Nasopharyngeal angiofibroma presents with symptoms of nasal obstruction and epistaxis. The treatment of choice is embolization followed by surgery.A 52-year-old man underwent surgery for nasopharyngeal angiofibroma after adjuvant radiofrequency-induced thermotherapy. To the best of the authors' knowledge, this is the first case of angiofibroma with clinical follow-up after thermocoagulation therapy supported by quantitative, double immunohistochemistry. We found this case of angiofibroma to be of interest owing to the presentation of symptoms leading to biopsy, the pathohistological observations obtained with synchronous Ki67/cluster of differentiation 34 and Ki67/smooth muscle actin immunohistochemistry and high pericyte proliferation.Coagulation of angiofibroma vessels followed by acquisition of a thick mantle of pericytes in a patient with a nasopharyngeal growth suggests that radiofrequency-induced thermotherapy could be a useful, palliative therapy for bleeding nasopharyngeal angiofibroma, supporting vessel maturation prior to surgical tumor removal. Nasopharyngeal angiofibroma is considered to be a reactive, malformed, benign but aggressive neoplasm. Clinical staging and tumor embolization reduce surgical morbidity. The therapy protocol is influenced by hospital-related factors. Radiofrequency-induced thermotherapy (RFITT) is a minimally invasive surgical procedure that causes thermal ablation through coagulation and is used in the treatment of both head and neck diseases. We were unable to find reported cases of angiofibroma that were treated with RFITT, subjected to follow-up evaluation and had documented histological changes with time.We present an unusual case of a 52-year-old man with nasopharyngeal angiofibroma that first appeared as a nasal polyp. Coagulation, thrombosis, sclerosis and pericyte proliferation occurred after RFITT. We looked for a change in angiofibroma cell proliferation through biopsies obtained before and after RFITT when the patient was free of bleeding episodes. The cell origin of vessel formation after thermocoagulation therapy was investigated. Our results are of interest for surgeons applying pre-operative thermal ablation therapy.3) was diagnostic for nasopharyngeal angiofibroma after routine hematoxylin and eosin (H&E) staining  showed a soft tissue lesion in the mediosagittal line, suggesting a nasal polyp. A biopsy (18 February 2005) of the polyp revealed that it was immovable and provoked bleeding. The provided tissue , the patient was subjected to RFITT using a Celon AG medical instrument . The therapy was performed three times over a 2-month period . The lesion did not bleed but hardened. The second surgical specimen (21 September 2005) was 5 cm3 of residual tumor with an overall Ki67 proliferation index of 10%. Plump SMA-positive and predominantly Ki67-negative cells were detached from the vessel wall and formed sheets resembling angiomyofibroblastoma after H&E staining. The second and third biopsies respected the recovery time from RFITT and were not complicated by hemorrhage.A third biopsy 10 months after RFITT provided 0.075 cmOne year after RFITT, angiography found no arteries feeding the residual tumor. The patient underwent surgery at another institution without prior embolization (no hypertrophic feeding arteries were found at repeated angiography before the operation).The primary intention was to reduce the tumor and alleviate the symptoms using RFITT before the operation. Double immunostaining was planned later because of increased Ki67 staining observed in the control biopsy after RFITT. Ki67 is a proliferation marker providing nuclear staining when the cell is in the S phase preparing to enter mitosis. To determine which cell type is proliferating in a tissue, a second differentiation marker is added, that is, CD34 for endothelial cells or SMA for pericytes. The immunohistochemical analysis of all three angiofibroma biopsies was repeated with a double-staining technique for both Ki67/CD34 and Ki67/SMA to distinguish between endothelial cell and pericyte proliferation over time Figure  and 4C. Double immunohistochemical staining revealed higher proliferation indices for cells of the vessel compartment compared with single Ki67 staining of each routine biopsy. The EPI slightly decreased while the PEPI increased 10 months after RFITT. The third biopsy contained a large number of detached SMA-positive cells. There were scattered Ki67-positive nuclei of cells outside the vessel wall that were defined by neither CD34 nor SMA in all three biopsies. The MVD increased 20 days after RFITT and further increased with time. The PCI also increased with time. Measurements and images were obtained using a BX-40 Olympus microscope, Sony CCD-Iris color video camera and ISSA 3.1 software .Nasopharyngeal angiofibroma is considered a malformation in juveniles -3, but dThe case presented here is of interest from both the clinical and the pathological points of view. The nasopharyngeal and sinonasal tracts are sites of different pathologies prone to epistaxis, such as the angiofibroma, angiectatic nasal polyp , and necIn our case, two pathologies were present synchronously, a mucosal nasal polyp and an angiofibroma, making the diagnosis more complex as noticed by other authors . The assThe origin of angiofibroma is still under investigation. Zhang et al.  presenteHistologically, both endothelial cell and pericyte proliferation were more accurately expressed with double immunohistochemistry compared with routine Ki67 staining. Pericyte proliferation was stronger than endothelial cell proliferation prior to therapy . While the PEPI increased upon coagulation and progressed with time, the EPI did not. These results support the theory of angiofibroma as a maturing vasoformative lesion. Vessel formation is observed in inflammation, malformation, neovascularization of neoplasia and as a neoplastic event. Proliferation in vascular malformations has been studied previously ,11. VessWe have presented a rare case of angiofibroma in a 52-year-old man with pericyte proliferation, supporting the maturation of the vessel compartment and revealing active angiogenic machinery . We observed the divergent behavior of endothelial cells and pericytes after RFITT adjuvant therapy prior to surgery. Further studies of RFITT related to vessel behavior are needed. We found thrombosis and coagulation resulting from RFITT to function as equivalent to embolization prior to surgical therapy for angiofibroma. An analysis of vessel cell proliferation in tissues treated with thermal ablation might have broader clinical impact across medicine.CD; Cluster of differentiation; CT: Computed tomography; EPI: Endothelial cell proliferation index; H&E: Hematoxylin and eosin; MVD: Microvessel density; PCI: Proliferating capillary index; PEPI: Pericyte proliferation index; RFITT: Radiofrequency-induced thermotherapy; SMA: Smooth muscle actin.The authors declare that they have no competing interests.MKr is the author of this study and performed the quantitative analysis of the double-stained immunohistological slides. MKu, TB and NC are surgeons who treated and observed the patient and provided the angiofibroma biopsy specimens. AH is our radiologist responsible for the acquisition of data and analysis and interpretation of data.