the scapula, it should be kept in mind that the most common benign tumor of the scapula is osteochondroma. Osteochondroma is the most common type of benign bone tumor arising from the surface of a bone. Osteochondromas commonly affect the proximal humerus, pelvis, and knee but are rarely seen on flat bones . HoweverMany cases of osteochondroma with ventral location causing pseudo-winging and snapping scapula have been reported in the literature -8. In coHerein, we present a case of osteochondroma located at the dorsal aspect of the scapula.A 15-year-old female patient was admitted to the Orthopedics and Traumatology Department with the complaint of a mass on the left upper back for five years. The patient complained of the inability to sleep in a supine position, pain with shoulder motion, and cosmetic discomfort for two years. She reported that there had been no growth in the size of the mass in the last three years. The patient did not have a history of trauma, family history, a history of osteochondroma in another region, weight loss, systemic signs, or any other known disease. On examination, a hard, oval solid mass was palpable with a size of about 6×4 cm on the superior-medial dorsum of the scapula Figure . A pedunUnder general anesthesia, the patient was placed in the prone position. After cleaning the operation field with iodine, a sterile drape was applied. An incision was made over the mass. The soft tissues were dissected carefully and the borders of the mass were exposed Figure . The pedOsteochondromas are benign tumors that commonly affect the proximal humerus, pelvis, and knee but are rarely seen on flat bones . They arThere have been some cases of solitary osteochondroma of the scapula reported in the literature. In most cases, the affected side was the volar aspect of the scapula ,3,5-8. SNathan et al.  performeYadkikar and Yadkikar  presenteIn another case presentation, Jadhav et al.  presenteNekkanti et al.  presenteOsteochondromas may cause different symptoms depending on their localization. While osteochondroma is not common in the scapula, it should be kept in mind that the most common benign tumor of the scapula is osteochondroma. Although osteochondroma is more common in the ventral region and causes clinical snapping scapula, the dorsal involvement of the osteochondroma may also cause clinical symptoms depending on the size of the mass.


We present a unique case of intercostal muscle flap (ICMF) ossification mimicking an intrathoracic rib diagnosed 3 years after oesophageal perforation repair. A 58-year-old male presented with complaints of mild chest discomfort. Three years ago he had undergone left thoracotomy and primary repair of post-emetic oesophageal perforation. An ICMF had been used to strengthen the repair. Chest X-ray identified a linear calcific density within the left hemithorax. Subsequent thoracic CT characterized the anomaly as ossification of the ICMF. The lesion had the appearance of a well-differentiated intrathoracic rib coursing through the left lower lobe. We discuss the typical appearances of ossified ICMFs and the potential complications resulting from this ossification. Such ossification can have a variety of appearances on CT and can result in complications such as anastomotic stricture and ischaemia. Surgical technique has been shown to determine whether an ICMF ossifies.4Although ossification of intercostal muscle flaps (ICMFs) has been reported in the literature,Three years before the present findings, a 58-year-old male had been referred to the thoracic surgery department at Toronto General Hospital with acute onset of post-emetic sharp central back pain. On admission the patient’s vital signs showed tachycardia, hypotension with a systolic blood pressure of 90 mmHg and a fever of 38.4 °C.On thoracic CT angiogram performed to exclude aortic dissection, free air was identified in the posterior mediastinum adjacent to the distal oesophagus. Subsequent Gastrografin oesophagography revealed a small contained leak at the distal oesophagus. Left thoracotomy was performed. When entering the chest the left sixth intercostal muscle bundle was mobilised and after primary repair of the perforation this ICMF was sutured over the repair acting as a buttress.The patient recovered well post-operatively and was discharged after 12 days. The initial post-operative follow-up was uneventful. Nevertheless, 3 years later, the patient was referred back to the thoracic surgery clinic by his family doctor because of mild chest discomfort.Chest radiograph was first performed and showed a linear calcific density within a band of soft tissue density in the left hemithorax . CompariThe patient’s mild discomfort did not hinder the activities of his daily life. He was discharged from the thoracic surgery clinic with the advice to return should his symptoms worsen.5 Compared with pleural or pericardial flaps, which have limited vasculature, the ICMF is easily mobilized within the thoracic space and its potential for neovascularisation ensures long-term viability.8 The use of ICMFs is also indicated in the repair of tracheoesophageal fistulas, buttressing of tracheal and bronchial anastomoses, repair of bronchopleural fistulas and for reconstruction post intrathoracic cancer resection.11Post-emetic oesophageal perforation is uncommon and has a high mortality rate if untreated. Primary repair with the use of a pedicled ICMF as a buttress has been described in the literature, as local oedema, necrosis and inflammation could compromise the site of primary repair.1 Therefore, calcification and ossification of the muscle flap is a known post-operative phenomenon.10 Different surgical techniques for the preparation of ICMFs have been associated with varying degrees of ossification post-operatively. In most studies, periosteum from both adjacent ribs is harvested together with the ICMF. On follow-up, the majority of these exhibited some degree of calcification or ossification.10 Cerfolio et al4 described a technique in which the ICMF was harvested utilising cautery instead of the subperiosteal approach for bronchial buttressing and none of the 301 cases described at 6-month follow-up exhibited any ossification.The ICMF is composed of intercostal vessels, nerves, skeletal muscle and frequently periosteum of the adjacent