Background. Inflammatory Fibroid Polyp (IFP) of the colon is an exceedingly rare condition. Since 1952 till now only 32 cases have been reported worldwide of which only 5 were giant (>4 cm) polyps mostly found in the caecum (15 cases) with only 3 in the descending colon. Case Presentation. A 36-year-old female with no previous illness presented to the emergency unit with an acute onset pain over the right hypochondrium for 3 days associated with intermittent fever and anorexia. As she had evidence of localized peritonitis she underwent a diagnostic laparoscopy and subsequently an exploratory laparotomy. A mass measuring 8 × 7 × 5 cm arising from the hepatic flexure of colon was noted. Right hemicolectomy with ileotransverse anastomosis was performed. The mass was subsequently reported to be IFP. Conclusion. IFP is a very rare condition with clinical presentation depending upon its size and location. Definitive diagnosis is possible with histopathological examination of tissue aided by immunohistochemical studies. Surgical resection has been the most common method of treatment especially for large and giant colonic IFPs owing to challenges in terms of diagnosis and technical difficulties associated with endoscopic methods. The first case of IFP was described by Konjetzny in 1920 as “Polypoid Fibroma” . The etiThe presentation of IFP varies and because of its rarity the correct preoperative diagnosis is often difficult and delayed. We report an interesting, rare case of IFP of the hepatic flexure of colon in an adult female. Its clinical presentation, investigative findings, and management are discussed and relevant literatures are reviewed. The rarities of this case are the atypical site of its occurrence and acuteness of its presentation.A 36-year-old female with no previous illness presented with an acute onset pain over the right hypochondrium for 3 days associated with intermittent fever and anorexia. Physical examination revealed a tender and guarded right upper abdomen. Hematological and biochemical tests were normal. Abdominal radiographs were unremarkable. Abdominal sonography revealed a double walled solid mass in the right upper abdomen very close to the liver and bilateral ovarian cysts. In view of patient's general condition and lack of facilities, CT scan and Colonoscopy could not be done. With differential diagnoses of ruptured hydatid cyst, duodenal ulcer perforation, acute acalculous cholecystitis, and an ulcerated GIST  an emergency diagnostic laparoscopy followed by midline laparotomy was performed. At diagnostic laparoscopy purulent and fibrinous reaction in the subhepatic region was noted based on which decision to proceed further was made. At laparotomy, a mass measuring 8 × 7 × 5 cm arising from the hepatic flexure of colon was noted as shown in Histopathologically, gross examination confirmed the operative findings and the cut section revealed an obvious bulge in the serosa caused by the mass that seemed to involve the full thickness of bowel wall, besides its glary myxoid appearance as shown in Microscopically, there was an intact mucosal lining and the mass was located in the submucosa spreading up to the serosa. There was low cellularity with proliferation of spindle-to-stellate shaped cells in a myxoid-to-pink hyalinized background. The stellate cells had plump nuclei and conspicuous nucleoli. The intervening vascularity was prominent with associated moderate mixed inflammatory infiltrates with large number of eosinophils. The serosal aspect showed necrosis with collections of acute inflammatory exudates. There was no evidence of cellular atypia, pleomorphism, or increased mitotic activity as shown in Immunohistochemical staining was found negative for CD34, CK PAN, and CD117 as shown in Figures The patient had an uneventful recovery and was discharged on the 10th postoperative day. At one-year follow-up, she remained symptom-free.IFPs are rare benign mesenchymal gastrointestinal tumours , 6. AlsoRegarded by many as reactive tumours of nonneoplastic origin until 2008, the neoplastic nature of IFPs became evident after the detection of activating PDGFRA mutations .IFPs are found mostly in the stomach (70%) and the small intestine (20%). Colonic IFPs are exceedingly rare and most commonly located in proximal colon, especially in the caecum. They can be sessile or pedunculated and usually contain blood vessels, fibroblasts, and an edematous stroma rich in eosinophils , 6.Clinical presentation depends on size and location generally. With enlargement they can cause abdominal pain, hematochezia, anemia, weight loss, diarrhea, and intussusceptions . DefinitAn extensive search in PubMed, Medline, and Google in reference to colonic IFPs showed that from 1952 till now only 32 cases have been reported worldwide of which only 5 were giant (>4 cm) polyps mostly found in the caecum (15 cases) with only 3 in the descending colon.Treatment approach was surgical in 20 58%) while endoscopic resection was done in only 8 (23%). There was no reported recurrence in the colon [8% while Surgical resection has been the most common method of treatment specially for large and giant colonic IFPs owing to challenges in terms of diagnosis and technical difficulties associated with endoscopic methods such as limited view due to large size, morphology (sessile or pedunculated), and location  and concerns regarding completion of procedure, recurrence, and cure.In a setup like ours where the presentation was of an acute abdomen and the possibility of CT scan and Colonoscopy was remote, we opted for an open resection. However, provided that latest technology exists, it will be worthwhile attempting the least invasive methods.In conclusion, Inflammatory Fibroid Polyp of the colon is an uncommon presentation of an uncommon diagnosis. The clinical and radiological picture may mimic carcinoma and definitive diagnosis may be possible with histopathological evaluation aided with immunohistochemical analysis. Once resected with negative margins, IFP does not require further treatment and has a good clinical outcome. Therefore, awareness of the clinical presentation and good pathological expertise are important adjuncts in the diagnosis. Surgery is the mainstay of treatment in the acute presentation.


Introduction: Cartilaginous nasal septal agenesis is a rare finding. In fact, just one case has been reported to have congenital agenesis of all nasal cartilages in a 6-year-old child by Bakhshaee et al. The literature review shows another case that was reported by Ozek et al in Turkey, where they reported a case of total nasal agenesis that was associated with Tessier no. 30. We could not find a similar case in the literature where only agenesis of the nasal cartilaginous septum was present. Methods/Case Report: This is a case report of a 5-year-old child presenting to our clinic with agenesis of his nasal septum and attenuation of the upper and lower lateral cartilages. His parents were seeking a corrective procedure to improve the shape of his nose. He was a male child with a right unilateral cleft lip and palate that were corrected surgically in 2009 (lip repair) and 2010 , respectively. Results: On postoperative week 3, the patient's mother brought him to the emergency department with a history of falling on his face while playing at home. Examination revealed swelling of the nose but no breathing difficulties. He still had an acceptable augmented nose but with sings of deviation and collapse. Discussion: We report this case to find an answer to how such cases can be approached in the future in terms of the surgical intervention required and to study the options of redoing such cases. Cartilaginous nasal septal agenesis is a rare finding. In fact, just one case has been reported to have congenital agenesis of all nasal cartilages in a 6-year-old child by Bakhshaee et al.This is a case report of a 5-year-old male child presenting to our clinic with agenesis of his nasal septum and attenuation of the upper and lower lateral cartilages. His parents were seeking a corrective procedure to improve the shape of his nose. He was a male child known with a right unilateral cleft lip and palate that were corrected surgically in 2009 (lip repair) and 2010 , respectively. He had no other medical problems, no family history of similar conditions, and had undergone no surgical procedures to his midface other than those mentioned earlier. There was no history of facial trauma, and the patient had no known allergies. No other congenital anomalies were present, and he never suffered from any infections of the face.No breathing difficulties were reported. The nasal deformity was causing him stress at the school, and it had a great impact on his social behavior.His physical examination revealed an otherwise healthy child with a paramedian scar on his lip from previous surgery and an obviously collapsed nose in terms of projection with a deviated columella to the right side and discrepancy of the nostrils, with the left nostril being bigger than the right one. His nose was rotated downward, and he also had a short, thin, and curved columella. His nasolabial angle was 80° and his nasofrontal angle was 160° -3. His iA preoperative computed tomographic (CT) scan of the facial bones was obtained to assess his nasal bone structure and position. CT scans showed flattening of the nasal bones and the presence of the bony septum. Nasal cartilages were not visible.After obtaining surgical consent from the family, the patient was taken to the operating room and the senior author (M.A.M.) decided to correct the deformity with a rib bone and cartilage graft. During the dissection, we were unable to identify any cartilaginous septum and his lower and upper lateral cartilages were attenuated. In addition, the medical crura of the lower lateral cartilages were entirely missing.A 6-cm rib bone and cartilage graft (1-cm bone and 5-cm cartilage) was harvested from the right fifth rib. Through a left intercartilaginous incision, the osseous end of the graft was fixed to the nasal bones using 2 percutaneous screws. With this maneuver, we achieved dorsal nasal augmentation while also correcting the collapsed nose -6. BecauOn postoperative week 3, the patient's mother brought him to the emergency department with a history of falling on his face while playing at home  and 8. TOur case patient had a history of cleft lip and palate with clinical identification of the absence of the nasal septal cartilage but the presence of the lower lateral cartilage, though, deformed and attenuated. No genetic workup was done for the case patient, although it would be interesting to see if this finding has any genetic association. We appreciate the presence of midfacial retrusion, although this can be explained by the effect of the palatoplasty that was performed earlier.We report this case to find an answer to how such cases can be approached in the future in terms of the surgical intervention required and to study the options of redoing such cases. In addition, we could not relay this finding to any embryological abnormality that would result only in the deficiency of the cartilaginous nasal septum. Robinson and Hilger