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Non-occlusive mesenteric ischemia is not uncommon in chronic hemodialysis patients and is the major cause of an acute abdomen in this population. Intensive ultrafiltration and intradialytic hypotension are usually the precipitation factors. A definite diagnosis is usually late and associated with high mortality. We present a rare case of a patient who developed abdominal symptoms during his first week on HD without having obvious hypotensive episodes.th post-operative day.A 76-year-old man was admitted with pulmonary edema and renal failure developed abdominal symptoms during his first week on hemodialysis without having obvious hypotensive episodes. Abdominal diagnostic procedures were all unrevealing. Mesenteric ischemia was diagnosed during laparoscopy done on the basis of physical findings and clinical suspicion. Ischemic small bowel of the distal ileum was resected and histopathology examination of the small bowel demonstrated transmural ischemic necrosis with hemorrhages and non-occluded mesenteric artery. Patient maid a steady recovery, and was discharged on the 11Mesenteric ischemia should be systematically suspected in dialysis patients experiencing even mild and nonspecific abdominal symptoms with or without hemodialysis-induced hypotensive episodes. Identification of patients at risk and prevention of intradialytic hypotension may help to reduce the incidence of this potentially fatal complication in hemodialysis patients. Recen century ,4-6, wit century , since m century ,4.We present a rare case of a 76-year-old man who developed abdominal symptoms during his first week on HD without having obvious hypotensive episodes. Abdominal diagnostic procedures were all unrevealing. Laparotomy showed NOMI of small intestine.2) and was not on dialysis.A 76-year-old man with known history of chronic kidney disease, cardiac disease, hypertension and chronic obstructive lung disease was admitted to our hospital with pulmonary edema. The patient had kidney disease of unknown etiology with baseline serum creatinine of 4.0 mg/dL  scan revealed distended gallbladder with sludge and diverticulosis of the sigmoid colon. Upper endoscopy (EGD) showed gastritis. Intravenous antibiotics with ceftriaxone and metronidazole started for presumed acute acalculous cholecystitis. Over next week the patient had few hypotensive episodes during HD with systolic blood pressure of 70–80 mm Hg associated with marked increase of abdominal pain. Rebound abdominal tenderness and leukocytosis with WBC count of 24.5 × 103/μL with 90% neutrophiles noted. Repeated abdominal CT scan with intravenous contrast showed mild ascites and increased small bowel distention  count was 12 × 10In recent years, NOMI has become increasingly recognized as a highly dangerous complication in dialysis patients. Patients with end-stage renal disease have many risks factors for the development of mesenteric ischemia. These include generalized atherosclerosis, congestive heart failure, diabetes mellitus and increased time on dialysis , the useMesenteric ischemia may appear more frequently among dialysis patients than in the non-dialysis population. In the general population, mesenteric ischemia is of an occlusive type, usually due to atherosclerotic thrombosis of the proximal portion of the superior mesenteric artery affecting the left colon and sigmoid . In contThe diagnosis of mesenteric ischemia is difficult and requires a high level of suspicion since a delay in an early diagnosis leads to a high mortality rate ,5. John Angiography findings can confirm diagnosis of arterial occlusion and indicate NOMI when 'defoliated tree' that refers to the absence of contrast dye in the smaller mesenteric vessels is present . AngiogrThe known difficulty in diagnosing intestinal ischemia and the role of early, definitive surgery was exemplified in the group of 12 chronic HD patients admitted to the John Hopkins Bayview Medical Center because of acute abdominal pain with leukocytosis . AbdominMesenteric ischemia is a potentially fatal complication in chronic HD patients and is the major cause of an acute abdomen in this population. An early diagnosis is difficult to establish. A definite diagnosis is usually late and associated with high mortality. To our knowledge, this is a first report of NOMI that occurred at the fist week of chronic HD therapy and without hypotensive episodes. As our report illustrates, mesenteric ischemia should be systematically suspected in patients experiencing even mild abdominal symptoms during or after HD sessions, especially in elderly patients with atherosclerotic cardiovascular disease even without HD-induced hypotensive episodes. Identification of patients at risk and prevention of intradialytic hypotension may help to reduce the incidence of NOMI in chronic HD patients.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.ZZB and MB analyzed and interpreted the patient data. HKO performed histopathological studies. JFW was responsible for manuscript editing. All authors read and approved the final manuscript.


Cholesterol granuloma (CG) may involve the middle ear, the mastoid bone and the petrous apex. However, CG presenting as a mass obstructing the external ear canal (EEC) is relatively rare and it can be a diagnostic challenge.We report a case of a CG occupying the mastoid antrum and presenting as a mass into the EEC. Temporal bone computerized tomography showed a soft tissue mass which eroded the posterior-superior bony wall of the EEC. On magnetic resonance imaging, the mass revealed a high signal on both T1 and T2-weighted images. The CG was removed by a mastoidectomy procedure and the histopathologic report confirmed the diagnosis of CG. A type III tympanoplasty was performed.The postoperative course was uneventful. Cholesterol granuloma (CG), or cholesterol cyst, is a clinical entity that was first reported by Manasse in 1894 ,3. This A 65 year old male presented to our ENT clinic as an outpatient for a routine yearly check up. He had a history of chronic otitis media with subtotal ear drum perforation in both ears. Left myringoplasty was performed thirty years ago. Otoscopic examination revealed stenosis of the right EEC due to the presence of a mass occupying the superior-posterior part of the canal, which prevented visualization of almost half of the perforated tympanic membrane Figure . The midTemporal bone CT Figure  and 3 shBlood work was normal. The serum cholesterol levels of our patient were within normal limits. The cyst was drained and the brownish semi-liquid material was cultivated; no microorganisms where found. Cholesterol was found to be present in the effusion (274 mg/dL).Through a postauricular approach the patient underwent mastoidectomy. The mastoid antrum was entered and the cyst was excised along its direction to the EEC. The posterior canal wall was reconstructed with a piece of auricular cartilage. Then a type III tympanoplasty was performed. The postoperative course was uneventful. The histopathologic report confirmed the diagnosis of CG. The cyst had a fibrous lining and contained cholesterol crystals, haemosidirin and fibrin which were surrounded by foreign-body giant cells.The first theory about the pathogenesis of the cholesterol cyst was proposed by Friedman in 1974  suggesteMicroscopically, the cyst presents cholesterol clefts (the cholesterol dissolves during tissue processing) that are surrounded by histiocytes and foreign-body giant cells . In factThe clinical presentation of the CG is variable since it is encountered in different locations and dimensions. It could be discovered incidentally, as in our case, or can be presented with hearing loss , tinnitus, vertigo, cranial nerve deficits, headache, facial pain/numbness, diplopia and CSF leak ,14. The Computed tomography scanning and magnetic resonance imaging plays a significant role in the diagnosis of CG. The CG appears as a well-marginated lesion in the CT, isodense with the brain, with no significant contrast enhancement while bone erosion may be present. MRI is helpful in order to make differential diagnosis from other non-contrast-enhanced masses such as cholesteatomas and brain hernias and to conduct a proper preoperative plan. The CG has short T1 and long T2 features and amplified signal intensity in both T1 and T2 weighted images .Regarding the treatment of CG, variable approaches are plausible. The treatment plan depends on the location and size of the lesion. There are reports of conservative therapy such as simple patient follow-up with serial neurological exams and MR imaging . MedicalWritten informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.VN study design, involved in drafting the manuscript and revising it critically. HM study design, involved in drafting the manuscript. DP study design, involved in drafting the manuscript. SM study design, involved in drafting the manuscript and revising it critically. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1472-6815/10/4/prepub