ribs, which includes pluripotent mesenchymal cells that have the potential to develop into osteoprogenitor cells.2 performed a retrospective review on the radiological records of 23 patients in whom an ICMF was utilised for bronchial stump reinforcement post lung resection. At follow-up, all ICMFs showed discontinuous linear calcifications either appearing as a single narrow stripe resembling a surgical staple line, or as two parallel stripes with a lower attenuation central portion, resembling a rib. Calcification was shown as early as 1 week post-operatively and reached a maximal density at a mean of 5 months.Kwek et al3 If this technique has been utilised, bronchial anastomotic stricture should be excluded on post-operative CT scans. A consequence of this complication is recurrent pneumonia of the affected lung or lobe. If the underlying structure is the oesophagus, stenosis can be excluded on barium oesophagography or thoracic CT with oral contrast.Ossification has been reported to cause bronchial anastomotic stricture or tracheal stenosis if the ICMF is wrapped circumferentially around the underlying structure.This case illustrates ICMF ossification post oesophageal perforation repair. On CT, ossification of the flap in this patient resembled an intrathoracic rib arising from the posterior left sixth intercostal space, coursing through the left lower lobe to the distal oesophagus.Ossification of ICMFs is a recognised phenomenon when the periosteum of adjacent ribs is removed together with the flap.This can have several appearances on CT, the most common being a single narrow high-density stripe within the flap or two parallel high-density stripes with a lower density central portion (mimicking a bone).Ossification of ICMFs is usually an incidental finding with no clinical consequences.If the ICMF is wrapped around a bronchus, the trachea or the oesophagus, ossification may result in a stricture, which should be excluded on imaging.Written informed consent was obtained from the patient for publication of this case report, including accompanying images.


An 11-year-old female spayed domestic shorthair cat was referred with a2-month history of ptyalism, hyporexia and weight loss. Physical examinationrevealed reduced body condition score (2/9) and decreased skin turgor.Laboratory abnormalities included mild erythrocytosis, elevated creatinekinase, hypercobalaminaemia and hypofolataemia. CT of the head and abdominalultrasonography were within normal limits. Gastroesophagoscopy revealedmucosal ulceration and possible stenosis of the distal oesophagus. Thoracicradiographs and iodine oesophagram showed a soft tissue opacity in thecaudodorsal thorax compatible with a parietal oesophageal mass causingluminal stenosis or an extra-parietal mass causing ventral displacement andcompression of the oesophagus. Pulmonary nodules were observed in thecranial lung lobes. CT of the thorax confirmed the oesophageal origin of themass and the presence of pulmonary nodules scattered throughout the lungparenchyma. The patient was euthanased given the imaging findings andperceived guarded prognosis. Post-mortem examination revealed multifocalnodular lesions affecting the oesophagus, lungs, kidneys and pancreas.Histopathological examination identified atypical round cells characterisedby eosinophilic cytoplasm and pale nuclei with prominent nuclear grooves,compatible with neoplastic histiocytic cells. Immunohistochemistry revealedstrong expression for CD18, Iba-1 and vimentin. Transmission electronmicroscopy demonstrated intracytoplasmic organelles consistent with Birkbeckgranules of Langerhans cell origin in lesional histiocytes. These findingswere compatible with a diagnosis of disseminated Langerhans cellhistiocytosis.To our knowledge, this is the first report of disseminated Langerhans cellhistiocytosis with oesophageal involvement in a cat. Ptyalism wasintermittent and had no association with feeding. Empirical treatment with cefovecin, meloxicam  and famotidine  was notbeneficial. On presentation the cat was bright and responsive, weighed 2.5 kg (bodycondition score 2/9) and had lost 500 g weight over the previous month. Physicalexamination revealed a mild generalised muscle atrophy. Oral and thyroidexamination, thoracic auscultation and abdominal palpation were normal. A 6%dehydration was estimated based on a mild loss of skin elasticity.Gastrointestinal disease was suspected. Complete blood count and serum biochemistryidentified mild erythrocytosis  and elevated creatine kinase . In-house testingfor feline immunodeficiency virus and feline leukaemia virus were negative. Agastrointestinal panel revealed mild hypercobalaminaemia and hypofolataemia . AmmoniaThe owner was offered a CT of the head and thorax of the cat to better assess forpossible hidden dental/oral disease, and assess the oesophagus, respectively,followed by abdominal ultrasound and an upper gastrointestinal endoscopy toinvestigate oesophageal and gastrointestinal disease. Abdominal ultrasound waschosen over CT to better assess the gastrointestinal wall layering. The ownerinitially declined the thoracic CT owing to financial concerns, given the absence ofrespiratory signs and because the oesophagus would be assessed during endoscopy.CT study of the head  with contrast medium and abdominal ultrasound showed nosignificant abnormalities. The upper gastrointestinal endoscopy  was suggestive of stenosis in the distalthird of the oesophagus with a focal area of ulceration in the mucosal surface. Thestomach and duodenum appeared macroscopically normal.Given the suspicion of oesophageal stenosis, thoracic radiographs were obtained. They shTo further investigate the origin of the mass, an iodine oesophagram was performedusing an oesophageal tube. Cranial to the mass, the oesophageal lumen was distendedwith diluted contrast medium . At the level ofthe mass, the lumen was slightly narrowed but patent. On the lateral views, the masswas seen surrounding the oesophageal lumen dorsally and ventrally . The dorCT of the thorax was performed. The mediastinum was markedly widened by awell-defined, homogeneously attenuating parietal oesophageal mass  extendinGiven the severity of clinical signs, diagnostic imaging findings and perceivedguarded prognosis, the owner elected for euthanasia. On post-mortem