We report the case of a large lumbar schwannoma eroding the vertebra and originating from spinal canal with invasion of the retroperitoneal space. We also review all the cases in literature reporting lumbar schwannomas eroding the vertebral bodies and invading the retroperitoneal space focusing on the surgical strategies to manage them. Spinal CT-scan revealed a 44 mm × 55 mm inhomogeneous soft-tissue mass arising from the right L5-S1 neural foramen and its most anterior portion had a clear colliquative aspect. Magnetic resonance image showed a neoplastic lesion with homogeneous low signal in T1WI, heterogeneous signal in T2WI, and strong enhancement in postgadolinium examination. It developed as well in the retroperitoneal space, posteriorly to the iliac vein, up to the psoas muscle with wide erosion of the omolateral conjugate foramen. We performed a one-step combined approach together with the vascular surgeon because the lesion was too huge to allow a complete resection via a posterior approach and furthermore its tight relationship with the psoas muscle and the iliac vessels in the retroperitoneal space should be more safely managed via a retroperitoneal approach. We strongly suggest a 1-step surgery first approaching the dumbbell and the intraspinal schwannomas posteriorly achieving the decompression of the spinal canal and the cleavage of the tumor cutting the root of origin and the vascular supply and valuating the stability of the spine for potential artrodesis procedure. The patient must be then operated on via a retroperitoneal approach achieving the complete en bloc resection of the tumor. Lumbar schwannoma is a common lesion affecting the peripheral nervous system moreover in the spinal canal or into the vertebral foramina.Giant lumbar schwannomas have been rarely described in literature , 2 and pThe goal of surgery is the complete removal of these lesions to avoid recurrence but giant retroperitoneal schwannomas present more difficulties to choose the best surgical route and to manage the close relationship with vital abdominal structures.Furthermore, the erosion of a lumbar vertebra also complicates radical surgery for its challenging approach because of the potential following instability and the inherent literature is quite exiguous.We report the case of a large lumbar schwannoma eroding the vertebra and originating from spinal canal with invasion of the retroperitoneal space through the neural foramen.Such lesion involved the psoas muscle and the abdominal vessels requiring first a posterior approach and then a retroperitoneal route together with the vascular surgeon.We also review all the cases in literature reporting lumbar schwannomas eroding the vertebral bodies and invading the retroperitoneal space focusing on the surgical strategies to manage them.A 64-year-old male came to our observation complaining of pain localized in the posterior side of his right inferior limb since at least 2 years. Over the last 6 months, he progressively developed numbness in the same dermatome. There was neither obvious relief nor precipitating factors.At the admission, the neurological examination was unremarkable. Physical examination revealed no palpable mass within the abdomen.Spinal CT scan without contrast medium  revealedMagnetic resonance image (MRI) showed  a neoplaElectroneurography and electromyography at the lower extremities showed neurogenic damage with denervation signs of the right L5 nerve.We decided to perform a one-step combined approach with the vascular surgeon because the lesion was too huge to allow a complete resection via a posterior approach and furthermore its tight relationship with the psoas muscle and the iliac vessels in the retroperitoneal space should be more safely managed via a retroperitoneal approach.First we dissected free the nerve root from the tumor's capsule debulking the lesion and decompressing the spinal canal and then removed the residual portion after the control of the psoas muscle and the iliac vessels.During the first step, we performed an L5-S1 hemilaminectomy, a facetectomy, and a resection of L5 transverse process. The tumor clearly originated from the L5 nerve root and invaded the vertebral foramen. We debulked the lesion using the ultrasonic surgical aspirator clearly maintaining the capsular surface to keep the correct margins of resection allowing a complete removal of the tumor.Such approach allowed us not to sacrifice completely the nerve root, and after debulking, the mass was removed from the vertebral foramen up to the psoas muscle and the bony resection was minimal.Immediately after the neurosurgical procedure, the vascular surgeon excised the residual retroperitoneal portion of the tumor by a right semilunar, subumbilical approach. He exposed the common iliac artery and the common iliac vein as well as the major part of the external iliac vein and of the hypogastric vein. The distal segment of the inferior vena cava and the aorta were also accurately visualized. Resection of the lesion medially to the psoas muscle was impossible because of its firm adherence to the muscle itself and the risk of vascular injury.The mass was firmly impinged to the bone and to the posterior aspect of the common iliac vein in its passage to extern iliac vein .In order not to damage the iliac veins and to save the hypogastric vein, the last lumbar vein was tied and the vena cava was raised and moved laterally. The iliac vein was pulled upwards and medially with a widening of the operative field and the mass was finally removed, without traction, laterally .The spine was considered stable and no additional fixation was performed because of a standard unilateral posterior approach and of the small portions of eroded vertebral body.Five days after the operation, the patient was discharged and neurological examination revealed mild numbness in the right L5 dermatome that recovered 3 months later. There were no walking difficulties or back pain. The postoperative magnetic resonance image (MRI) confirmed  the compClinical follow-up at 3, 6, and 12 months demonstrated the complete recovery of the patient.Microscope examination revealed a mesenchymal spindle-cells tumor with rare mitotic activity. Tumor cells strongly and diffusely express S-100 protein, providing the diagnosis of benign schwannoma (WHO I).Giant schwannomas are very rare accounting for 0,3–5% of all these kind of tumors and affecting particularly the retroperitoneal space , 2.Giant lumbar schwannomas eroding the vertebral body and extending into the retroperitoneal space are also rarer and in literature we found only 10 cases [We focused our attention on the review of the cases of lumbar schwannomas eroding the vertebral bodies and invading the retroperitoneal space to evaluate the different surgical strategies and the considerations leading to 1 or 2 steps approaches.The schwannoma is a typical benign tumor, well-encapsulated and without adherence with the surrounding tissue often presenting a delayed diagnosis due to their insidious behavior and slow growing, but large lesions present a more aggressive behavior with local compression, adherence, and bone rearrangement , 2.Large retroperitoneal schwannomas often demonstrate such elements with important relationship with the large abdominal vessels accounting for high risk of intraoperative venous injury so that, in our opinion, involving vascular surgeon in the surgical team is strongly recommended.However, such giant retroperitoneal lesions could mimic other aggressive intraabdominal tumors and osteoblastoma, chondrosarcoma, aneurismal bone cyst, or giant cell tumors must be considered in differential diagnosis.Rapid growth, bone erosion, and invasion of the surrounding structures are similar for high grade sarcomas that, in doubtful cases, must be excluded preoperatively to choose the optimal strategy to avoid recurrence and metastases.Invasion of the retroperitoneal space with bone erosion and myofascial plane extension defines a malignant schwannoma  and somePreoperative biopsy should be reserved to the quite doubtful cases because the cellular pleomorphism of the degenerated areas could produce the misdiagnosis of malignancy  and SakaSchwannoma demonstrates some radiological features suggesting the appropriate diagnosis as well circumscribed mass with heterogeneous contrast enhancement, calcifications, and cystic portions up to 40% in large tumor , 10.MRI represents the exam of choice for preoperative diagnosis demonstrating hypointensity in T1 weighted images and hyperintensity in T2 weighted images [After administration of contrast medium, schwannoma shows wide enhancement.Angio-CT and abdominal CT study are absolutely mandatory in order to clarify the relationships of the tumor with the aorta, vena cava, and retroperitoneal structures.The goal of surgery of these lesions is the complete removal preserving neural and intra-abdominal structures avoiding recurrence and postoperative adjuvant therapy  but liteIn 1971, Dickson et al.  reportedIn 1997, Kádár et al. [Napolitano et al.  describeIn 1998, Chang et al.  describePaderni and Boriani  in 2002 Case 3 of Daneshmand's series  in 2003 Sofia et al.  in 2008 In 2008, Sakalauskaite et al. [In 2009, Chiang et al.  reportedIn 2009, Park et al.  report aThe few papers in literature produce a lack of guidelines in treatment of such tumors so that surgical strategy must be discussed case by case relating to surgeon's experience and lesion's characteristics and many topics must be considered as the extension and the kind of the resection, the one-step or two-step approach, the kind of laparotomy, the involvement of abdominal structures and vessels, and the vertebral stability .Usually, retroperitoneal location is correlated to lesion's large size (>3 cm) producing strong adherence with the surrounding tissue, vessels, and organs and behaving like a malignant schwannoma.These lesions appear to be firm, adherent to surrounding structures, and often difficult to mobilize so we believe that a previous posterior approach can reduce their vascular source but furthermore produces an easier mobilization of the mass.The cleavage and the disconnection of the tumor from its posterior vascular support and its radicular origin, achieving at the same time an optimal decompression of the spinal canal, dramatically produce an easier en bloc resection via the subsequent anterior approach.Complete neurological deficit after complete sacrifice of the involved spinal root has been not so often reported with an incidence of about 23%  due to tSuch kind of approach allows the surgeon to achieve a safer anterior en bloc resection because of the frequent involvement of abdominal structures as vena cava, aorta, iliac vessels, ureter, and kidney that could be easier managed after the reduction or the elimination of the posterior vascular supply and after the debridement of the lesion from the posterior neural structures.We believe the posterior approach is also optimal to plan a vertebral stabilization when the stability of the spine is altered, so we advocate a 1-step surgery with an initial posterior approach followed by anterior removal of the lesion.However, in literature, 6 cases , 10, 11 In 4 of these, no spinal canal compression was reported and 3 authors performed only an anterior approach , 8, 11 wThe authors  report aCase 3 of Daneshmand's series  sufferedThe patient reported by Chang et al. , after aWe suggest a multidisciplinary approach involving two surgical teams  to complete the surgery in a single session with decompression of the spinal canal, disconnection of the tumor from its origin, and eventual stabilization when necessary.This approach leads to an easier en bloc resection of the lesion via a subsequent retroperitoneal approach with important reduction of patient's affliction, medical expenses, and faster discharge.Preoperative vascular study and a vascular surgeon being part of the surgical team is in our opinion mandatory because such lesions are often adherent to vital structures as vena cava and aorta; involvement of iliac and mesenteric vessels must be carefully considered.Vascular identification and preparation of these vessels must be the first step of this surgery to prevent hemorrhage or acute bleeding.Eventual spinal instability must be taken into account when a large vertebral amount must be resected and 8 on 10 patients of our review underwent spinal stabilization by anterior plating and grafting or posterior pedicle screws.Two cases of 10 did not require a postoperative stabilization of the spine and according to these also our patient did not require stabilization.Stabilization must be considered after wide bony resection or vertebral collapse and if postoperative dynamic X-rays films demonstrate instability.We strongly suggest a 1-step surgery first approaching the dumbbell and the intraspinal schwannomas posteriorly achieving the decompression of the spinal canal and the cleavage of the tumor cutting the root of origin and the vascular supply and valuating the stability of the spine for potential artrodesis procedure. The patient must be then operated on via a retroperitoneal approach achieving the complete en bloc resection of the tumor. Only anterior approach could be evaluated for lesion not involving the spinal canal and not eroding a large amount of vertebral body not requiring a posterior stabilization.