Background. Intraabdominal lesions such as mesenteric cysts are uncommon disorders. Most are discovered incidentally during routine abdominal examinations. Methods. We report a patient with a mesenteric cyst masquerading as tuberculous peritonitis and ascites. Conclusion. Mesenteric cysts generally do not show typical clinical findings. They may also present with peritoneal tuberculosis findings such as low albumin gradient ascites with high ascitic adenosine deaminase levels. Surgery is used to remove a wide variety of mesenteric cysts. A correct diagnosis can be made by the combined use of radiographic and sonographic examinations in conjunction with the clinical signs. Mesenteric cysts (MCs) are rare intra-abdominal lesions arising with an incidence of 1/100,000 admissions in adults and 1/20,000in children . Herein μL, consisting predominantly of lymphocytes (>70%). Tumor markers were with the normal range in serum but the ascitic CA-125 level was 336 U/mL . Cultures of the ascitic fluid and blood were negative for mycobacterial colonies. She refused to undergo laparoscopy, most likely due to her busy schedule. The findings were compatible with peritoneal tuberculosis and conventional antitubercular therapy was started. She returned to the hospital six months later. At the time of her second admission, she was well but her abdominal dullness and ascites were persisting. She had not taken any kind of medication for four months. Levels of serum electrolytes, lactate dehydrogenase, creatine kinase, albumin, and total protein and results of renal- and liver-function tests were normal. Repeated abdominal and pelvic CT scanning with contrast medium showed a large MC  showed multiloculated ascites. A computerized tomographic (CT) scan showed ascites with thin intraascitic septations. The serum-ascites albumin gradient was 0.4 g/dL and the adenosine deaminase level of ascites was 49 U/l . Moreover, the ascitic fluid was straw colored with protein >3 g/dL and total cell count of 550 /large MC . Exploralarge MC . The posAlthough MCs can occur at any age, they are common in people between 40 and 70 years old; however, they also affect children younger than 10 years old. MCs usually range in size from a few centimeters to 10 cm but they can also be very large . In our Small MCs may be asymptomatic but large cysts can present as acute abdominal emergencies such as acute intestinal obstruction, or volvulus, or sudden hemorrhage into the cyst, or infection . In our The cyst often has a flaccid consistency and tends to “flow out” and “fill out” the dependent parts of the abdominal cavity and interposes between the structures. Usually the diagnosis of mesenteric cyst is made after the exclusion of other diseases . With the development of US and CT, the preoperative diagnosis of abdominal cystic disorder has become the rule. US can distinguish between solid and cystic masses and CT can determine extension and cystic content , 8. On UMCs are rare and benign lesion. Although physical examination and abdominal US indicate remarkable ascites, giant MCs may be mistaken for ascites on physical examination and imaging studies . TherefoAdenosine deaminase (ADA) is an aminohydrolase that converts adenosine to inosine; so it is a purine-degrading enzyme and allows for preliminary diagnosis of peritoneal tuberculosis. Previous studies have demonstrated that ADA (cutoff 30–33 U/l) has a sensitivity, specificity, and diagnostic accuracy in peritoneal tuberculosis that were 100%, 97%, and 98%, respectively . A TurkiThe treatment of choice is complete excision to avoid recurrence and possible malignant transformation . HoweverIn conclusion, MCs are rare benign abdominal tumors. The pathogenesis is not uniform and the clinical and radiologic diagnosis is difficult. The symptoms of this condition vary from acute abdominal signs to nonspecific abdominal features or incidental findings. With an accurate history, a thorough clinical examination, a high index of suspicion, and the use of radiological examination, a definitive diagnosis can be made. The thin wall of the MCs can be better demonstrated with CT examination after intravenous contrast medium, and a correct radiological diagnosis of the lesion can be maintained.


Colorectal cancer (CRC) is a common and lethal disease with thousands of new cases of large bowel cancer diagnosed in the United States each year. Colorectal cancer frequently causes obstruction of the large bowel. Cases of obstructions and perforations have been documented with masses well below 6 cm in diameter. Obstruction and/or perforation are important predictors of prognosis with respect to colorectal carcinomas.The following is the presentation of an unusual case of a 13 × 12 × 16 cm colonic adenocarcinoma without any signs of obstruction or perforation.Perioperative mortality rates for colorectal cancer with obstruction have been documented anywhere from 5% to 47.6%, making obstruction an important predictive factor in long-term patient survival. Although a correlation between obstruction and patient outcome has been determined, a correlation between colorectal tumor size/staging and obstruction has yet to be investigated. Colorectal cancer (CRC) is a common and lethal disease with over 150,000 new cases of large bowel cancer diagnosed each year in the United States. Bowel oA 58 year old Caucasian male with a history of iron deficiency anemia and severe depression presented with weakness and shortness of breath, along with a loss of appetite and thirty pound weight loss. He also has a long history of psychological issues, including extensive illicit drug use, bipolar personality disorder, anxiety, chronic depression, and suicidal tendencies. He denied any constipation, diarrhea, tenesmus, melena, hematochezia, nausea, vomiting, or abdominal pain. He did, however, report feeling a firm mass in his right upper and lower abdominal quadrants, which he claims has grown in size over the years.Upon physical examination, the patient was found to appear thin, pale, but comfortable. He was found to have normal bowel sounds in all four quadrants; however, there was a large firm, non-tender mass palpated in the right upper and lower quadrants. Also, bilateral mildly reducible inguinal hernias and a large umbilical hernia were detected. Patient's exam was otherwise normal.The patient presented with a hemoglobin/hematocrit ratio of 7/25.7 and an albumin level of 2.9. He was therefore admitted for blood transfusions and further workup of a mass detected in his upper and lower right abdominal quadrants. Computed Tomography (CT) with contrast was performed which showed a large 13 × 12 × 16 cm right-sided ill-defined soft tissue mass involving the ascending colon , according to the American Joint Committee on Cancer staging system for colorectal cancer Table 9]..9].In this case, the tumor had invaded the retroperitoneal tissue (T4), zero out of sixteen nodes were found positive (N0), and no distant metastasis was discovered (M0). It is highly unusual that this Stage IIB  tumor was found without any signs of obstruction or perforation.Nevertheless, one cannot dismiss the fact that the patient's mental status could have "blunted" his true physical presentation. In other words, with chronically depressed individuals, such as in this case, it is difficult to determine whether the loss of appetite and weight loss is secondary to their mental illness(es) or cancer. Even with a thorough history and physical, the clinician can be misguided, especially if the patient is not presenting other symptoms that would make him/her more suspicious of other underlying conditions.The incidence of colorectal cancer makes it a very common disease process across the world. Perioperative mortality rates for colorectal cancer with obstruction have been documented anywhere from 5% to 47.6%, making obstruction an important predictive factor in long-term patient survival. Although a correlation between obstruction and patient outcome has been determined, a correlation between colorectal tumor size/staging and obstruction has yet to be investigated.Written and verbal informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The views expressed in the submitted abstract/manuscript are those of the author(s) and do not reflect the official policy or position of the Department of the Army, Department of Defense, or the U.S. Government. Also, the authors undersigned disclose that there are no financial, economic, commercial, and/or professional interests related to the topics presented in the manuscript. The authors declare that they have no competing interests.AE and KL were both involved in direct patient care, the surgery, perioperative management, literature review, and writing the manuscript. RG was a contributor in writing the manuscript. All authors helped read, edit, and approve the final manuscript.