examination thedistal third of the oesophagus was severely enlarged due to the presence of acircumferential mass. This mass was firm and white, with evidence of mucosalulceration .SeveralHistopathological examination of the aforementioned organs revealed the presence of apopulation of round cells arranged in densely cellular sheets exhibiting distinctcell borders, and a moderate amount of eosinophilic cytoplasm with one oval,prominent, vesicular nucleus, which was frequently indented. Anisokaryosis andanisocytosis were moderate to severe. There were three mitoses per high-power field.High numbers of neutrophils and small lymphocytes were frequently associated withproliferating round cells. Immunohistochemical investigation was performed and theround cells showed strong expression for CD18 , Iba-1 and vimentin , and were negative forchromogranin A , CD20 , CD3 ,cytokeratin  and synaptophysin  .Selected formalin-fixed paraffin-embedded tissue samples were submitted fortransmission electron microscopy (TEM). Ultrastructural morphology was suboptimalowing to the formalin fixation and paraffin embedding; however, diagnostic featureswere recognised. Atypical histiocytic cells in multiple locations revealed evidenceof intracytoplasmic electron-dense bilaminar structures characterised with aninternal zipper-like pattern of striations consistent with BGs of LC origin.Occasionally, terminal bulbous structures were observed (tennis racquet morphology). Thesef12 Oesophageal involvement in disseminated LCH in cats has notbeen previously described. Reported cases have presented primarily with respiratorycomplaints, lethargy, anorexia or weight loss.9 In our case hyporexia, weightloss and ptyalism were the presenting clinical signs. While ptyalism is a commonclinical sign of oesophageal disease, pseudoptyalism from an involuntary disruptionof the swallowing mechanism is also a likely consequence of mechanical obstructionfrom the oesophageal mass.13Gastrointestinal involvement in LCH is rare in humans and oesophageal involvement ispoorly documented.9 In the present case, the milderythrocytosis was considered secondary to dehydration and the elevated creatinekinase due to oesophageal damage. Hypofolataemia was most consistent with decreasedintake. Hypercobalaminaemia has been associated with solid neoplasms in cats, andseems the most likely cause in the absence of supplementation.14There are no typical clinicopathological findings associated with LCH, and no knownassociation with retroviruses.9In this case, pulmonary nodules were detected within the cranioventral lung lobes.Peribronchial alveolar infiltrates on both sides of the mass in combination withreduced lung volume was interpreted as lung atelectasis. The anatomical origin ofthe caudodorsal thoracic mass could not be defined based on survey radiographsalone. It might have been originating from the caudal mediastinum or lungparenchyma. The iodine oesophagogram demonstrated the relationship between the massand the oesophageal lumen that was surrounded dorsally and ventrally by the mass.This might have been explained by a parietal oesophageal mass or a paraoesophagealmass. As oesophageal masses are rare in cats, the decision was made to perform CT ofthe thorax to define the anatomical origin of the lesion. CT was the definitiveimaging modality to define the anatomical origin of the mass and allowed a bettercharacterisation of the size and distribution of the pulmonary nodules.Thoracic radiographs of pulmonary LCH report mixed pulmonary patterns that arebronchointerstitial, alveolar or interstitial, with or without nodularopacification.9 Althoughlesional histiocytes exhibited similar cellular and nuclear descriptions ofpleomorphism, there are differences between the present case and reports ofpulmonary LCH. Pulmonary LCH has been characterised by cohesive infiltration ofhistiocytic cells, which obliterate terminal airways and extend to the pleuralsurface.9Accompanying inflammatory cells are dominated by lymphocytes, with fewer plasmacells and occasional macrophages.9 In our case a predilection for terminal airways was not observed andneutrophilic infiltration contributed significantly to the proportion ofaccompanying inflammatory cells.Infiltration of the affected organs by multifocal-to-coalescing nodular masses in thepresent case are similar to the characteristic feature described in pulmonaryLCH.15 Expression of Iba-1 has been determined as a useful marker for histiocyticproliferative, neoplastic and inflammatory disorders in cats, although it is unableto differentiate between macrophage and dendritic antigen presenting cells.16 In addition, the use of CD18 expressed on all leukocytes, as a histiocyticmarker, is dependent upon exclusion of lymphocyte differentiation as demonstrated inthis case.9 In keeping with previous reports of pulmonary LCH, lesional histiocytesexpressed vimentin, Iba-1 and leukointegrin CD18, alongside negative immunolabellingof cytokeratin and lymphoid differentiation antigens CD3 and CD20.9 Expression of CD1a and adhesionmolecule E-cadherin was not assessable; however, a diagnosis was achieved by theidentification of BGs on TEM, which are a hallmark of histiocytes of LCorigin.18Immunohistochemical profiles of histiocytic cells are a cornerstone in the diagnosisof LCH.Financial restraints were a limitation and influenced the diagnostic investigationsperformed in this case. Initially, performing an oral examination and thoracicradiographs after anaesthesia would have given relevant information to guide thecase, then followed by CT of the head and thorax to rule out hidden oral disease andbetter characterise the thoracic changes seen on radiography, respectively.Endoscopic biopsies of the affected oesophagus were obtained but not analysedbecause a full post-mortem examination was performed. Given that the neoplasticcells involved all layers of the affected oesophagus these biopsies would likely besuitable to reach a diagnosis in an ante-mortem setting.We present a novel case of disseminated LCH with oesophageal involvement in a cat.Feline LCH should be included in the differential diagnosis list of ptyalism andwhen there is a clinical index of suspicion of oesophageal disease. Ante-mortemdiagnosis and management of LCH in the cat remains undescribed and requires futurestudies.