Hydatidosis is a public health problem in endemic countries. Hydatid cysts are located usually in the liver and the lungs. Primary pancreatic hydatid cyst is in unusual location and rarely causes acute pancreatitis. In this paper, we report a case of a 34-year-old man who admitted with recurrent acute pancreatitis. Following the preoperation investigations, the primary impression was a pancreatic pseudocyst. During surgery, a primary hydatid cyst was detected in the pancreas measuring 35 × 20 × 15 cm. The treatment consisted of evacuation and external draining of the cyst. In addition, we summarized 14 cases of primary hydatid cyst of the pancreas associated with acute pancreatitis reported in the literature. Echinococcus. It is endemic in Mediterranean countries, the Middle East, South America, and the Indian subcontinent [Echinococcus lead to infection in humans; Echinococcus granulosus is the most common parasite causing cystic echinococcosis with larval stage that represents more than 95% of cases [Echinococcosis, hydatid disease, and hydatidosis are alternative names of a zoonotic parasitic disease caused by the tapeworm ontinent –3. Four of cases . It can of cases .Acute pancreatitis is commonly associated with alcohol intake or disorders of the pancreaticobiliary tracts . Acute pWe report a case of hydatid cyst diagnosed after acute pancreatitis episode, and we reviewed and summed the data from other reported cases of hydatid cyst manifested as acute pancreatitis in the English and French literature.A 34-year-old man was admitted to the Surgery Department in our centre in July 2016 with diffuse abdominal pain, dyspnea, general fatigue, and weakness. In his medical history, he had been admitted to another centre two months ago for an episode of acute pancreatitis.Abdominal ultrasonography revealed a heterogeneous area of 5 cm in size in the body of the pancreas, peripancreatic fluid, gallstones with thickness in the gallbladder wall, and multiple cysts in the left kidney. Abdominal computed tomography (CT) showed heterogeneous collection of fluid with a thick wall of 12 × 4 cm in size along the body of the pancreas and left colic angle (often an abscess or a pseudocyst) with infiltration of adipose tissue around it and mild thickness at the wall of the colon. A simple renal cyst was also reported in the left kidney .Laboratory investigations were within normal levels except an elevation in C-reactive protein value (18.1 mg/dl) and amylase (765 U/L). The hepatic tests were within the normal range  was 0.64 mg/dl, alanine aminotransferase (ALT) was 20 IU/L, and aspartate transaminase (AST) was 19 IU/L). The patient was treated as an episode of acute pancreatitis.Other investigations were performed; upper gastrointestinal endoscopy (UGI endoscopy) demonstrated an esophagitis (grade A) at the lower esophagus, incompetence of the lower esophageal sphincter (LES), diffuse congestion of the mucous membrane of the stomach, and aphthous ulcer at the fundus of the stomach. Lower GI endoscopy was normal until the terminal ileum.A month later, the CT scan for thorax and abdomen data were similar to the previous finding, and the pancreatic cyst measuring 13.5 × 7 cm stretched down through the peritoneal cavity in front of the mesenteric vessels.The laboratory values were normal, and a primary diagnosis of pancreatic pseudocyst was probable, and the decision of surgical intervention was decided.Intraoperatively, an extreme oedema in the pylorus, the transverse mesocolon, the head and body of the pancreas, and the hepatoduodenal ligament was found. A cholecystitis required cholecystectomy. After entering the lesser sac, a large mass of 35 × 20 × 15 cm in size was found, located in the space between the tail of the pancreas, spleen, left colic angle, left kidney, stomach, and diaphragm. The mass was hard to dissect from the neighboring structures. In puncture, a clear pure liquid was aspirated proposing the existence of a hydatid cyst. Cyst fenestration was performed, and multiple daughter cysts were evacuated; the endocyst membrane was removed . A FoleyThe patient had another episode of acute edematous pancreatitis after a month of surgery, and the amylase level was over 1000 U/L. The development of local retroperitoneal abscess required puncture and drainage by CT; the patient also developed a deep venous thrombosis and was treated by anticoagulants.During 18 months of follow-up, the patient was well with no episodes of recurrence or other complications.Besides our case, 14 cases of a pancreatic hydatid cyst with acute pancreatitis were reported. Three of them are available with abstract and one is not accessible. The ratio of women to men was 3/10. The mean age of the patients was 30.2 years. The location was solitary in the pancreas in 10 patients. The cyst was found in the body (7.1%), tail (28.5%), body and tail (21.4%), or head (28.5%) .On clinical examination, no specific complaints or signs were found to distinguish hydatid cyst from other etiology of acute pancreatitis so that the final diagnosis was made preoperatively in 4 cases by ultrasonography or computed tomography. In our case, the final diagnosis was made intraoperatively.3, and the amylase median was 1718 U/L.Laboratory investigations varied between cases, and they were not available in all cases. The average of white blood cells was 17,943/mmSurgical procedures that have been carried out varied according to every situation; for example, left pancreatectomy with splenectomy was performed in 5 cases. Only 2 patients were treated with albendazole before surgery.n=1), complex pancreatic fistula , and portal vein thrombosis (n=1). Fistulae were treated with parenteral nutrition, antibiotic therapy (ampicillin), and adequate local treatment.After surgery, there were some complications such as external pancreatic fistula  .Hydatid cysts can localize virtually in any organ and structure of the body. The highest rate of cysts location exists in the liver and the lungs, which assess to 70% and 20% of patients, respectively. Other organs present in a small proportion of patients , 22. It According to a previous review in 2012 , the cysThe clinical manifestations of cystic echinococcosis (CE) depend on the size of the cyst, site, and potential complications such as rupture, compression on adjacent tissue or organs, infections, and intestinal or biliary fistula .Hepatic hydatid cysts rupture into the biliary tree in 5–17% of the cases . This coDiagnosis of cystic hydatidosis is based on imaging. Ultrasonography allows classification of the cysts, as the WHO classification tabulates cysts in 3 groups: the first one is active and includes CE1, which appears as unilocular anechoic cystic lesion with double-line sign, and CE2, which is a multiseptated “rosette-like” “honeycomb” cyst; the second one is transitional and contains CE3a cyst with detached membranes (water-lily sign) and CE3b cyst with daughter cysts in the solid matrix; and finally, the third group is inactive and has CE4 and CE5 cysts with heterogeneous hypoechoic/hyperechoic contents and no daughter cysts and solid plus calcified wall .CT scan demonstrates the features of hydatid cysts better than US. CT scan can identify the number, size, shape, margins, anatomic location, and calcification. Furthermore, the abdominal CT scan may be used to evaluate complications like rupture into the main pancreatic duct. It also assesses the lesions during therapy . MagnetiThe management of the cystic echinococcosis (CE) includes surgery, percutaneous treatment, antiparasitic drug therapy, and observation . SurgeryDespite pancreatic hydatid cyst being rare, it may be treated as a reason to acute pancreatitis. Hydatid cysts should be counted as a significant differential diagnosis for cystic lesions of the pancreas and other organs, especially in endemic regions. Clinicians must know about complications of hydatid cyst in its uncommon locations.


Neuroendocrine tumors (NETs) arising in the duodenum are rare neoplasms that are often classified as indolent and have a low potential to metastasize. Although rare, multiple reports cite an increasing incidence of duodenal NETs. Symptoms are usually nonspecific and the diagnosis is made via endoscopy. Endoscopic resection is the mainstay of therapy. The prognosis is usually favorable. We describe a case of a duodenal NET that presented with vague symptoms in order to increase the awareness of this rare but increasing in frequency entity. Neuroendocrine tumors (NETs) are neoplasms of enterochromaffin cell origin that display neurosecretory capacity, which may result in a carcinoid syndrome . Well-diA 48-year-old woman, with a past medical and surgical history of hypertension, hyperlipidemia, hypothyroidism, anxiety, depression, and abdominoplasty, presented with a five-month history of intermittent diffuse abdominal pain and bloating. The symptoms were exacerbated by food intake and associated with nausea and alternating diarrhea and constipation. Physical examination was normal, except for a mild diffuse abdominal tenderness. The complete blood count, complete metabolic panel, and the thyroid-stimulating hormone (TSH) were within normal limits. The computed tomography (CT) scan of the abdomen and pelvis with intravenous contrast was unremarkable as well as the colonoscopy. The esophagogastroduodenoscopy (EGD) showed esophagitis in the distal third of the esophagus, chronic gastritis in the body of the stomach and antrum with negative H. pylori on an immunohistochemical stain, and a 0.2 cm duodenal bulb polyp that was resected Figure . The patDuodenal NETs comprise 2-3% of all gastrointestinal (GI) endocrine tumors . AlthougWHO has classified NETs into well-differentiated NETs; well-differentiated neuroendocrine carcinoma, which has a low malignant potential, and poorly differentiated neuroendocrine carcinomas, representing small cell neuroendocrine carcinomas of high malignancy potential . HistoloEndoscopic resection may be considered for non-metastatic duodenal lesions measuring up to 2 cm if the tumor is confined to the mucosa and submucosa on EUS examination . SurgicaThe five-year survival for duodenal carcinoid lesions is 60% . Some stOur case illustrates a unique and atypical clinical presentation that has led to the diagnosis of a rare location of NET. Physicians treating patients presenting with non-specific gastrointestinal symptoms should keep this entity in mind. An early diagnosis establishes the tumor stage and predicts the prognosis, which eventually leads to the appropriate therapeutic management.