Melanotic variant of oncocytic metaplasia of the nasopharynx is an extremely rare condition.A 73-year-old Japanese man presented with nasal congestion and chill. Nasoscopic examination revealed multiple black nodules around the bilateral torus tubarius. The nodules were biopsied to determine the histology. The clinical differential diagnosis was malignant melanoma or hemangioma. Microscopically, there were oncocytic plump cells with abundant brown pigmented granules showing glandular pattern. No significant atypia was found. The pigment was positive for Fontana-Masson staining, and negative for Berlin blue staining, showing that it was melanin pigment. Immunohistochemically, S100-positive HMB45-negative dendritic cells were also found.Such a pigmented variant of benign oncocytic lesion is very rare, and only 15 cases have been reported in the English literature. As a benign mimicker of malignant melanoma, melanocytic oncocytic metaplasia should be always taken into consideration in the clinical setting. In 1995, Shek et al. originally reported two cases of melanotic oncocytic metaplasia of the nasopharynx -5. We prA 73-year-old Japanese man presented with the feeling of nasal congestion (obstruction) and chill. His past medical history was unremarkable and he was a non-smoker. During nasoscopic examination multiple black nodules, measuring several millimeters, were discovered around the bilateral torus tubarius Fig . While tOncocytic cells are large epithelial cells with eosinophilic, granular cytoplasm. Oncocytic metaplasia (change) is most frequently encountered in certain epithelial organs, such as the salivary gland, the lacrimal gland, the parathyroid gland, the thyroid gland, the pulmonary tree, and the kidney . OncocytOncocytic change in the upper respiratory tract is an uncommon finding and melanotic variant of oncocytic metaplasia of the nasopharynx is an extremely rare condition. To our knowledge, 15 cases have been reported in the literature and our case is the 16th case.The lesion may be single or multiple. Most of the lesions are asymptomatic and tend to be incidentally discovered during examination of the nasopharynx. Some of these lesions may occasionally produce symptoms. When the mucosa around the opening of Eustachian tube is affected, ear or nose symptoms may occur. Eustachian tube dysfunction can be caused when the tissue edema accompanying the lesions impairs the tube function. In this case, the feeling of nasal congestion (obstruction) may be due to Eustachian tube dysfunction, and the chill may be coincidental.All reported cases including our case are elderly males in their sixties or seventies. The lesion may be misinterpreted clinically as early nasopharyngeal carcinoma, nevus, or malignant melanoma. The origin of the melanin pigment is in question. The exact nature of melanin-containing oncocytes in the nasopharynx awaits further clarification.In conclusion, we report the 16th case of melanin-containing oncocytic metaplasia of the nasopharynx. As a benign mimicker of malignant melanoma, melanocytic oncocytic metaplasia should be always taken into consideration in the clinical setting.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.TK and KM performed histological examination and analyzed the case. TK was a major contributor in writing the manuscript. SO and SM treated the patient and provided clinical images. All authors read and approved the final manuscript.


A 58-year-old-man presented with painful rapidly progressive bilateral proptosis with restricted ocular movements of 15 days duration. There was history of significant weight loss in the recent past. Computed tomography scan of the head and orbit revealed bilateral multiple, well-defined, round, soft tissue masses, isointense with muscles in intraconal and extraconal space. Fine needle aspiration cytology and incision biopsy from the lesion, urine for Bence-Jones proteins and immunofixation clinched the diagnosis of multiple myeloma. Skeletal survey did not reveal any bony involvement.The diagnosis of multiple myeloma should be kept in mind in cases of bilateral proptosis. Bony involvement is not universal in cases of orbital myeloma. Early diagnosis can be established with extensive biochemical and histopathological investigations and timely treatment is life saving for these patients. Multiple myeloma is a subgroup of plasma cell dyscrasias in which there is neoplastic proliferation of plasma cells or their precursors. Unilateral proptosis is a well-recognized but somewhat rare presentation of multiple myeloma in ophthalmic practice.2 BilaterA 58-year-old-man presented with painful rapidly progressive bilateral proptosis with restricted ocular movements of 15 days duration. There was history of about 10-kg weight loss amounting to 15% of his body weight over a period of the past one month. Ophthalmic examination revealed visual acuity of 20/30 in both eyes. His right eyeball was pushed inferiorly and laterally and left eye showed axial proptosis . Hertel'The differential diagnosis of secondaries from an unknown primary, blood cell-related tumor including lymphoma or plasma cell dyscrasias was kept. Hemogram and coagulation profile showed hemoglobin of 7.8 g/dl  though his blood counts and coagulation profile was normal. To look for an unknown primary from the lung, prostate, gastrointestinal tract; endoscopy, colonoscopy, stool examination and prostate examination was done. Examination of the gastrointestinal tract (GIT) and prostate was unremarkable. CT abdomen revealed paravertebral soft tissue mass and contrast enhanced CT (CECT) chest revealed a soft tissue mass in the parahilar region.Fine needle aspiration cytology (FNAC) followed by incision biopsy of the orbital mass and FNAC from the left thigh nodule showed cellular sheets of mature and immature plasma cells, many binucleate and multinucleate forms were seen, suggestive of plasmacytoma . KeepingPatient was started on chemotherapy in consultation with the oncologist . Patient responded dramatically to treatment with remission of eye manifestations  and skinPlasma cell dyscrasias are a rare cause of proptosis. Solitary extramedullary plasmacytoma (SEMP) tends to be locally invasive and does not often metastasize. Multiple myeloma is a malignant systemic plasma cell neoplasm that is aggressive and metastasizes. It is a malignancy of plasma cells characterized by monoclonal proliferation of a single clone of highly specialized B-lymphocytes engaged in the production of a single immunoglobulin. It is associated with production of paraproteins and/or light chain moiety. The criteria for diagnosis of SEMP include negative lymph node assessment, skeletal survey, bone marrow biopsy and CT. In the present case the patient though presented with proptosis and no skeletal involvement, had positive bone marrow biopsy, renal insufficiency, anemia and multiple plasmacytomas. Orbital involvement is a rare presentation of the disease.24 Unilat23Orbital myeloma most commonly presents as a unilateral solitary soft tissue intraorbital tumor which is an extension of bony deposit and is associated with bone destruction. In the p310The diagnosis of multiple myeloma should be kept in mind in cases of bilateral proptosis. Bony involvement is not universal in cases of orbital myeloma. Early diagnosis can be established with extensive biochemical and histopathological investigations and timely treatment is lifesaving for these patients.