Taking a systematic and comprehensive patient history is undoubtedly an essential tool for a proper diagnosis. We report a unique case of a patient presenting with persistent buccal swelling, a rare presentation of two unrelated pathologies occurring concurrently. In this case, the presence of a lipoma was concealed by a dental abscess and an incomplete patient history. Lipomas are benign tumors composed of a well-circumscribed mass of mature adipose tissue . AlthougA 53-year-old man sought treatment at the department of oral medicine and radiology at Sri Ramachandra University in Chennai, India, for a painful and progressive swelling involving the right mandibular region for four days. It was associated with a toothache on the right lower jaw. The pain in the tooth was dull, continuous, and aggravated on taking hot or cold beverages. The swelling was small when he initially noticed it and gradually increased to the presenting size. The patient had visited a private dentist a day before, where an orthopantomogram was taken, and he was advised to undergo extraction after a course of antibiotics and non-steroidal anti-inflammatory drugs. His medical history was noncontributory. Extra orally, a single, well-defined swelling was evident in the right lower jaw region, with signs of inflammation. The swelling was 4 cm x 5 cm, warm, tender, soft, compressible, and not fluctuant. Intraoral examination revealed dental caries in 47, with gross destruction of the crown and mucobuccal fold obliteration in relation to 47. A provisional diagnosis of dental caries in 47, with a dentoalveolar abscess, was promptly made. The previous orthopantomogram revealed radiolucency in 47 with gross destruction of the crown associated with radiolucency around the roots Figure . No otheGiven that the patient was asymptomatic, we extracted tooth 47 while he was under local anesthesia. The postoperative period was uneventful. When the patient was evaluated one week later, the asymptomatic swelling remained the same size Figure .There was no cervical lymphadenopathy and, intraorally, the mucosa was normal and moist Figure . The mouConsidering the history of painless swelling lasting one year that was well-defined, soft, nontender, with no intraoral manifestation, a lipoma of the right submandibular region was suspected. Fine needle aspiration cytology was performed under ultrasound guidance. Ultrasonography revealed a well-circumscribed hyperechoic mass measuring 3.2 cm x 2.7 cm separated from the right submandibular gland Figure .The smear showed mature fibro-adipose tissue, which would be consistent with a lipoma Figure .Surgical removal of the lesion was done by the submandibular gland approach under general anesthesia. The excised mass showed mature adipocytes, a few capillaries, and fibro-collagenous tissue. The physical features were again consistent with a lipoma Figure .A complete and accurate history is the basis for an effective care plan. Despite today’s medical advancements, a patient’s history remains the mainstay for diagnosis. This case presented a rare situation where two distinct, unrelated pathologies  coexisted, invariably making the clinical course complex. While the buccal aspect of the mandibular body remains an infrequent site for lipomas, its possible presence was masked by the unambiguous signs of odontogenic infection and an incomplete patient history, which naturally led to considerable uncertainty about the source of persistent swelling.Lipomas are benign tumors of soft tissue origin occurring from mature adipose tissue . It is tAccording to World Health Organization, benign adipocytic tumors are classified into “Lipoma, Lipomatosis, Lipomatosis of nerve, Lipoblastoma/lipoblastomatosis, Angiolipoma, Myolipoma of soft tissue, Chondroid lipoma, Extra-renal angiomyolipoma, Extra-adrenal myelolipoma, Spindle cell/pleomorphic lipoma, and Hibernoma” . The cauGiven that a lipoma is a soft tissue neoplasm, ultrasonography (USG) and magnetic resonance imaging (MRI) serve as valuable diagnostic tools. Although lipomas show variable echogenicity, they are usually hyperechoic, containing linear echogenic lines under USG . In T1-wHistologically, a lipoma is composed of mature adipocytes arranged in a lobular pattern usually surrounded by a thin fibrous capsule. Although they resemble normal fat cells, they are distinguished from normal adipocytes by the fact that they are not utilized for metabolism. Surgical excision remains the mainstay of treatment, and the recurrence rate is very low ,12.Our patient presented with dental pain and an abscess in the right lower jaw region, a site with a preexisting lipoma. The presence of the lipoma was concealed by the definite signs of a dental abscess. Though the features of lipomas are usually straightforward, systematic clinical examination and diagnostic workup are essential for successful management. We were fortuitous to handle both the abscess and the lipoma successfully, without causing discomfort to the patient. However, we may not always be so fortunate. Therefore, accurate and detailed information regarding a patient’s history is indispensable for optimal patient care.