Appendiceal mucinous cystadenoma can present in various ways, and it is most commonly encountered incidentally during appendectomy, but mechanical intestinal obstruction secondary to an appendiceal mucocele has been rarely reported.We report a case of mechanical intestinal obstruction secondary to appendiceal mucinous cystadenoma. After nasogastric decompression and initial aggressive intravenous fluid resuscitation, an emergency operation was performed under the diagnosis of acute mechanical intestinal obstruction.We performed an appendectomy and intraoperative enteral decompression without anastomoses. The pathologic examination (PE) revealed appendiceal mucinous cystadenoma. After the operation, the patient's recovery went smoothly, and the patient was discharged on the fifth postoperative day. No tumor recurrence was recorded over an 8 month follow-up period.Early operative intervention should be recommended to the patient with acute mechanical complete intestinal obstruction, especially the patient who had no previous abdominal surgery. And it is vital to discriminate benign and malignantappendiceal mucocel in determining the extent of surgery. Here, we present a case report describing a 76-year-old man who was diagnosed with mechanical complete intestinal obstruction secondary to appendiceal mucinous cystadenoma.Mucocele is an uncommon disease of the appendix. Appendiceal mucocele can present in diversified ways, but it is most commonly encountered incidentally during appendectomy.2A 76-year-old previously healthy man, who had no history of abdominal surgery, presented to our emergency room with a 38-hour history of intermittent abdominal pain, nausea, and the disruption of bowel movements. His abdomen was mildly distended and tympanic with diffuse slight tenderness, particularly in the periumbilical areas. He had sonorous bowel sounds. A computed tomography (CT) scan of the abdomen revealed dilated small bowel loops with air-fluid levels suggestive of a small bowel obstruction Fig. . The pat3 observed in 0.2% to 0.3% of appendectomies and 8% to 10% of appendiceal tumors. Appendiceal mucoceles are more frequent in women than in men, and are often discovered in patients older than 50 years. Mucocele is an uncommon pathology of the appendix characterized by a cystic dilation of the lumen as a result of an abnormal accumulation of mucus, and it usually results from either benign (cystadenoma) or malignant (cystadenocarcinoma) epithelial proliferation. Clinical manifestations of appendiceal mucoceles are quite diversified. 30% to 64% of patients present right lower quadrant pain, similar to the acute appendicitis, which is the most common manifestation.–9 A quarter of patients are asymptomatic which are more frequently found in benign appendiceal mucocele, while appendiceal mucoceles are discovered incidentally during physical examinations , at surgery or during imaging study for other problems.,11 Other clinical manifestations mainly include intussusception, genitourinary symptoms, low gastrointestinal bleeding, and so on. ,12 However a bowel obstruction secondary to an appendiceal mucocele has been rarely reported.–16 And the early diagnosis is challenging. Imaging with CT has better apply to establish a preoperative diagnosis in similar cases.Appendiceal mucocele is a relatively rare disease,Although it would have been almost impossible to make the right diagnosis prior to the operation in this case, there are still many aspects that are worth reflecting on. First, the patient came into the emergency room with signs and symptoms of acute mechanical intestinal obstruction, and he had no previous abdominal surgery. Therefore, we almost could exclude the diagnosis of adhesive intestinal obstruction. The differential diagnosis at initial presentation included a colonic tumor, benign or malignant small intestinal tumors, and a stricture secondary to other conditions , all of which often required an operation. Second, when we take the medical history of a patient with an unexplained intestinal obstruction, we should be more careful with the patient's past medical history and potential causes of the symptoms, these may help us make right diagnosis. In this case, we reinquiry the patient's case history on postoperative day, the patient had had a right lower quadrant abdominal intermittent slight chronic pain symptoms in the past 20 years, but there was no other influence in his work and daily life, so he did not care about the symptoms at all. Now if we integrated the medical history and CT, it seemed that we may have made the right diagnosis before operation. Third, we support the clinical application of total abdomen CT scanning in the case of an unexplained mechanism of abdominal obstruction. An abdominal CT is a rapid, simple, and effective means for diagnosing the location, cause, and degree of obstruction in cases of unexplained intestinal obstructions. It is particularly useful for orienting the clinician to the location of obstruction, and it would help surgeons decide where to make an incision, as an appropriately placed incision is crucial to the success of an operation. If malignancy cannot be exclusive, right hemicolectomy should be considered.,18 In this case, we should exclude the possibility of the intestine necrosis; otherwise, we must resect the highly suspected necrotic intestinal tissue further. The key-point of surgical treatment is to make resection margin clear and keep the appendiceal mucocele intact. The prognosis for benign appendiceal mucocele following complete ablation is excellent and the 5-year survival rate is almost 100%. A patient with malignant mucocele, however, is much poorer, and the 5-year survival rate has association with the degree of extension of the tumor and varies between 30% and 80%.So how did we determine the extent of surgery for the patient with appendiceal mucocele? It is vital to discriminate benign and malignant appendiceal mucocele in determining the extent of surgery. Appendectomy is appropriate therapy for unruptured benign appendiceal mucoceles, like this case. When benign appendiceal mucocele protrudes into cecal lumen, partial cecectomy may be curative. “If either cecal wall or ileum is invaded by tumor or adequate surgical margins cannot be secured, ileocecal section or right hemicolectomy may be required.”4For acute mechanical complete intestinal obstruction, the patient's past medical history and potential causes of the symptoms should be paid attention to, and an abdomen CT is very necessary; Besides early operative intervention should be recommended to the patient with acute mechanical complete intestinal obstruction, especially the patient who had no previous abdominal surgery. Moreover, it is vital to discriminate benign and malignantappendiceal mucocel in determining the extent of surgery.


Dear SirCecal duplication cyst is one of the rarest forms of alimentary tract duplication. Occurring intraluminally can mimic as intramural mass and intussusception. Thus it becomes quite daunting to make a specific diagnosis preoperatively. A 7-day-old male child with normal birth history and uneventful neonatal period was brought with complaints of bilious vomiting, abdominal distension and constipation. On examination, abdomen was soft with visible peristalsis; a lump was palpable in right iliac fossa. X-ray abdomen showed multiple air fluid levels. Ultrasonography abdomen revealed a mass in right iliac fossa. After adequate resuscitation, the child was taken up for surgery. On exploratory laparotomy, there was a firm intramural mass of size 3×3 cm palpable in ileocecal region causing obstruction with dilatation of whole of the small intestine proximal to the lesion and collapsed distal colon . Resection of the gut along with lesion was performed and ileo-colic anastomosis done. Grossly, a cystic globular structure was identified sharing a common wall with caecum and filled with mucoid material. Postoperative period was uneventful. The histopathological examination showed the cyst has all layers of large intestine, without any ectopic tissue, and shared a common wall with the caecum. Cecal duplications are exceedingly rare on literature search. [3-5] Cecal duplications can either simulate or lead to intussusceptions. [2] It can also present with features of intestinal obstruction. Ijaz et al [4] in and Keum et al [5] reported cases of cecal duplication presenting with acute intestinal obstruction. In present case, it was mimicking as intramural tumor mass. Cecal duplication cysts are although rare but should be kept in mind in neonates with intestinal obstruction.Source of Support: NilConflict of Interest: None


Introduction. Skin disorders can be the first manifestation of occult diseases. The recognition of typical paraneoplastic dermatoses may anticipate the cancer diagnosis and improve its prognosis. Although rarely observed, the sudden appearance and/or rapid increase in number and size of seborrheic keratoses can be associated with malignant neoplasms, known as the sign of Leser-Trélat. The aim of this report is to unveil a case of a patient whose recently erupted seborrheic keratoses led to investigation and consequent diagnosis of bladder cancer. Case Presentation. A 67-year-old man was admitted to the intensive care unit due to an exacerbation of chronic obstructive pulmonary disease (COPD). On physical examination, multiple seborrheic keratoses on the back of the hands, elbows, and trunk were observed; the patient had a 4-month history of these lesions yet was asymptomatic. The possibility of Leser-Trélat syndrome justified the investigation for neoplasia, and a bladder carcinoma was detected by CT-scan. The patient denied previous hematuria or any other related symptoms. Many of the lesions regressed during oncologic treatment. Conclusion. Despite the critics on the validity of the sign of Leser-Trélat, our patient fulfills the description of the disease, though urinary malignancy is a rare association. That corroborates the need of further investigation when there is a possibility of paraneoplastic manifestation. Paraneoplastic diseases may be defined as hormonal, neurological, or hematological disturbances and as clinical and biochemical imbalances related to the presence of malignancies, with no direct association with primary tumor invasion or metastasis . If promThe aim of this case report is to describe a cancer diagnosis made after following the clue given by the patient's skin condition—a case of Leser-Trélat syndrome.A written informed consent of the patient has been obtained for the publication of this case report.A 67-year-old man was admitted to the intensive care unit (ICU) due to a second exacerbation of COPD in two weeks. He came from a first aid unit, had already been through orotracheal intubation, and was receiving mechanical ventilation and antibiotics. The patient was a heavy smoker (120 pack-years of tobacco exposure) and started to claim dyspnea to great efforts over the previous few months; he had also lost 8 kg in less than a month. There was no other known comorbid condition in his clinical history. After some days of stabilization in the ICU, the patient was referred to the general internal medicine unit, where another exacerbation took place, along with fever and productive cough, likewise controlled with bronchodilators and antibiotics.On physical examination, multiple seborrheic keratoses on the back of the hands , elbows,The search for an occult neoplasia was then initiated. The investigation comprehended thorax CT-scan, upper gastrointestinal endoscopy, colonoscopy, and blood analysis, which pointed no malignancy. Colonoscopy showed four sessile polyps, one in the ascending colon (measuring 10 mm), histopathologically defined as a tubulovillous adenoma with high-grade dysplasia, and three in the transverse colon (the biggest measuring 9 mm), corresponding to tubular adenomas with high-grade dysplasia. Ultimately, abdomen CT-scan revealed one irregular-shaped vegetation on the bladder floor, measuring 22 × 18 × 16 mm, with no apparent adipose tissue invasion . The patDue to an incomplete stabilization of the pulmonary condition, the patient was discharged and returned after a month for cystoscopy and transurethral resection of the bladder tumor. Histopathological examination of the removed tissue confirmed a noninvasive low grade papillary urothelial carcinoma. Control cystoscopy did not show any residual mass. Adjuvant therapy with intravesical BCG administration was then commenced.Nine months after the start of the treatment, the patient still presented residual lesions of seborrheic keratoses, which are then pruriginous .Indirect involvement of the skin by visceral tumors can cause a variety of inflammatory, proliferative, metabolic, and neoplastic changes without the actual presence of tumor cells . In thesSix criteria have been described to consider a cutaneous alteration as paraneoplastic, known as Curth's postulates: 1) both conditions (neoplasia and paraneoplasia) are of concurrent onset, 2) both follow a parallel course, (3) the skin lesion is not part of a genetic syndrome, 4) a specific tumor occurs with a certain dermatosis, 5) the dermatosis is not common in general population, and (6) there is a high percentage of the association between both conditions [ both con both fol the derm a specifThe sign of Leser-Trélat is defined as the sudden appearance and/or rapid increase in number and size of seborrheic keratoses, secondary to a neoplasia , 6, 7. TIn 1988, Holdiness gathered the available data (60 cases) to analyze clinical features of the sign. The average age at the time of reported onset was 60.7 years. The average time of evolution of the cutaneous lesions was 14.9 weeks, highlighting its eruptive presentation as a requisite that get most of the patients to be aware of. Acanthosis nigricans and pruritus were simultaneously present in 16.7%. Of these patients, 55% had adenocarcinomas, of which 36.4% were adenocarcinomas of the stomach; lymphoproliferative neoplasias were the second most recorded neoplasms, accounting for 18.3%. Metastatic disease was reported in 56.7% of the patients, with an average survival time of 11.5 months after cancer diagnosis [The sign of Leser-Trélat has also been associated to breast, prostate, lung, ovary, and kidney cancer and melanoma . Yaniv eIn comparison to the literature, our patient was near the average age (67 years) and time of skin eruption (approximately 16 weeks) and presented pruritus in the follow-up. There was no sign of associated acanthosis nigricans on his dermatological examination. The low-stage papillary urothelial carcinoma was a rare association.Interestingly, the link between seborrheic keratoses and bladder carcinoma has also been searched at genetic level. Fibroblast growth factor receptor 3 (FGFR3) currently appears to be the most frequently mutated oncogene in bladder cancer and implies a good prognosis –14. FGFRWhether the sign of Leser-Trélat is a genuine paraneoplastic phenomenon is still discussed. Critics advocate that in the elderly both seborrheic keratoses and cancer are common and their coexistence may be fortuitous , 19. LinHeaphy et al. suggested distinguishing between a “sign of Leser-Trélat” and a “syndrome of Leser-Trélat”: the sign would be defined as an acute efflorescence of seborrheic keratoses, may be present with or without occult malignancy, and is detectable on history and physical examination alone; furthermore, the term syndrome would then be used to describe a paraneoplastic syndrome in patients with the sign in whom an occult malignancy was identified after the appearance of the sign .Many authors believe that the cumulative weight of published cases of the association of seborrheic keratoses with subsequent demonstration of malignancy shows the need for an exhaustive evaluation of all such individuals, including physical examination, radiologic and imaging studies, endoscopic studies, and other laboratory evaluations , 8, 21. Most paraneoplastic dermatoses disappear when the primary tumor is removed and reappear in the case of recurrence or metastases of the cancer . In abouThereby, though a rarely observed entity, our case supports the validity of the Leser-Trélat sign and its relevance to the affected individuals, as the investigation may lead to an early diagnosis of yet low-stage cancer and then, with prompt treatment, offer a good prognosis.