Extraskeletal osteosarcoma is an uncommon high-grade malignant soft tissue sarcoma. Well-differentiated extraskeletal osteosarcoma is thought to have a better prognosis than classical extraskeletal osteosarcoma, but dedifferentiation after recurrence has also been reported. We present a case of a primary retroperitoneal extraskeletal osteosarcoma in a 62-year-old Japanese woman. Abdominal CT revealed a large mass with diffuse calcification in the right retroperitoneal space and tumor resection was performed. The histopathological diagnosis was well-differentiated retroperitoneal extraskeletal osteosarcoma. She was followed up by CT every 6 months without adjuvant radiotherapy and chemotherapy for 31 months until anaplastic high-grade spindle cell sarcoma recurred in the retroperitoneum. Our case is the seventh reported description of well-differentiated extraskeletal sarcoma, and the first to arise in the retroperitoneum and recur as an entirely dedifferentiated spindle cell sarcoma. Well-differentiated extraskeletal osteosarcoma is a rare soft tissue sarcoma, with only 6 cases reported in the English literature –4. The bA 62-year-old woman was admitted to our hospital complaining of severe right abdominal pain for 4 months. A fixed stony-hard tumor of about 100 mm in diameter was found on palpation, with slight tenderness on the right side of her abdomen. A full blood examination gave values within the normal limit. Abdominal CT showed a large mass with diffuse calcification of size about 130 × 110 mm in the right retroperitoneal space between the right kidney and the inferior vena cava, with slight hydronephrosis . Lymph nComplete surgical resection of the primary tumor was performed. The tumor was situated behind the mesenterium of the ascending colon, compressing the right kidney, but no direct invasion of the surrounding organs was observed: there was no lymph node swelling, ascites or peritoneal dissemination, and the tumor was not present in the parenchyma of the liver and the kidney. The surgical margin was determined based on the rough connective tissue surrounding the tumor. Dissection of lymph nodes was not performed.Gross examination revealed a rounded, bony, and hard tumor of size about 140 × 110 × 80 mm and weight 1100 g that was yellowish-white in color . HistoloThe postoperative course was uneventful. We recommended adjuvant therapy because of the probable poor prognosis, but the patient refused radiotherapy and chemotherapy. The patient was followed up by CT every 6 months without receiving adjuvant radiation therapy or chemotherapy.Thirty-one months after the operation, she was readmitted to our hospital due to severe pain in the lumbar region and the right lower leg. Abdominal CT revealed a large mass in the right retroperitoneal space. No calcification was seen, but recurrence of the primary tumor was strongly suspected. Surgical resection of the recurrent tumor was performed via a transabdominal approach, in which a tumor with adherence to the right kidney was completely removed, a procedure of tumor resection and right nephrectomy. Grossly, the recurrent retroperitoneal tumor adherent to the right kidney was of size 310 × 300 mm, with necrosis and hemorrhage and a yellowish-white color on the cut surface. The recurrent tumor was characterized histologically by proliferation of atypical nonepithelial cells with hyperchromatic nuclei and a high nuclear-cytoplasmic ratio. The mitotic count of the neoplastic cells was about 50 per 10 high-power fields. Anaplastic features predominated, but a few foci of matrix production looked like osteoid or hyalinized collagen within the recurrent neoplastic tissues. These findings led to a diagnosis of high-grade malignant anaplastic spindle cell sarcoma, consistent with the recurrence of extraskeletal osteosarcoma . There wExtraskeletal osteosarcoma is defined as a malignant osseous neoplasm arising from soft tissue without attachment to skeletal bones. This neoplasm is rare compared with bone osteosarcoma, accounting for 1-2% of all soft tissue sarcomas , and hasExtraskeletal osteosarcoma is generally a highly aggressive neoplasm with a grave prognosis, and its histology is basically high-grade, similar to most skeletal osteosarcomas. Most patients die from metastatic disease within 2 to 3 years of the initial diagnosis , 8, 9, wIt is not always easy to distinguish extraskeletal osteosarcoma from other benign or malignant bone- and cartilage-forming soft tissue lesions . DiffereTreatment for extraskeletal osteosarcoma is usually restricted to local intervention, including surgery alone or a combination of surgery and radiation therapy. Systemic therapy including adjuvant chemotherapy has not been established. Patel et al.  summarizWell-differentiated (low-grade) extraskeletal osteosarcoma is extremely rare. This tumor was first described in 1953 by Umiker  and appeWell-differentiated extraskeletal sarcoma may have a better prognosis than classical extraskeletal sarcoma, but some cases may recur as a high-grade malignancy with rapid progression leading to death. In our case, regular Follow-up was performed only with a plain abdominal CT scan at 6-month intervals. Based on our experience, we recommend closer follow-up using a chest-abdominal CT scan, abdominal magnetic resonance imaging (MRI), or ultrasound enhanced with a contrast agent.Our case suggests that careful observation for the recurrence of the tumor is necessary if the pathological diagnosis is well-differentiated extraskeletal osteosarcoma.


Intestinal tuberculosis is a common problem in endemic areas, causing considerable morbidity and mortality. An isolated primary caecal perforation of tubercular origin is exceptionally uncommon.We report the case of a 39 year old male who presented with features of perforation peritonitis, which on laparotomy revealed a caecal perforation with a dusky appendix. A standard right hemicolectomy with ileostomy and peritoneal toileting was done. Histopathology revealed multiple transmural caseating granulomas with Langerhans-type giant cells and acid-fast bacilli, consistent with tuberculosis, present only in the caecum.We report this extremely rare presentation of primary caecal tuberculosis to sensitize the medical fraternity to its rare occurrence, which will be of paramount importance owing to the increasing incidence of tuberculosis all over the world, especially among the developing countries. Intestinal tuberculosis is an extremely common occurrence in India. Any portion of the gastrointestinal tract may be affected by it, and various pathogenic mechanisms are involved. However, a primary isolated caecal perforation due to tuberculosis is an unusual occurrence, without the concomitant involvement of the ileum or ascending colon. We report this extremely rare presentation, the diagnosis of which was made retrospectively, with a view to sensitize the entire world to this entity.A 39 year old male presented to the emergency department, in the month of May 2009, with complaints of right lower quadrant abdominal pain and low grade fever of 2 days duration. He was a chronic alcoholic since 10 years and had no other significant prior medical, surgical history, or any evidence of a traumatic event. On examination, the patient had tachycardia, and slight tenderness and guarding, localized to the right iliac fossa. The rest of the abdomen was non tender. His routine blood tests were within normal limits, with completely normal counts. His HIV and HBsAg tests were negative. An x ray of his abdomen (erect film) showed a ground glass appearance. Ultrasound examination of his abdomen however revealed features suggestive of terminal ileitis or ruptured appendix  with a normal liver. Clinical suspicion was of a recently ruptured appendix. The patient was taken up for an emergency laparotomy, via a right paramedian incision (considering his USG findings). Intra operatively, a 3 × 2 cm caecal perforation was present along the antero lateral wall with a dusky appendix attached to the caecum via a narrow base. There was no peritoneal contamination and the rest of the bowel appeared completely normal. The liver and spleen were normal and there was no lymphadenopathy. A right hemi colectomy with ileostomy and transverse colonic mucous fistula was done and the specimen was sent for a histopathological diagnosis. The patient was started on anti-tubercular therapy , with satisfactory results as regards the improvement in the overall patient condition, with 6 kgs weight gain over two months. The patient became comfortable, ambulatory; Ileostomy was functional and viable and main wound healing occurred via secondary intention. The patient was followed up on a regular 2 weekly basis with ileostomy closure performed after 3 months, without incident.Isolated caecal perforation, that too tubercular in origin, is a rare entity. Caecal perforations are commonly encountered as a part and parcel of various associated disease processes, in accordance with Laplace's law. Laplace's law dictates that the intraluminal