A 64-year-old man consulted the emergency department with intermittent abdominal pain during the past 10 days. Clinical examination demonstrated tenderness in the right iliac fossa towards the inguinal region. Blood testing showed no signs of inflammation.Ultrasound (US) examination of the abdomen revealed a direct inguinal hernia protruding anteromedially and inferiorly to the inferior epigastric vessels. A small tubular structure compatible with the appendix was present within the hernia sac Figure , arrow. Complementary computed tomography (CT) of the abdomen confirmed Amyand’s hernia . Clinical symptoms can be misleading and more often resemble those of a (strangulated) inguinal hernia than the classic signs and symptoms of appendicitis. Sometimes a palpable inguinal mass may be present [Ultrasound is an excellent technique to evaluate the inguinal region and can be used to detect all types of inguinal hernias, including Amyand’s hernia. US is safe and cheap, but remains operator-dependent.Computed tomography (CT) can be used to confirm the diagnosis if necessary. At the same time, possible intra-abdominal complications like perforation and abscess can be ruled out, especially in a preoperative setting .When diagnosis remains unclear, surgery can both be diagnostic and therapeutic. Treatment depends on the surgeon’s personal choice. Removing a healthy appendix is subject to a medical debate which has not reached a consensus yet .


Testicular cancer represents 1% of all malignant tumors in men. About 95% of testicular cancers are germ cell tumors (GCTs). These can be divided into nonseminomatous GCTs (NSGCTs) and seminomas. NSGCTs include teratomas, yolk sac tumors, embryonal carcinomas, choriocarcinomas, and mixed tumors. Only 2–6% of testicular teratomas are pure teratomas. Pure teratomas can be subdivided into prepubertal and postpubertal. The prognosis is significantly different between these two age groups. Different from teratomas in ovary, the immaturity in a teratoma is not an indication of their biologic behavior; the age of the patient is of greater importance. Malignant transformation of teratoma occurs in only 3–6% of testicular GCTs. The most frequent transformed histologic types consist of rhabdomyosarcoma, adenocarcinoma, and primitive neuroectodermal tumors. We report a rare case of pure postpubertal testicular teratoma with a secondary somatic malignancy that was an incidental finding in a patient presenting with lower back pain and testicular torsion. Primary testicular tumors are rare, but they are the most common solid malignancy in men 20–35 years of age . Over thAmplification of chromosome arm 12p, such as isochromosome 12p, is nearly universal in TGCTs. The pathogenesis of TGCTs is complex. It is believed that they arise from premalignant intratubular germ cell neoplasia (IGCN), which in turn develops from failure of normal maturation of fetal gonocytes [Testicular teratomas have been divided into 2 groups: prepubertal and postpubertal . The preMalignant transformation of teratoma is rare, occurring in only 3–6% of TGCTs. The most frequent transformed histologic types consist of rhabdomyosarcoma, adenocarcinoma, and primitive neuroectodermal tumors [We report a rare case of pure postpubertal testicular teratoma with a secondary somatic malignancy that was an incidental finding in a patient presenting with lower back pain and testicular torsion.Patient is a 38-year-old white male with a past medical history significant only for schizophrenia, currently on Abilify. He presented to the emergency department at the University of Toledo Medical Center with lower abdominal and back pain  that had been intermittent for the past 2 weeks, with no particular inciting factor. On further questioning, he admits that a week earlier he fell and experienced sharp pain in his testicle. This pain was severe but subsequently subsided. Physical examination revealed height of 69 inches and weight of 220 pounds. His vital signs were within normal limits. The remaining physical examination was normal except for a firm enlarged left scrotum, which is nontender. Patient was given pain medications and imaging was ordered.Bedside duplex ultrasound showed unremarkable right testis with good Doppler flow. However, the left testis had minimal to no blood flow and was extremely heterogeneous with small fluid collection, which was concerning for testicular torsion.Testicular mass could not be excluded from the ultrasound study. Hence serum beta HCG, ALP, AFP were ordered, all of which were within normal range. In addition, CT of the chest, abdomen and pelvis with contrast showed left adrenal mass measuring a maximum of 1.9 cm, most likely a simple adenoma. There was no evidence of retroperitoneal or inguinal adenopathy. The left hemiscrotum showed enlarged contents with calcifications . A diagnOn gross examination, the testicle was enlarged , measuriMicroscopic examination of the mass revealed predominantly overt malignant spindled cells displaying pleomorphism, hyperchromasia, and increased mitosis, with extensive necrosis in the background . There wMalignant transformation of teratoma is defined as the transformation of a somatic teratomatous component of a germ cell tumor (GCT) to a nongerm cell malignant tumor . It was The most common examples of malignant transformation are sarcoma , carcinoma (adenocarcinoma and squamous cell carcinoma), primitive neuroectodermal tumor, as well as hematologic malignancies .A pitfall in histopathological diagnosis is the potential of misinterpreting atypical epithelial or stromal cells for malignant transformation. Davey et al. established that cytological atypia with no expansile growth and infiltration of the surrounding tissue is not enough to make the diagnosis. Carcinoma or sarcoma component should consist of an expansile component that is exclusively occupying at least one low-power field  , 11.Unlike conventional GCTs, which respond significantly to platinum-based chemotherapy with an estimated 90% survival, teratoma with malignant transformation is a very aggressive tumor that is resistant to chemotherapy and is usually metastatic at presentation . It was This case report signifies the importance of maintaining a broad differential with a testicular torsion presentation and includes proper histopathologic diagnosis and impact of early diagnosis on prognosis of testicular teratoma with malignant transformation.