Parathyroid carcinoma is an extremely rare malignant endocrine neoplasm that is very challenging in its diagnosis as well as its treatment. Clinically the disease is detected earlier in patients who present with hyperparathyroidism with signs of profound hypercalcemia. Differentiation between benign and malignant disease of the parathyroid is challenging both for the clinician and for the pathologist. Complete surgical resection at the time of first operation offers the best chance of cure. Even after radical excision which is the standard management, local recurrence and metastases are frequent. The disease usually has a slow indolent course and most patients suffer from complications of hypercalcemia rather than tumor invasion or metastasis.We report a case of a 31-year-old woman who presented with renal colic. Various hematological, biochemical and radiological investigations were performed and a slightly enlarged right parathyroid was found. A clinical diagnosis of parathyroid adenoma was made and a right parathyroidectomy was done. Intraoperatively the surgeon had no suspicion of malignancy but microscopically the lesion was malignant and a final diagnosis of parathyroid carcinoma was rendered based on the criteria of invasion. Since there is no gold standard, a multidisciplinary approach, including the entire clinical, biochemical, radiological and pathological profile of the disease aids in an accurate diagnosis. Here we are reporting a case of a functional parathyroid carcinoma presenting in a relatively young patient with all the biochemical and radiological investigations and intraoperative findings pointing towards a benign parathyroid disease.  Parathyroid carcinoma (PCA), a rare endocrine malignancy first described by de Quervain in 1904, occurs either sporadically or as a part of a genetic syndrome . Prior nWe herein report a case of a young female patient who presented clinically with features of hyperparathyroidism. A 31-year-old woman presented to the emergency department with renal colic. She also complained of nausea, vomiting, heart burns and persistent pain in the back and legs for several months. Routine hematological investigations revealed a slight elevation of the serum calcium levels to 11 mg/dl and serum creatinine to 1.7 mg/dl. In view of the raised serum creatinine, calcium and persistent bone pain, a serum parathyroid hormone assay was performed and a raised value of 148 pg/ml was obtained. On clinical examination no neck mass was found. Ultrasonography and 99mTc-sestamibi scintigraphy of the neck revealed only a slightly enlarged right PT. With a clinical diagnosis of parathyroid adenoma a right parathyroidectomy was performed. Macroscopically, the PT was reddish-brown, soft to firm, measured 2 cm and weighed 2.5 g. Cut section revealed a 1 cm whitish zone which appeared fibrous with hemorrhagic areas. Microscopic examination showed normal thyroid and an encapsulated tumor in the parathyroid gland , cyclin D1, parathyroid adenomatosis gene 1 (PRAD1), retinoblastoma (RB), and hyperparathyroidism 2 (HRPT2) tumor suppressor genes have been implicated in the pathogenesis of PCA. Shattuck et al. have documented HPRT2 mutation in 10 of 15 patients with sporadic PCA , cyclin , 7]..7].Differentiating a PCA from an adenoma is challenging for the pathologist. Grossly, parathyroid carcinomas can be indistinguishable from adenomas as in our case though they usually present as larger >3 cm), firm, lobulated, grey-white masses weighing 2–10 g, with a dense, fibrous capsule adherent to adjacent structures. Adenomas in contrast are round to oval, reddish-brown in colour and of a soft consistency  cm, firm. Due to  Since PCA is a slow-growing tumor with distant metastases occurring late in the disease course, aggressive surgical approach is recommended to avoid local recurrence . CompletRecurrent disease occurs in more than 50% patients of PCA . MetastaSurvival rates vary from 90% at 5 years to 67% at 10 years in patients who undergo complete en bloc tumor resection , 8]. Ly. Ly8]. LPCA is a very rare disease with a delayed diagnosis especially when the patient has non-specific symptoms, the tumor is non-palpable and the serum calcium and parathyroid hormone are not too high. In this case neither the clinical symptoms nor the serum calcium and parathyroid hormone levels indicated severe hyperparathyroidism as usually seen in PCA. The patient’s young age for carcinoma was also quite unusual. Moreover, the preoperative localizing studies and intraoperative gross evaluation did not raise a suspicion of the presence of a parathyroid carcinoma. Postoperatively, only the histopathological examination of the clinically suspected adenoma revealed a diagnosis of PCA. Even the gross findings of the resected parathyroid favored an adenoma. For the clinician challenge lies in differentiating hyperparathyroidism caused either by a PCA or its benign counterpart which is much more common. Patients of PCA with persistent hypercalcemia should undergo thorough investigations with a lifetime follow-up since recurrences upto 20 years have been reported . Due to The authors declare that they have no competing interests.


Verrucous proliferation arising from odontogenic cysts is a rare entity. We report an unusual case of an infected odontogenic cyst with verrucous proliferation and melanin pigmentation in a 13-year-old male patient who presented with an intraoral swelling in relation to impacted teeth 26 and 27. The enucleated lesion was diagnosed as an odontogenic keratocyst and the patient died within two years of presentation due to multiple recurrences. The clinical, radiological, and microscopic features of the lesion are presented with an attempt to discuss the etiopathogenesis. The case hereby reported is uncommon with only eight cases reported in the literature. Odontogenic cysts have a diverse morphology. Though their origin and clinical presentations have been well documented, they continue to puzzle the pathologists with few rare clinicopathological presentations. Verrucous proliferation in a cystic lining is one such rarity. To the best of our knowledge only eight cases of odontogenic cysts with verrucous proliferation have been reported in English literature –8, of whA 13-year-old male patient reported with a complaint of swelling and fetid discharge in upper left posterior tooth region since few months. On clinical examination, a solitary swelling of the left maxillary molar segment with a nonulcerated mucosa (except for a draining sinus) was evident with missing 26 and 27. The swelling was firm, had well-defined borders, and was nontender on palpation. Radiographically, a well-defined radiolucent cystic lesion surrounding impacted 26 and 27 was seen . Based oGrossly, the excised tissue measured about 6.6 × 2.5 × 2.2 cm and the whitish-brown cystic lining showed focal areas of nodular/papillary proliferation .Microscopic examination of the specimen revealed a keratin-filled cystic lumen lined by a discontinuous five to fifteen cells thick para- to orthokeratinised epithelium Figures  and 4 wiThe supporting collagenous connective tissue capsule showed moderate to dense mixed inflammatory cell infiltrate and numerous dilated blood vessels. Many melanophages were also seen juxta-epithelially . Focal aThe intraosseous location of the cyst in close association with impacted teeth suggested an odontogenic origin. The histopathological appearance was closest in compatibility to the diagnosis of an odontogenic keratocyst while not being typical of it. After due diligence and recognising the variation from classical appearances, a diagnosis of inflamed odontogenic cyst exhibiting verrucous proliferation was given. Despite the presence of a prominent verrucous component, a distinct lack of endophytic growth and “pushing” epithelial ridges precluded a diagnosis of verrucous carcinoma. Gel electrophoresis (GeneRuler ThermoFisher Scientific) confirmed the presence of human papilloma virus DNA in the tissue specimen. The lesion reportedly recurred thrice over the next two years and curettage was performed but the specimen was unavailable to us for histopathological examination at each of these recurrences. The lesion subsequently extended to the nasoethmoid region involving the orbit and perforating the base of skull anteriorly. The lesion eventually metastasized to the brain resulting in death.Verrucal proliferation in odontogenic lesions is an extremely rare occurrence with only a handful of cases reported in the literature till date. The demographic, clinical, radiographic, and histopathological diagnostic information of the eight reported cases of odontogenic cysts with verrucous proliferation (including verrucous carcinoma) has been compared in Aldred et al. (2002) reported the first case of verrucous proliferation in an odontogenic cyst that occurred in a 13-year-old female patient in right maxillary alveolus, in relation to an impacted canine and the root of the lateral incisor. It was enucleated and did not recur . The preConventionally, a cystic lesion like an OKC/KCOT is known for its aggression but is not commonly known to metastasize. Cases of OKC have been described in the literature wherein maxillary lesions have extended to involve the orbit, skull base, and infratemporal fossa –11.The occurrence of verrucous proliferation in an intraosseous cyst causes one to speculate on the supposed cause. Various etiologies for verrucous growth in odontogenic lesions proposed thus far include the presence of HPV (human papilloma virus), candidal infection, and the habit of tobacco consumption as these factors have correlated well with oral mucosal verrucous lesions . HoweverHuman papilloma virus (HPV) is strongly associated with oral verrucous lesions, verrucous cysts seen in skin, and even verrucous proliferation in ameloblastoma –14. HPV Another histopathologically notable variation seen in the present case was melanin pigmentation and melanin incontinence which is unusual in an odontogenic lesion. One may surmise that the origin of the dental lamina  from primitive oral mucosa may justify the presence of melanocytes , 17. NeuAldred et al. have suggested the use of the term “verrucous odontogenic cyst” . HoweverRegardless of whether such a lesion arose de novo or through transformation of a preexisting lesion, an “odontogenic cyst with verrucous proliferation” is certainly a rare entity. Of the nine cases reported with verrucous growth (including the present case) two have been reported in 13-year-old children and six were reportedly signed out as verrucous carcinoma. The patient in the present case succumbed within two years of diagnosis of the lesion due to recurrences, thus giving an insight into the aggressive behaviour of the lesion which was most likely to be due to infection by HPV. While only three of the nine cases (including present case) were assessed for presence of HPV, positivity for the HPV genome was seen only in our case see .The report of the present case emphasizes the need for careful enucleation and thorough histopathological examination of aggressive cystic lesions. It also warrants the need for running advanced diagnostics to identify presence of HPV infection as it might be a factor determining the prognosis of such lesions.Odontogenic cysts are known to present with numerous variations, verrucous proliferation, and pigmentation being one of the rarest histological presentations. These lesions tend to be aggressive in behaviour even bearing a diagnosis of verrucous carcinoma.The likely association of these lesions with HPV infection warrants careful and thorough investigations including PCR for detection of HPV genome and PET scans as follow-up for recurrence. Through the present report we record the ninth case of odontogenic cyst with verrucous proliferation, with a review of the previous cases in terms of histopathology and possible etiopathogenesis.