pressure needed to stretch the wall of a hollow tube is inversely proportional to its radius. The cecum has the largest diameter of the colon, and as such, requires the least amount of pressure to distend -3. The dCaecal perforations are usually seen associated with entities such as diverticular disease, inflammatory bowel diseases, ogilville syndrome, closed In dealing with an emergency colonic obstruction leading to a caecal perforation, few authors favour some form of primary anastomosis as opposed to others favoring creating of an initial stoma after a standard right hemi-colectomy. Many others have recommended subtotal or extended right hemicolectomy for emergency left colon or distal transverse colon obstruction. This is advisable in cecal perforations complicating large bowel obstruction, and in those patients who have a high risk of a metachronous lesion developing. This isIleocecal tuberculosis has always been elusive to physicians, patients often ending in the surgical ward with acute or subacute obstruction, intractable vomiting and diarrhea and often chronic abdominal masses. The precise incidence of gastrointestinal tuberculosis is not known, due to a lack of random samples of populations studied. However, the reported incidence varies from 0.02% to 5.1% in various autopsy series. Furtherwhat the mind knows is what the eyes see'.A spontaneous primary caecal perforation can be tubercular in origin even in the absence of features suggestive of the same, and thus this unusual entity must be borne in mind for 'The patient has granted his written and informed consent for publication of this work.The authors declare that they have no competing interests.PSL, SM, GA, NJ were the operating team of surgeons and DKJ was the overall team incharge, under whose guidance the entire surgery was performed and who guided the subsequent management of the patient. All authors have read and approved the manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2482/10/12/prepub


The concept of compression of the rostral ventrolateral medulla as a cause for hypertension is gaining more and more interest. This report is about a 36-year-old male with a three years history of hypertension who presented with a posterior fossa mass suggestive of a hemangioblastoma. Laboratory and imaging studies ruled out the presence of von Hippel-Lindau disease and/or concomitant pheochromocytoma. Post-surgical blood pressure monitoring revealed a 40 mmHg decline in blood pressure. It could be hypothesized that alleviation the compressive effect of the tumour on the rostral ventrolateral medulla as proposed by previous studies could be a contributing factor. Hemangioblastomas  constitute around 2-3% of all primary nervous system tumors and although they can occur at all sites from the cerebral cortex to peripheral nerves, are most commonly found in the cerebellar hemispheres ,2. PreseA 36-year-old Iranian male with Caucasian ethnicity referred to the emergency department of our institute with a chief complaint of severe suboccipital headache, vomiting and frequent drop attacks. He had arterial hypertension for 3 years and was taking atenolol 50 mg/day. Family history was negative for secondary causes of arterial hypertension including adrenal disease. Physical examination revealed blood pressure of 180/100 mmHg, bilateral papilledema and truncal ataxia with gaze-dependent horizontal nystagmus. Lab tests showed polycythemia (Hemoglobin = 18 g/dL). Contrast enhanced brain MRI showed a round extra-axial heterogeneous densely enhanced 4 × 4 cm mass with irregular margins. It was located in the left cerebellar hemisphere, posterolaterally obliterating the fourth ventricle, causing non-communicating hydrocephalus. The mass encroached onto the cavity of the fourth ventricle anterolaterally, abutting the right half of the brainstem Figure . AbdominA midline posterior fossa craniectomy was performed. Following durotomy, the mass appeared as a reddish lobulated tumor with multiple feeders from cerebellar cortex and the bridging PICA which were coagulated and divided. Not surpassing the surrounding gliotic border, the tumor was totally resected. Post-operative period was uneventful and the obtained MRI revealed no residual tumor Figure . SurprisSecondary hypertension may be caused by a myriad of secretory disorders, including some tumors such as pheochromocytoma and aldostrone-secreting masses. However, not all these hypertensive states could be attributed to humoral factors. In 1979, Janneta and Gendell introduced the concept of neurogenic hypertension caused by pulsatile vascular compression of the left lateral medulla . Animal Mitigation of arterial hypertension after resection of the tumor in our patient, as well as negative laboratory and imaging studies for secondary causes for HTN supports the proposed mechanism of RVLM compression, as the pathogenesis of HTN.There had been reports of posterior fossa tumors associated with arterial hypertension. The plausible mechanism of rostral ventrolateral medullary compression is gaining interest as a cause for hypertension. The present case is another clue advocating the proposed mechanism.VHL: von Hippel-Lindau disease; MRI: Magnetic resonance imaging; CT: Computed Tomography; PICA: Posterior inferior cerebellar artery; VMA: Vanillylmandelic acid; EMA: Epithelial membrane antigen; NSE: Neuron Specific Enolase; RVLM: Rostral ventrolateral medulla; HTN: Hypertension.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.The authors contributed to surgery and preparation of the manuscript. The primary draft and its final revision was done by ATM. HS was the attendant surgeon and prepared the primary draft and revised the manuscript. MZ aided in surgery and revised the manuscript.


A 51-year-old woman with severe asthma underwent bronchoscopy and endobronchial ultrasound (EBUS) for investigation of a 15-mm peripheral lung nodule. Histology demonstrated a typical carcinoid tumor. Pulmonary location is the second commonest site for carcinoid tumors. Diagnosis of peripheral carcinoid tumor of the lung is difficult due to its small size, poor accuracy of cytologic diagnosis, and low sensitivity of positron emission tomography in detecting it. EBUS has a high diagnostic yield and a low complication rate in the evaluation of small solitary pulmonary nodules. The ultrasound appearance of carcinoid tumors is identical to that of lung carcinomas. Prompt diagnosis of carcinoid tumor is desirable as regional lymph node metastasis is seen in 10% of patients and is associated with a reduced 5-year survival. We feel that, where possible, all patients presenting with solitary pulmonary nodules should be investigated initially using EBUS due to its high diagnostic rate and the very low incidence of adverse events. CT scanning of the chest was performed to exclude any factors that may have been responsible for exacerbating her illness. A lobulated, well-defined, soft tissue density nodule, measuring 15 mm was identified in the left lower lobe [A 51-year-old lady presented with persistently poor control of her asthma symptoms. She had a long-standing history of difficult asthma, frequently requiring oral prednisolone therapy. Respiratory function testing demonstrated moderate fixed airflow obstruction with a forced expiratory volume in 1 second . Examination in the posterobasal segment of the left lower lobe demonstrated a solid lesion . FluorosHistologic examination of biopsy specimens showed soft tissue infiltrated by sheets and cords of cells with slightly irregular nuclei, intranuclear inclusions, and fine granular chromatin. ImmunohCarcinoid tumors are low-grade malignancies comprising neuroendocrine cells. The commonest site for such lesions is the gastrointestinal tract, with a pulmonary location being the second commonest site. They are2The majority of pulmonary carcinoids are centrally located; when loEBUS has been demonstrated by numerous groups to improve the diagnostic evaluation of solitary pulmonary nodules (SPNs). Its value is most apparent with smaller lesions,8 where tEven large series examining the efficacy of EBUS report only very rare diagnosis of carcinoid tumour. Only one carcinoid was diagnosed in a cumulative 577 EBUS procedures.–13 Carci14Without the histological diagnosis that was made possible by EBUS our patient may have gone undiagnosed for some more time. Firstly, over one-third of carcinoid tumors are diagnosed only at thoracotomy, and this17While 5-year survival from typical carcinoid tumors is as much as 91%, delay inThis case illustrates the significant value of bronchoscopy using EBUS in the investigation of small peripheral pulmonary nodules. Peripheral pulmonary carcinoid tumors may reliably be diagnosed using transbronchial biopsy, and their ultrasonographic appearance seems to be similar to other bronchogenic carcinomas. We feel that, where possible, all patients presenting with solitary pulmonary nodules should be investigated initially using EBUS due to its high diagnostic rate and very low incidence of adverse events.