Coxsackievirus, a common pathogen causing pediatric infection, typically causes a mild, nonspecific illness with low-grade fever, but can cause severe illness on rare occasions. Encephalitis is an uncommon but important complication of coxsackievirus infection. The magnetic resonance imaging (MRI) findings of coxsackievirus B3 (CVB3) encephalitis have not been reported in the literature, and there are few descriptions of MRI findings of encephalitis induced by coxsackieviruses of other serotypes. We report the MRI findings of CVB3 encephalitis in a two-year-old girl, review the MRI findings in previously reported cases of pediatric coxsackievirus encephalitis, and present the MRI characteristics of coxsackievirus encephalitis. Coxsackievirus is a single-stranded RNA virus and belongs to the family Picornaviridae and the genus Enterovirus, which also includes poliovirus and echovirus. Coxsackieviruses are divided into group A and group B viruses. At least 23 serotypes of group A viruses and six serotypes of group B viruses are recognized. Most coxsackievirus infections are asymptomatic or result in only mild illness, such as nonspecific febrile illness, rashes, or mild upper respiratory tract infections, but they can also cause serious diseases such as pericarditis, myocarditis, pancreatitis, or encephalitis. Because encephalitis is a rare presentation of coxsackievirus infection, there is very limited information on the MRI findings of coxsackievirus encephalitis in the literature. We present a case of CVB3 encephalitis in a two-year-old girl and compare the MRI findings to previously reported cases of coxsackievirus encephalitis and other types of enteroviral encephalitis.A two-year-old girl had a fever up to 40°C for six days before admission. She had no cough, rhinorrhea, vomiting, diarrhea, or abdominal pain. She had previously been treated with amoxicillin and cefixmycin prescribed by a local medical doctor for five days, but a low-grade fever persisted. On admission, the results of the hematological and biochemical studies, urinalysis, and adenovirus rapid test were normal. Two days after admission, conscious disturbance was observed. Analysis of cerebrospinal fluid (CSF) revealed white blood cells of 144/μl , but protein and glucose were within normal limits. Brain MRI revealed hyperintense lesions in the midbrain, dorsal pons, and cerebellar dentate nuclei on fluid-attenuation inversion recovery (FLAIR) and T2-weighted images (T2WI) (Figure To the best of our knowledge, this is the first report demonstrating the MRI appearance of CVB3 encephalitis. To date, there have been four cases in three reports describing the MRI appearances of pediatric coxsackievirus encephalitis in the English-language literature 23. The p2The brain MRI in our case revealed abnormal signal intensity in the midbrain, dorsal pons, and cerebellar dentate nuclei. This MRI manifestation is strikingly similar to those of EV71 encephalitis reported by Shen et al.  and HuanHowever, in three cases of coxsackievirus encephalitis (cases 1, 2, and 4; Table Based on our review, the MRI manifestations of pediatric coxsackievirus encephalitis are age-dependent and similar to those of enteroviral encephalitis caused by other serotypes. Two patterns of involvement are recognized. In neonates, the lesions are distributed bilaterally in the cerebral white matter, without brainstem involvement. Characteristic brainstem involvement, most commonly in the posterior pons, are observed in older children. Although a definitive diagnosis of coxsackievirus encephalitis needs virus identification, familiarity with MRI features is helpful in guiding the management of these patients.


Idiopathic brachial plexus neuritis or neuralgic amyotrophy is a rare neurological condition whose true etiology currently remains unknown. Epidemiologically, the incidence of this condition is exceptionally rare with only 1.6 cases for every 100,000 people . Symptoms present an initial acute and sudden pain to the shoulder girdle and upper arm which is followed by a sense of profound weakness and numbness to the upper arm . Localized neuropathy within the arm-pit region may also be presented. The pain often exacerbates upon movement of the shoulder. Due to the anatomic location affected and the nature of the clinical symptoms presented, accurate diagnosis of brachial plexus neuritis poses a challenging diagnostic task for physicians due to remarkably similar symptoms expressed by differential diagnoses.Here, we report the case of a 55-year-old woman who underwent surgery entailing cervical laminectomy with instrumented fusion. She presented with postoperative symptoms of severe pain in the left arm with significant weakness within 24 h after surgery. A diagnosis of brachial plexus neuritis was made based on the symptoms presented and upon review of imaging scans.After a six-month follow-up visit, the patient recovered from the brachial neuritis but has residual numbness in the hand. The presentation of this case serves to transmit three fundamental purposes. First, this case serves to establish an intriguing possible association of the post-surgical period of cervical laminectomy with acute brachial neuritis and signifies the importance post-operative linkage with brachial neuritis in general. Second, this case also highlights the importance of close clinical monitoring of patients with unique symptoms within the postoperative follow-up period to ensure successful improvement and accurate diagnosis.As an underdiagnosed