In this report, we seek to shed light on a 44-year-old Caucasian male with a known history of an esophageal diverticulum, who was transferred to our facility after an upper endoscopy at an outside hospital suggested a purulent discharge emanating from the mouth of a mid-esophageal diverticulum. A barium swallow done at the outside institution had reportedly demonstrated an 8 cm long barium collection parallel to and anterolateral to the mid-and distal esophagus which terminated several centimeters proximal to the gastroesophageal junction. At our facility, antibiotics (piperacillin/tazobactam) were continued, and a double-contrast esophagram was performed. The presence of an unusual mid-esophageal diverticulum was confirmed. He clinically improved after a 3-day course of intravenous broad-spectrum antibiotics. No surgical or endoscopic repair was elected as the patient opted for continued medical management. While esophageal diverticula are not rare in humans, to our knowledge, this is the first report of development of esophageal diverticulitis in humans. We believe that antibiotic coverage in addition to dietary restriction is the logical mainstay of acute therapy. Optimal antibiotic coverage should likely include oral flora aerobes and anaerobes. Once symptoms resolve, diverticula may be managed expectantly. While esophageal diverticula are not rare in humans, development of esophageal diverticulitis in humans has not been reported in the literature. In this report, we aim to describe a 44-year-old Caucasian male who was confirmed to have esophageal diverticulitis at our facility and underwent successful management. After description of the case, we shed light on pathogenesis and clinical presentation of this condition followed by a brief discourse on management options.A 44-year-old Caucasian male with a known history of an esophageal diverticulum, gastroesophageal reflux (GER), and dysphagia since the 1980s was transferred to our hospital after an upper endoscopy at an outside facility. Upper endoscopy there suggested a purulent discharge emanating from the mouth of a mid-esophageal diverticulum. Of note, he had presented to the outside facility with moderate to severe chest pain radiating to the back accompanied by fever to 38.9 °C and leukocytosis. A barium swallow revealed an 8 cm long barium collection parallel to and anterolateral to the mid-and distal esophagus which terminated several centimeters proximal to the gastroesophageal junction. CT imaging demonstrated mild circumferential distal esophageal wall thickening. He described a twenty five year history of periodic, stereotypic episodes of prodromal pleuritic chest pain followed by fever to 38.9 °C, and sustained chest and epigastric pain. Such episodes had been occurring 1 - 2 times yearly and would resolve over 3 - 5 days with oral antibiotics. More severe episodes had been requiring hospital admission for IV antibiotics. Long-standing intermittent solid food dysphagia had required him to drink liquids when ingesting solid foods.At our facility, antibiotics (piperacillin/tazobactam) were continued, and a double-contrast esophagram was performed. The presence of an unusual mid-esophageal diverticulum was confirmed . EsophagAerococcus viridians infection in a loggerhead sea turtle (Caretta caretta) has been described [Esophageal diverticula are not rare but to our knowledge, this is the first report of esophageal diverticulitis in humans to be reported in the literature. However, an esophageal diverticulum associated with an escribed .Zenker’s diverticulum [Esophageal diverticula can be congenital or acquired . Acquirerticulum , terminarticulum , and thorticulum .As esophageal diverticulitis has not been reported in humans, the mechanism of its development in our patient is speculative. Ingested food particles may lodge in the distal recesses or mouth of the diverticulum resulting in stasis. In regard to this case, the predisposing factor may have been the very unusual anatomy of the diverticulum with very long length and small entrance likely predisposing to stasis.Signs and symptoms may include severe inspiratory chest pain, fever with chills and rigors, dysphagia, odynophagia, and leukocytosis, along with regurgitation, choking, nausea, and vomiting.Differential diagnoses include angina/myocardial infarction, pleuritis/pericarditis, gastroesophageal reflux disease, motility disorders , paraesophageal hernia with infection, esophageal stricture, and esophageal cancer. Diagnosis of esophageal diverticulitis may be made with the combination of clinical assessment, CBC with differential testing, barium swallow, CT scan and upper endoscopy.Antibiotics and dietary restriction are the logical mainstay of acute therapy. Optimal antibiotic coverage should likely include oral flora aerobes and anaerobes; it is unclear whether gram negative rod coverage is necessary. Once symptoms resolve, diverticula may be managed expectantly. Recurrent or severe episodes may warrant endoscopic or surgical  management options as have been described for such symptomatic lesions outside of an acute diverticulitis scenario .


The adrenal gland is the most common site, but myelolipomas are also rarely present in extra-adrenal sites, including the pelvis, mediastinum, retroperitoneum, and paravertebral region, as an isolated soft tissue mass. These tumors account for 2.6% of the primary adrenal masses with equal sex distribution and in our reviewed material of a decade they constitute about 5% in a series of surgically treated adrenals for various neoplastic processes of the adrenals. Although usually small in size, in our series a remarkable large size of the tumors examined was observed, ranging from 7 - 15 cm. Adrenal myelolipoma is often an “incidentaloma”, since its diagnosis is frequently based on autoptic findings or made during surgical interventions and imaging procedures performed for other purposes, as happened in our cases.Myelolipomas are unusual benign tumors or tumor-like lesions, composed of hematopoietic cells and mature adipose tissue. They usually are asymptomatic and behave as non-functioning, unilateral, small adrenal tumors often found incidentally on imaging studies. We report the clinicopathological characteristics of four cases of adrenal myelolipomas treated in our hospital, worth to mention because of their rarity and their significant size. Myelolipomas were first described by Gierke in 1905, and the term  Myelolipomas are unusual benign tumors or tumor-like lesions, composed of hematopoietic cells and mature adipose tissue. Most cases develop in the adrenal glands but extra-adrenal lesions are also reported, arising in the retro-peritoneum, mediastinum, liver and lungs, as well as in heterotopic adrenals . They usA 57-year-old male complaining of chronic back pain underwent an abdominal computed tomography (CT-scan), which revealed a large retroperitoneal tumor probably of adrenal origin. Open surgical approach was considered appropriate as long as malignancy could not be excluded definitely. Right adrenalectomy was finally performed and pathological examination showed a 7 × 5 × 4 cm adrenal myelolipoma composed of mature adipose tissue and hematopoietic cells and weighting 250 g. On the tumor periphery a narrow zone of healthy adrenal tissue was observed. The patient was discharged on the fifth postoperative day, and remains disease free three years later.A 40-year-old white woman with severe anemia of unknown origin was investigated because of enlarged spleen. During investigation an abdominal CT-scan showed a large right adrenal tumor ranging about 15 cm, which was an incidental finding. After proper hormonal laboratory studies, and exclusion of pheochromocytoma and Cushing’s syndrome, operative treatment was decided. The patient underwent an uneventful open right adrenalectomy. Pathology report revealed a 15 × 7 × 6 cm yellow tan, round and firm mass, weighting 420 g, which microscopically consisted of mature adipose tissue mixed with elements of bone marrow, finally diagnosed as myelolipoma. Patient is healthy and disease free 5 years after surgery.A 45-year-old woman with a personal history of cholelithiasis, arterial hypertension and elevated urine cathecholamines was admitted to our hospital for further imaging diagnostic examinations and treatment. An abdominal CT-scan revealed an about 10 cm right adrenal mass, which was considered to be a pheochromocytoma. After appropriate preoperative preparation with the use of phenoxybenzamine and propranolol the patient was operated in an open fashion. Pathological examination showed a large adrenal tumor weighting 185 g tumor and measuring 10 × 8 × 3.5 cm. Grossly the tumor was yellow-brown, with elastic consistency and microscopically presented the typical myelolipoma features.A 74-year-old woman with the history of breast carcinoma, under treatment, was diagnosed with a large adrenal tumor, about 8 cm in diameter during routine follow-up ultrasound of the abdomen. Abdominal CT-scanning followed and the adrenal tumor was radiologically heterogeneous with non-specific imaging features. Surgical intervention was decided because of inability to safely exclude malignancy, either primary or metastatic. Pathological examination of the specimen showed a myelolipoma weighting 120 g and atrophic changes of the adrenal cortex.myelolipoma was coined by Oberling in 1929 [Myelolipomas are rare benign tumors composed of mature adipose tissue and hematopoietic elements. They were first described by Gierke in 1905, and the term  in 1929 .The exact etiology of adrenal myelolipomas is still unknown and their histogenesis is uncertain. According to some authors, the tumor develops from residual embryonic cells of the bone marrow that, after embolization reach the adrenal gland. According to a recent theory, myelolipoma originates from a metaplastic process of the reticuloendothelial cells within adrenal capillaries. Some authors even consider the development of adrenal myelolipomas as a response to various endocrine stimulations . This hyThese tumors account for 2.6% of the primary adrenal masses with equal sex distribution . They arAlthough most adrenal myelolipomas are usually asymptomatic, abdominal/flank pain and an abdominal mass are the most common presenting symptoms. The compression of neighboring structures by large tumors (mass effect), infection, tumor necrosis and intra-tumoral hemorrhage are the main complications associated with it. Intestinal obstruction and spontaneous retroperitoneal hemorrhage are rare complications usually encountered with tumors > 10 cm in diameter , 12, 13.Adrenal myelolipoma is often an “incidentaloma”, since its diagnosis is frequently based on autoptic findings or made during surgical interventions and imaging procedures performed for other purposes, as happened in our cases. However, with the advent of cross sectional imaging, the diagnosis of this neoplasm has improved remarkably, both in asymptomatic and symptomatic patients. The imaging features of myelolipoma depend on the varying proportion of fat, hematopoietic tissues, hemorrhage or calcification present within it. Calcification may be seen in these masses in around 27% of cases .Although a predominantly hyperechoic adrenal mass strongly suggests the diagnosis of myelolipoma, ultrasonography is not specific and computed tomography (CT) or magnetic resonance imaging (MRI) should be obtained for further evaluation -16. CT iMyelolipomas are well circumscribed in gross inspection, but are rarely encapsulated. Their color varies from pale yellow to deep red or brown depending upon the relative proportion of fat and hematopoietic elements. The contour of the adrenal myelolipoma is smooth, wavy, or irregular and may shoe intermingling of cortical cells with elements of the myelolipoma. The surrounding cortical cells can be relatively normal in appearance or compressed . There iPreviously, most patients with myelolipoma underwent surgical resection for a suspected malignant neoplasm. Nowadays, with the frequent detection of these myelolipomas incidentally, the treatment has been a matter of much debate. Asymptomatic, small myelolipomas (< 5 cm) are treated conservatively with 6 - 12 months interval follow-up with ultrasound or CT, whereas symptomatic and large tumors (> 10 cm) should be extirpated because of risk of malignant change and spontaneous rupture with retroperitoneal bleed. With the advent of minimally invasive surgery, laparoscopic adrenalectomy has become a standard treatment for both functioning and non-functioning adrenal tumors. In the case of bilateral adrenal myelolipomas, a staged tumor removal is preferable, removing the larger one and continuing to observe the contralateral myelolipoma as long as possible in an effort to avoid adrenal insufficiency and a lifetime of steroid replacement .