Sir,Elephantiasis nostras verrucosa (ENV) is a rare, chronic, deforming disorder characterized by hyperkeratosis and papillomatosis of the epidermis with underlying woody fibrosis of the dermis and subcutaneous tissue. No standard treatment for this rare cutaneous manifestation is available. In this A 51-year old male was presented to our clinic with a 15-year history of a pendulum-like mass arising from the anterior abdominal wall in its lower part covering penis and scrotal region . The devPhysical examination revealed generalized thickening and lichenification of the skin as well as multiple fistulas. No lymph nodes were palpable due to the woody nature of the tissue. Rectal examination revealed a slightly enlarged prostate without any palpable masses. Test results were negative for fecal occult blood. The patient underwent surgery to remove the mass. A compression stocking was used to treat the lymphedema. Ten months after the operation, we saw no signs of disease recurrence. The result was also aesthetically satisfactory .Biopsy specimens revealed pseudoepitheliomatous hyperplasia with dilated lymphatic spaces in the dermis, accompanied by chronic inflammation and fibroblast proliferation. No evidence of neoplastic change was observed.Elephantiasis is a clinical description of distinct cutaneous changes associated with a chronic underlying dysfunction in the lymphatic system. Many factors may lead to chronic lymphatic obstruction and stasis. The comm11ENV and papillomatosis cutis carcinoides share many features. The latter can morphologically resemble ENV with single or multiple verrucous, partially ulcerated tumors on the chronically irritated edematous extremity. Both are histologically characterized by pseudoepitheliomatous hyperplasia. The diagnosis of papillomatosis cutis carcinoides is established by biopsy.Therapeutic efforts should aim to reduce lymph stasis, which will also improve the cutaneous changes. The mainstay of therapy remains prophylactic antibiotics, compression, and elevation of the affected limb. SystemicENV should be included in the differential diagnosis of chronic lymphedema and be differentiated from papillomatosis cutis carcinoides. Early diagnosis is paramount. Without appropriate early intervention, ENV continues to worsen and may result in gross deformity requiring amputation.3


Epididymal inflammatory myofibroblastic tumor, also known by various other synonyms is a rare benign disease. Only eight cases have been reported to date. The most common presentation is a scrotal mass of variable duration. For a scrotal mass it is difficult to distinguish a benign or malignant etiology, in addition to the origin whether from testis or epididymis. As a result the definitive diagnosis can only be established by surgical exploration.We report the ninth case of epididymal IMT who based on clinical and radiological findings underwent radical orchidectomy, with the histology suggestive of inflammatory myofibroblastic tumor. At 4 years follow up the patient is free of disease recurrence.IMT though rare should be considered in the differential diagnosis of epididymal mass. Clinically it is often difficult to distinguish the origin of mass and even though the disease has benign nature and course it is crucial to counsel patients for orchidectomy as definitive diagnosis is established on surgical exploration. Inflammatory Myofibroblastic tumor (IMT) of epididymis is a distinct but rare entity. IMT is also described by other synonyms, more commonly as inflammatory pseudotumor. Extra genitourinary and genitourinary sites are well documented with various proposed etiological theories . EpididyA 22 year old healthy Caucasian male noticed a swelling and a palpable mass in the right scrotum for a period of one week. Patient denied any history of fever, trauma, urethral discharge and any previous history of recurrent urinary tract or sexually transmitted infections. There was no past history of exposure to tuberculosis. Physical examination revealed a nontender indurated solid mass in the lower pole of right testicle possibly also involving the epididymis.Scrotal ultrasound demonstrated a solid heterogeneous mass involving right testicle with possible extratesticular extension into the epididymis. Quantitative serum Beta -human chorionic gonadotropin, alpha-fetoprotein and LDH (lactate dehydrogenase) were within normal limits.With the presumed diagnosis of testicular tumor a right radical orchidectomy was performed. On gross pathologic examination the mass was abutting the tunica albugenia but further examination revealed being confined to epididymis with normal testicular parenchyma. Histology of the mass (4 × 2.2 × 1.8 cm) demonstrated a spindle myoepithelial and polygonal cell proliferation with intense lymphoplasmacytic infiltrate. Fig.  and CD 1The patient recovered well with no evidence of any recurrence at the site of resection or other sites after 4 years of follow-up.Based on such an unusual and rare finding a thorough Medline search revealed eight additional patients with similar presentation of scrotal lump. All patients had exploration of the scrotal mass due to its solid heterogenous features on ultrasound and clinical examination. All patients underwent either excision of mass or radical orchidectomy.Inflammatory Myofibroblastic tumor (IMT) is a well described disease and can occur in many organs such as lung, skin, soft tissues, breast, gastrointestinal tract, pancreas, oral cavity, bone and central nervous system. However various sites in genitourinary tract have also been reported but less commonly , epididyOnly those tumors with spindle myoepithelial cell proliferation and lymphocytic infiltrate qualify for IMT. Various synonyms like inflammatory pseudo tumor, plasma cell granuloma, plasma cell pseudo tumor, atypical Myofibroblastic tumor and post operative spindle nodule are used interchangeably . In spitThe pathophysiology of IMT is not well understood, various etiologies have been proposed including a reparative process related to delayed chronic response to remote or undetected trauma . InfectiPatients with IMT can present with fever, night sweats, weight loss, malaise or abnormal laboratory parameters such as elevated ESR (erythrocyte sedimentation rate), anemia, leukocytosis and site specific symptoms . HoweverThe diagnosis of IMT is based on the histological features of spindle myoepithelial cell proliferation, lymphocytic and inflammatory infiltration. Other immunomarkers could substantiate the diagnosis of IMT. Immunomarkers such as vimentin, actin and CD 68 are positive in 25% cases . A similVarious non surgical treatment options have been proposed at sites other than genitourinary tract including cyclosporine, corticosteroid, methotrexate, antibiotics -18 and rThe abdominal and retroperitoneal variant presents with more aggressive pattern compared to their extra abdominal counterparts, with recurrence rate of 23–37% ,20. The IMT though rare should be considered in the differential diagnosis of epididymal mass. Clinically it is often difficult to distinguish the origin of mass either from testis or epididymis. Radiological studies are unable to differentiate benign or malignant nature and as a result definitive diagnosis is established on surgical exploration. Depending on the gross characteristics and frozen section of clinically distinct masses, either a local excision or radical orchidectomy is offered. Thus even though the disease has benign nature and course it is crucial to counsel patients for orchidectomy as definitive diagnosis is established on surgical exploration.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.PPD was involved in conception and design, acquisition of data, data analysis, and interpretation, manuscript drafting and final approval. WPW was involved in acquisition of data, data analysis, provided pathologic imaging, interpretation of data and final approval. KSP was involved in conception and design, acquisition of data, data analysis, and interpretation, manuscript drafting and final approval.