and relatively obscure condition, this case serves as an imperative reference for physicians to illuminate differential diagnosis of similar symptomatic conditions and also to promote knowledge of brachial plexus neuritis which can lead to an early and precise diagnosis. Brachial neuritis is a form of peripheral neuropathy of the brachial plexus. The condition was originally described by Drs. Parsonage and Turner after they began to see an abnormal pattern of cases during army work for the United Kingdom and India Command . Althoug22.1A 55-year-old woman presented with neck pain for many years since a motor vehicle accident twenty-one years ago. Additionally, patient presented with paresthesia and pain in the right arm with a burning sensation near the shoulder girdle. Intermittent numbness in the toes and bilaterally hands was also reported.2.2Physical examination reports revealed restricted range of motion of the neck when turning the body to the left. Long tract signs were positive for Babinski and Hoffman signs with hyperreflexia in the lower extremities and the right arm. Weakness was presented in the right wrist extensor and the right hip flexor.A magnetic-resonance-imaging scan  showed d2.3Surgical intervention included cervical laminectomy from C3 to C7 with lateral mass fusion from C3 to C7. Neurophysiological monitoring (SSEP and EMG) was stable throughout the duration of the surgical procedure, satisfactory signal response was also obtained from all four extremities before and during the surgery. No complications developed during surgery and in the recovery room the patient was conscious and neurologically unchanged as preoperative exam.2.4Twenty-four hours following the surgical procedure, the patient presented with significant pain in the left para-cervical and shoulder area going into the arm. There was numbness in the entire arm in no particular dermatome. The next day she had more pain in the left arm with newly developed weakness in the deltoid, biceps, triceps, wrist extensor, grip and intrinsic muscles. The patient had difficulty using the arm both proximally and distally. An MRI was conducted post-operatively which shows the well decompressed cervical spine . An MRI 3Acute brachial neuritis is a rare and under-recognized diagnosis for post-operative cervical pain. Etiology of brachial neuritis is currently unknown, however, previous medical literature suggests that possible influences could be auto-immune, post-vaccination, post-surgical or genetic ,17,5,18.Although a link to the site of surgery has not been established, this report allows for a greater illumination to display a possible correlation between site of surgery and BN. Post-surgical brachial neuritis is typically seen to emerge several weeks to months following surgery, however, in this documentation the onset of brachial neuritis was observed within 24 h of surgery with the site of surgery being dominantly within the cervical area . This isThe novelty of this report rests partly in the premise of the distinctive timing and potency of the symptoms presented. The literature indicates a former report where a young boy was diagnosed with brachial neuritis 72 h post-operatively, following subcutaneous mastectomy . This prAccurate and timely diagnosis of brachial neuritis can become a challenging task for physicians. There are several other similar conditions including ulnar nerve neuropathy, cervical radiculopathy, adhesive capsulitis, shoulder arthritis, and calcifying tendonitis ,18. It iSurgical trauma has been well-documented influence for post-surgical complications and could be attributed to influencing factors for such cases . HoweverA review of the medical literature indicates that post-operative brachial neuritis may be triggered by an auto-immune influence after response of the immune system to a virus in the nerve roots, although these speculations are unsubstantiated . FurtherBrachial neuritis typically resolves fairly quickly in patients, and full range of motion with strength is usually returns within the course of a year . Initial4The patient described herein represents a unique case, and to the best of our knowledge, the first documentation of postoperative onset of brachial neuritis within 24 h of cervical decompression surgery. This case is significantly distinct from other cases of post-operative emergence of brachial neuritis as common differential diagnosis were ruled out early in the process due to the highly distinction of BN symptoms. Diagnosis was confirmed clinically and radiologically. Furthermore, it is imperative that the surgeon regards possible rare diagnosis in evaluating post-operative neurological deficits. Overall, the patient responded quite well to aggressive physical therapy to the left arm, successfully reversing the deficits of the symptoms within six-months. Recovery and prognosis of brachial neuritis is generally effective, but can often be slow-paced.None.All procedures/reporting has been done in ethical accordance. Ethical approval from the institution is not necessary in our particular case as our study is exempt.Patient consent has been obtained, identifying details have been omitted from the case report.1st Author: Wrote the majority of the paper.2nd Author: Performed surgery, reviewed/edited the discussion and case report components.N/A.Both authors accept full responsibility for the work and/or the conduct of the study, have access to the data, and controlled the decision to publish.Not commissioned, externally peer-reviewed.None.