Foveolar gastric metaplasia of the duodenum is a frequent but not as yet considered correlate of endoscopically detected duodenal polyps. The majority of foveolar gastric metaplasias associated with polyps presented a typical benign endoscopic appearance and they were diagnosed by biopsy. Here we report a case of a surgical-resected foveolar gastric metaplasia manifesting as a duodenal tumor with an atypical appearance.An asymptomatic 56-year-old Asian man who presented with a foveolar gastric metaplasia of atypical appearance and had previously undergone esophagogastroduodenoscopy was referred to our hospital. A biopsy revealed a normal duodenum with an inflamed mucosa. Narrow band imaging with magnifying endoscopy revealed normal microvessels with normal micromucosa, which indicated non-neoplasia. Endoscopic ultrasonography using a miniature probe system (20 MHz) revealed a hypoechoic mass with multiple anechoic lesions (16-mm diameter) located in the mucosal layer. The lesion was excised via laparotomy assisted by endoscopic techniques similar to endoscopic submucosal dissection. The pathology indicated foveolar gastric metaplasia.Foveolar gastric metaplasia can present as a duodenal tumor. We identified two important clinical issues. First, foveolar gastric metaplasia can present as a duodenal tumor with an atypical benign appearance. Second, both endoscopic ultrasonography and narrow band imaging are useful techniques to increase the diagnostic rate of this condition. Foveolar gastric metaplasia (FGM) of the duodenum is a frequent but not as yet considered correlate of endoscopically detected duodenal polyps. According to a comparison of findings from two institutes with upwards of 100,000 gastroenterological cases per year, the association of FGM with typical polyp morphology was shown to be at higher rates than previously identified [An asymptomatic 56-year-old Asian man who presented with an FGM of atypical appearance and had previously undergone esophagogastroduodenoscopy was referred to our hospital. He had taken antihypertensive medicine for hypertension. He denied alcohol intake and did not smoke tobacco. An abdominal observation revealed that his abdomen was soft and flat and without pain. His neurological findings were normal. The results of laboratory findings were normal Table . A biopsThe esophagogastroduodenoscopy showed no recurrence of the lesion after 10 months.We identified two important clinical issues: FGM can present as a duodenal tumor with an atypical benign appearance, and both EUS and NBI are useful techniques to increase the diagnostic rate of this condition.First, FGM can present as a duodenal neoplasia with an atypical benign appearance. FGM is common in the duodenum, with localized ectopic gastric mucosa . A searcSecond, both EUS and NBI were useful to increase the diagnostic rate of FGM. According to the validity report for EUS for ectopic gastric mucosa lesions, endoscopic resection may be used in cases of duodenal ectopic gastric mucosa when EUS can verify that the tumor is localized in the third layer, which corresponds to the submucosa of the duodenal wall . In thisIn our case, although the lesion was diagnosed as benign, surgical resection was scheduled because of the relatively large size and unusual morphology of the lesion. We must concede that because the lesion was identified as benign, an observation period could have been an acceptable alternative. However, multiple factors led us to proceed with our chosen course of action, and valuable information was obtained.FGM can present as a duodenal tumor for which both EUS and NBI are valuable diagnostic tools. FGM can present as a duodenal tumor with an atypical benign appearance, and this should be considered while treating patients in future. In fact, many occurrences may remain unrecognized and undetected. EUS and NBI are highly recommended to verify and diagnose FGM before resection is considered. Further reporting would be useful to support the evidence that the incidence of FGM may be higher than previously thought, resulting in undiagnosed and untreated cases.


The aim of this retrospective study was to investigate the frequency of intra-articular osteoid osteoma (iaOO) in a large study cohort and to demonstrate its clinical relevance as an important differential diagnosis of non-specific mono-articular joint pain.We searched the registry for bone tumours of the University Medical Centre Hamburg-Eppendorf for osteoid osteomas in the last 42 years. Herein, we present three selected iaOO which were detected in the three major weight-bearing joints. Computed tomography (CT) or magnetic resonance imaging (MRI) scans were performed for initial diagnosis.Out of a total of 367 osteoid osteomas, 19 (5.2 %) tumours were localized intra-articularly. In all three presented tumours, a history of severe mono-articular pain was reported; however, the mean time to correct diagnosis was delayed to 20.7 months. Clearly, the nidus seen in CT and MRI images in combination with inconsistent salicylate-responsive nocturnal pain led to the diagnosis of iaOO.Rarely, osteoid osteoma can occur in an intra-articular location. In cases of diffuse mono-articular pain, iaOO should be considered both in large and smaller joints to avoid delays in diagnosis and therapy of this benign bone tumour.The online version of this article (doi:10.1186/s12891-016-1313-3) contains supplementary material, which is available to authorized users. Osteoid osteoma (OO) was first described as a specific entity by Jaffe in 1935 . OO may Localizations within the skeleton may vary, although the diaphysis in long bones of the lower extremity is most commonly affected. Intra- and juxta-articular localizations have been described, but only account for around 10 % of OO –9. EspecBesides making the correct diagnosis, the challenge is that various therapeutical options including arthroscopic removal and radiofrequency ablation (RFA) are available , 12–14. This retrospective study searched the registry for bone tumours at the University Medical Centre Hamburg-Eppendorf for all detected osteoid osteomas from 1974 to 2016. Age, sex, radiological investigations, tumour location, histopathological features including type and dignity of the tumour and diagnosis were documented. The registry collected both slides and tissue blocks (both paraffin and methyl-methacrylate) from tumour biopsies and resection specimens as well. All clinically assumed OO that could not be confirmed histologically were excluded. Therefore, 367 cases with histopathologically diagnosed OO were included in this study. Age and gender distribution, as well as the frequency of iaOO among all OO was evaluated with Microsoft Excel software.Haematoxylin and eosin (paraffin blocks) and toluidine blue staining methods (methyl-methacrylate embedded blocks) were performed for the purpose of the present study. Microscopy was carried out using a Zeiss microscope  and photographs taken with AxioVision Rel.4.8 . Furthermore, three of the most recent cases of iaOO of the lower extremity were selected and are presented here. These cases were all seen and clinically diagnosed at our institution.n = 3) and distal (n = 1) tibia, proximal (n = 2) and distal femur (n = 1), spine (n = 2), talus (n = 4), calcaneus (n = 2) and hand (n = 4), respectively , followed by localization in the tibia (27.8 %) and in the vertebra  at rest, occurring predominantly at night. No typical response to salicylates was reported in the beginning. Physical examination showed no signs of ankle instability. Arthroscopy was performed due to suspected synovitis but histology revealed that this was not the case. Extended laboratory tests did not point to any rheumatological diseases. Furthermore, multiple intra-articular injections were unsuccessful in terms of pain reduction. The lesion seen in CT and repeated MRI scans was eventually interpreted as an intra-articular, subchondral osteoid osteoma (3.1 × 2.7 mm) in the anterior process of the talus Fig. , althougAnother 26-year-old patient presented at our clinic with non-specific anterior knee pain after having consulted different orthopaedic surgeons that had suspected meniscus injury, bone marrow oedema and anterior cruciate ligament instability. The patient had worsening pain in the lateral knee, but no trauma was reported. Clinical examination revealed no signs of knee instability. Partial meniscectomy had been performed due to a submeniscal bone impression seen in MRI images and a lateral meniscus tear seen in arthroscopy. However, this intervention did not achieve any improvement of symptoms.Based on the patient’s information about resolving nocturnal pain following aspirin medication, we performed a CT scan that showed the typical nidus at the lateral tibia plateau, which was not as obvious in MRI scans Fig. . Time unThis 22-year old female patient was referred to our outpatient clinic. The patient was an enthusiastic tennis player and had had diffuse pain in the right hip joint for more than 6 months. Nocturnal pain was not the leading symptom; rather, joint tenderness and a restricted range of motion was reported. External leading differential diagnoses in the beginning were synovitis and coxa saltans. Conventional radiographs were inconspicuous Fig. . Due to RF ablation of the OO was performed in order to induce osteonecrosis of the nidus. However, it led to subsequent osteonecrosis of almost the complete femoral neck, which was diagnosed in post-interventional MRI images 3 months after the intervention. In proton density (PD)-weighted images altered signal intensity around the insertion channel of the RF ablation probe pointed to an accompanying marrow oedema. In a 1 year follow-up MRI there was little joint effusion and a constant necrosis zone  was performed and did not point to any primary or secondary bone diseases which could have provoked the osteonecrosis. In fact, DXA revealed a normal bone mineral density (BMD). We advised the patient to avoid uncontrolled movements in order not to risk further necrosis of the femoral head, although she was by then nearly symptom-free at rest. Imbalanced vitamin D and calcium homeostasis in terms of a moderate vitamin D insufficiency (25-OH-D3 20 μg/l) was controlled by daily supplementation with 2,000 IE vitamin D.Osteoid osteoma is a bone-forming tumour with typical localization in the diaphysis of long bones; however, it can be particularly challenging when localized intra-articularly , 15. We Cases 1 and 2. Considering all diagnostic options, CT imaging remained the most specific technique to detect iaOO [Case 2 [Case 3. Radionuclide imaging, which was reported to be a sensitive diagnostic modality [Clinically, the iaOO shown here did not consistently show nocturnal pain. Furthermore, in all three cases plain radiographies were inconspicuous. Other differential diagnoses including synovitis, rheumatological diseases and meniscus or ligament tearing were initially taken into account before suspecting OO. These suspected diagnoses were often the reason for initially carrying out an MRI and subsequent misdiagnosis . Furtherect iaOO , 17, esp [Case 2 , 19 whicmodality , was notThere are several possibilities for therapy of the tumour: a main advantage of the surgical removal of iaOO is the possibility of histopathological diagnosis. The histology shows irregular immature woven bone trabeculae covered by activated osteoblasts and embedded in vascular fibrous stroma with scattered osteoclasts Fig. . In miniWe here present the first case in the literature where RFA led to excessive osteonecrosis of the femoral neck. Most likely this was because there was either too much heat administered, an inappropriate device was used or the total ablation time was too long. We conclude that RFA is only safe when the induced osteonecrosis can be controlled. Especially when localized intra- or juxta-articularly, we highly suggest considering RFA with care, as osteonecrosis was a direct consequence of this procedure. In our case, it has to be expected that the necrosis zone will only revitalize very slowly, if at all. The use of alternative methods including tissue impedance as a parameter of osteonecrosis  or magneIn conclusion, we found 19 iaOO within a total of 367 OO (5.2 %) in our large registry of bone tumours. Furthermore, we demonstrate three cases of iaOO, which all atypically presented with non-specific joint pain and subsequent initial incorrect diagnosis. Based on our data, we recommend considering iaOO especially in young patients presenting with persistent joint pain of both small and large joints. Furthermore, we suggest arthroscopy with subsequent removal of the tumour and histopathological work-up of the lesion.


Primary intracranial osteosarcoma is a extremely rare disease entity. We describe a case of primary intracerebral osteosarcoma in an adult brain.A patient who presented with a 1-week history of headaches, and MRI examination was performed. The immunohistochemical diagnosis confirmed primary intracerebral osteosarcoma. The patient was treated with a surgical resection of the tumor.Primary osteosarcomas occurring in the brain are extremely rare. The MRI images did not provide a specific pretreatment diagnosis, and the histopathology was the mainstay in establishing the diagnosis. Osteosarcoma is a highly malignant neoplasm of bone and can occur anywhere in the body, but is most commonly found in the long bones. Extraskeletal osteosarcomas occurring in tissues other than bone, are rare. Primary intracranial osteosarcomas are extremely rare, developing from leptomeningeal sheats around blood vessels or from vessel walls themselves  revealed a large, partially necrotic and calcified tumor in the parenchyma of the left frontal lobe with marked surrounding edema and mass effect on midline structures, but without any dural attachment. The tumor appeared hypointense on non-contrast T1-weighted images (Fig. Extraskeletal osteosarcoma is rare and is defined as a malignant mesenchymal neoplasm that produces osteoid as well as bone or chondroid material and is located in the soft tissue without any bony attachment (Chung and Enzinger We performed a PubMed search for all cases of primary intracerebral osteosarcoma up to September 2016. Cases were analyzed for basic demographic features including age, sex, chief complaint, location, treatment, and clinical outcome (Table Primary intracerebral osteosarcoma is an extremely rare tumor and its radiological appearance is not pathognomonic. Despite their rarity, primary intracerebral osteosarcomas should be included in the differential diagnosis, especially for other tumors such as meningioma and calcified glial tumor, and histopathology is the mainstay in establishing the diagnosis.


It is important for the dentists to make accurate diagnosis and appropriate treatment of odontogenic cutaneous fistula. Although large facial skin lesions may bring up malignancy on top of the differential list, careful evaluation including physical observation, imaging, and pathology can rule out malignancies. Intraoral examination showed gingival swelling at the level of left impacted mandibular tooth. Computed tomography images revealed cutaneous ulcer at left submandibular area creating a fistula continuing from left third molar pericoronitis, with osteosclerosis (Fig. An 87‐year‐old man with a history of diabetes presented to our hospital due to dysphagia with skin perforation on his left chin. He was previously diagnosed with a traumatic hairline fracture of the mandible 2 weeks before. Observation revealed a 5‐cm ulcerated lesion in left submandibular region Fig.  with purAB: drafted the article. AB, YO, TS, and HO: participated in critical review and revision of the article, gave the final approval of the article, and have accountability for all aspects of the work.None declared.


Calcified amorphous tumor of the heart is a rare, non-neoplastic cardiac mass characterized by nodular calcium in the background of amorphous degenerating fibrinous material. Clinical diagnosis of calcified amorphous tumor can be difficult, and current single imaging techniques do not specifically differentiate calcified amorphous tumor from other cardiac tumors such as calcified atrial myxoma, calcified thrombi, or vegetation. Complete surgical resection is the treatment of choice for both symptom improvement and prevention of embolization, as well as for pathological diagnosis.A 70-year-old Asian man with end-stage renal disease complained of chest discomfort during exercise. He had no history of thromboembolism or endocarditis. A transthoracic echocardiogram revealed mitral annular calcification as well as a highly mobile mass (8 × 6 mm) attached to the ventricular side of the posterior mitral valve leaflet. As the mass was highly mobile, suggesting a high risk of embolization, he underwent surgical resection. A histopathological examination revealed multiple nodular amorphous calcifications, along with fibrous connective tissue. There were no identifiable myxoma or malignancy cells. Consequently, the diagnosis of calcified amorphous tumor was confirmed.In the present case, a calcified amorphous tumor arose from mitral annular calcification. A characteristic of mitral annular calcification-related calcified amorphous tumor is its highly mobile nature, with a high risk of stroke or other systemic embolism. Therefore, surgical therapy should be considered for treatment of calcified amorphous tumors. Calcified amorphous tumor (CAT) of the heart is a rare, non-neoplastic cardiac mass characterized by nodular calcium in a background of amorphous degenerating fibrinous material . AlthougA 70-year-old Asian man with ESRD due to nephrosclerosis complained of chest discomfort during exercise and was referred to our hospital. He had been receiving hemodialysis for 20 years, and had a past medical history of hypertension, hyperuricemia, and reflux esophagitis. He had no history of thromboembolism or endocarditis. He smokes one pack of cigarettes per day, and drinks socially.He had no significant social and environmental history. His medication list included amlodipine, allopurinol, ferric citrate hydrate, and lansoprazole. He had no family history of cardiovascular diseases.3, and platelets 134,000/mm3. His liver and renal function tests revealed total bilirubin 0.4 mg/dl, aspartate aminotransferase 24 IU/l, alanine aminotransferase 34 IU/l, blood urea nitrogen 54.2 mg/dl, and serum creatinine 11.1 mg/dL. His C-reactive protein was 0.16 mg/dl. His serum electrolyte tests were within normal limits, including calcium and inorganic phosphorus. In addition, his brain natriuretic peptide was raised at 605.1 pg/dl. His blood cultures were negative.He was afebrile, with a blood pressure of 114/50 mmHg and a regular pulse rate of 61 beats per minute. His oxygen saturation was 96% on ambient air. There was no evidence of lung rales, cardiac murmur, and abdominal tenderness. His neurological examinations were unremarkable. Blood investigations showed: hemoglobin 14.0 mg/dl, white blood cells 4700/mmA chest X-ray showed mild cardiomegaly without pulmonary congestion Fig. . An elecMacroscopic findings of the resected mass showed a yellowish colored and partially calcified mass (7 mm in diameter; Fig. No evidence of tumor recurrence was detected after the surgery, and he is doing well after 6 months of follow-up.CAT is a rare non-neoplastic cardiac mass. Although the origin of CAT is unclear, approximately 20% of patients with CAT have ESRD , suggestIn the present case, the CAT arose from MAC. Previous cases of MAC-related CAT have been reported Table , and thiFurther, MAC-related CAT had a highly mobile characteristic, with a high risk of stroke or other systemic embolism. Finally, all of those cases had received surgical resection. Thus, because of the high risk of cardiovascular events in patients with MAC alone , MAC-relWe reported a case of MAC-related CAT. In general, MAC-related CAT is considered a high risk for stroke or other systemic embolism. However, it is difficult to diagnose CATs without pathological findings. Therefore, surgical therapy remains the optimal approach for treatment of CATs.