Osteosarcoma of the humerus is an unusual case. In order to salvage the limb, resection followed with total humeral replacement is preferred. In situations where those prostheses are not available, a technique of modified total humeral replacement could be used.A 20-year-old female patient came with osteosarcoma of the right humerus accompanied by pathological humeral shaft fracture, with the onset of six months. The lesion extended along the proximal to distal part of humerus. The patient underwent a neoadjuvant chemotherapy before surgery and a limb salvage surgery by wide excision of the humerus and reconstruction using modified total humeral replacement was performed. Modifications to the shoulder hemiarthoplasty and total elbow prostheses were made, so both prostheses could join as one unit.After the surgery, no disturbances of hand function were seen, and functional outcome was evaluated by using Musculoskeletal Tumour Society Score (MSTS) score with score of 83% (excellent). After 3 years of follow up, patient could work and resume daily activity with improved MSTS score. There were neither sign of tumor recurrence nor distant metastasis on follow up.Total Humeral Replacement for the treatment of humerus malignancy was feasible by using this modification. This procedure yielded good functional outcome. HoweverTotal humeral osteosarcoma remains a challenge for treatment. Currently, osteosarcoma of the humerus has been successfully treated with limb salvage procedure with modular prosthesis as the preferred implant . Limb saUnfortunately, the modular prosthesis for total humeral is not available in our place of practice . Therefo2A 20-year-old female presented to our center with a telangiectatic osteosarcoma of the humerus. There was no history of malignancies within the family.MRI examination and open biopsy were performed by the previous physician. At the time the results were thought to be malignant lymphoma. After several diagnostic trials, the patient visited our center to seek advice about her telangiectatic osteosarcoma A.Fig. 1Piso-hyperintense and expansile lesions along the humerus  were spared during the total resection of the humerus. Axillary nerve was sacrificed during the tumor resection. To reconstruct the humerus, both long shoulder hemiarthroplasty and total elbow prostheses were used. These two prostheses were joined using two long one-third tubular plate that worked as the long stem augmentation. Extension cerclage wire was used to make the implant as one unit A. FinallFrom the post-operative x-ray, the modified prosthesis sat well on the shoulder and elbow joint . Functio31 Osteosarcoma is mainly on the metaphyseal region of the bone, but in this case, it extended along the humerus, thus made it an unusual variant of osteosarcoma. Not many osteosarcoma of the whole humerus that can be found in other studies regarding total humeral replacement. The second decade of life is the peak incidence of osteosarcoma cases, which matches with our patient. Approximately 58% of osteosarcoma patients are male, it means that females are more often to get an osteosarcoma compared to males [The humerus, especially proximal part, is a common site for the primary osteosarcoma on the upper limb, being the third site to be the most affected after the distal femur and proximal tibia.to males .This patient came with a chief complaint of sudden pain on her right arm after she heard a crack sound when she lifted a baby. The patient had no complaint on her arm before. On osteosarcoma cases, pathologic fracture is rare as the chief complaint . Pain, aFrom radiological findings, we got the results of the mixed lesions on the x-ray. MRI showed a hyperintense extracompartemental lesions along the humerus. Laboratory findings showed an increased level of alkaline phosphatase and lactate dehydrogenase. These findings were consistent with characteristics of the osteosarcoma ,2. The mLimb salvage procedure was chosen for this case with resection of the whole humerus, followed by modified total humeral replacement. Before the surgery we had the patient completed the neoadjuvant chemotherapy regiment for three cycles in three months. Neoadjuvant or induction chemotherapy using multiple drugs, in this case Doxorubicin and Cisplatin without high dose methotrexate, is a modality used to increase the survival rate of limb salvage procedure by preventing the tumor metastases to the lung and reducing the emergence of drug-resistant tumor cells . ChemothThere is relatively better prognosis of telangiectatic osteosarcoma (TOS) than that of other variants of osteosarcoma. There is important involvement of CEACAM1 in angiogenesis. CEACAM1 is a major effector of vascular endothelial growth factor (VEGF) in early tumor microvessel formation. VEGF increases CEACAM1 expression on both mRNA and protein levels, and the administration of a monoclonal CEACAM1 antibody blocks in vitro VEGF-induced endothelial tube formation. Furthermore, a disease that produces severe local inflammation accompanied by accumulation of CD11b cells at the site of infection, a VEGF-independent role of CEACAM1 has been characterized. Both blood and lymphatic vessel formation appear to be affected by the loss of CEACAM1/CD11b cells, which control angiogenesis during inflammation. Due to its capacity to evade the immune system, as well as its potent proangiogenic effects, CEACAM1 appears to play an important role in tumor growth and progression.The tumor on this patient extended along the humerus, thus making it an excellent candidate for the total humeral replacement. Total humeral replacement itself is a rare procedure, and so are the studies that published the results of this procedure. All studies tried as they could to preserve the rotator cuff, biceps, deltoid tendons, and also the nerves including axillary, radial, median, and ulnar nerves . For theThe preferred prostheses following resection of the tumor is the Modular Replacement System (MRS) . At the Fabroni et al. used a custom-made endoprosthetic for adult and had long-term outcomes from his three cases of total humerus replacement ,9. The dIt is cheaper compared with the MRS and easily available. Our concern is the longevity of the prostheses, because it was assembled using a separated component with plate, wire, and bone cement. The construction was firm during the operation, but longer term of follow up is needed to evaluate the results.Post operatively, patient had a functional-scores of 83%, based on the MSTS for the upper limb. This finding corresponded to other studies who had average scores of 71.5% . Follow Modified total humeral replacement was performed for the unusual osteosarcoma of right humerus in twenty-year-old female with excellent post operative hand function. With long term follow up and good documentation, this modification could serve as cheaper alternative for MRS with good amount of availability and flexibility.The authors declare that there are no conflicts of interest regarding the publication of this article.The authors declare that the study sponsors had no involvement in the collection, analysis and interpretation of data, in the writing of the manuscript, and in the decision to submit the manuscript for publication.The study has been approved by institution’s ethical committee.Written informed consent was obtained from the patient for publication of this case report and accompanying images.Adisa Yusuf Reksoprodjo and Yogi Prabowo performed the treatment, performed measurement, and wrote the manuscript. Rizky Priambodo Wisnubaroto helped wrote the manuscript.N/A.Yogi Prabowo.Not commissioned, externally peer-reviewed.