Intravascular papillary endothelial hyperplasia is a benign nonneoplastic vascular lesion that consists of endothelial cells with abundant vascular tissue with papillary proliferation. An adult female had a painless growing dark red nodule on the left side of the lower lip and often touched and gnawed at it for more than 4 years. The lesion was a tender, smooth mass approximately 1 cm in diameter without discoloration reaction. Magnetic resonance imaging of the lesion showed specific findings. She was diagnosed clinically as having mimicked hemangioma, and the lesion was totally excised under local anesthesia. Histopathological examination revealed that papillary proliferated endothelial cells with venous pool, and the lesion was diagnosed as intravascular papillary endothelial hyperplasia associated with venous pool. There has been no recurrence for more than 1 year. Despite the benign nature of this lesion, it could have been mistaken for a malignant tumor because of its clinical course and radiologic findings. Hemangioendothelioma vegetant intravasculaire in 1923. IPEH was first named by Clearkin and Enzinger [Intravascular papillary endothelial hyperplasia (IPEH) is a benign nonneoplastic and vascular lesion. The lesion was first described by Masson  as a HemEnzinger , and it Enzinger , malignaEnzinger . The lesEnzinger , Masson'Enzinger , or MassEnzinger  and compEnzinger . This leEnzinger , and asyIn this report, an adult female was diagnosed as having mimicked hemangioma arising in the lower lip by clinical finding and magnetic resonance imaging, and the lesion was excised completely. The lesion was examined by histopathological and immunohistochemical methods.A 62-year-old female was referred for a painless, asymptomatic, nonpulsatile slow growing dark red nodule in the lower lip. For more than 4 years the patient was aware of the lesion and she had frequently touched and gnawed around it. There was no contribution of her general medical history, and the head and neck examination revealed no evidence of adenopathy, paresthesia, or motor nerve deficiency. Extraoral findings revealed that the appearance of her skin was normal and symmetrical. Clinical intraoral examination of the left side of the lower lip showed a tender, smooth mass measuring approximately 1 cm in diameter without discoloration reaction . The bouThe lesion mimicked a hemangioma and was examined by magnetic resonance imaging (MR image) and it was not examined by sonography. In an axial T1 weighed MR image, the marginal edge of the lesion showed an enhanced line with the center of the lesion showing zero signal intensity . In an aThe lesion was totally excised under local anesthesia. Surgical procedure revealed that the mass was denuded carefully and removed after the incision around the mass. And the finding revealed that the mass was not clearly continued to the surrounding tissue, there was extremely little bleeding around the mass. The continuity of the mass to the surrounding tissue was not clear. The mass was excised with ease, and minimal blood was lost during the surgery. No recurrence was observed more than 1 year postoperatively.  According to the histological examination, a thrombus was observed in an expanded blood vessel. In addition, it was observed that the endothelial cells showed papillary proliferation Figures . ExpandeIntravascular papillary endothelial hyperplasia is a benign, non-neoplastic, and vascular lesion . IPEH isHowever, to differentiate IPEH from malignant tumors, such as angiosarcoma , malignaAccording to the examination of MR images of hemangioma arising from soft tissues, Teo et al. described that they reviewed MR imaging studies of 22 patients with soft-tissue hemangiomas and 22 patients with malignant soft-tissue masses . They reIn the immunohistochemical examination of IPEH, Albrecht et al. described that IPEH is closely related to organizing thrombi using antibodies to Factor VIII-related antigen, ferritin, and vimentin, Bodner and Dayan , 4. In aIn conclusion, we diagnosed this lesion as IPEH with venous pool because the histopathological and immunohistochemical findings revealed vigorous proliferation and papillary structures of endothelial cells associated with thrombi. In this study, we examined this lesion by MR imaging and the diagnosis was based on histological and immunohistochemical findings.


Aggressive digital papillary adenocarcinoma is a rare variant of eccrine sweat gland malignancy with a propensity for metastases and recurrence. We report a 45-year-old female with aggressive digital papillary adenocarcinoma diagnosed by fine needle aspiration cytology (FNAC). The cytological findings were representative of the histological features. The recognition of aggressive digital papillary adenocarcinoma as a distinct clinicopathological eccrine sweat gland neoplasm is important because of the potential for aggressive local growth and distant metastasis. FNAC plays an important role in the preoperative diagnosis and management of these lesions. Aggressive digital papillary adenocarcinomas are rare eccrine sweat gland tumors which have the potential for highly aggressive biologic behavior. We report the cytological finding of a case of aggressive digital papillary adenocarcinoma. To the best of our knowledge, the cytology findings have not been reported in the English literature.A 45-year-old female presented with a recurrent swelling over the lateral aspect on the dorsum of the right hand, of 5 months duration, associated with pain for the past 5 days. The local examination of the swelling revealed a 3×2 cm, tense, cystic, nontender swelling with restricted motility in the subcutaneous plane.The general physical and systemic examinations were normal. No abnormality in the breast, thyroid, lungs, and abdomen was detected clinically. The routine laboratory investigations and chest radiographs and ultrasound scan of the abdomen were normal.Fine needle aspiration cytology (FNAC) of the swelling was done using a 24-gauge needle and 10-ml syringe and about 2 ml of blood mixed fluid was aspirated. Examination of hematoxylin and eosin (H and E) stained smears revealed highly cellular aspirates consisting of many tissue fragments of pleomorphic epithelial cells which were also scattered singly throughout the smears . The tisAn excision biopsy was done and subjected to histopathological examination. Grossly, the lesion was well circumscribed, measuring 2×1 cm. The cut section revealed solid and cystic areas. Microscopically, the lesion was characterized by tubulo-alveolar and ductular structures occupying cystic lumina. The stroma varied from thin fibrous septae to areas of dense hyalinized collagen. Fibrovascular core circumscribed the epithelial islands . ModeratAggressive digital papillary adenocarcinoma is a rare tumor arising from skin sweat glands. It occurs as a single painless mass, almost exclusively on fingers, toes and adjacent skin of palms and soles.–3 It occ3et al.,[These tumors have a high rate of local recurrence.5 In a stet al.[et al.[1These tumors can metastasizse, occasionally resulting in mortality. It was found to be 4% in a study by Jih et al. and 41.2.[et al.1 No evideThese tumors may simulate metastatic papillary carcinoma from breast, lung, thyroid, ovary and lesion from these areas need to be ruled out by clinical and radiological examination. The other malignant skin adnexal lesions can be differentiated by the site of occurrence and non-papillary nature of these lesions.Aggressive digital papillary adenocarcinomas are rare, distinct, eccrine sweat gland malignant tumors characterized by aggressive biologic behavior with a high rate of local recurrence and distant metastasis, occasionally resulting in mortality. FNAC aids in establishing an early diagnosis and management of these cases.


A 68-year-old woman with progressive paraparesis and altered sensation lasting approximately five days was admitted to our clinic. Magnetic resonance imaging (MRI) revealed an advanced stage T7-8 epidural mass ventral to the spinal cord, which was believed to be a metastatic tumor considering the patient's age. A highly enhanced epidural mass and pedicle appeared during the MR scan. However, the pathologic findings were compatible with the diagnosis of a primary meningeal melanocytic tumor. Primary epidural melanomas are extremely rare lesions. This case was finally diagnosed as a primary thoracic spinal epidural melanoma. Most malignant melanomas in the central nervous system (CNS) are metastatic lesions . Most spA 68-year-old woman with progressive paraparesis and altered sensation lasting approximately five days was admitted to our clinic. Neurologically, the patient showed paraparesis (IV/III), hypesthesia and hypoalgesia below the T9 dermatome, as well as preserved proprioception. The knee-jerk was hyper-reflexive and ankle clonus was identified bilaterally. T1-weighted magnetic resonance imaging (MRI) demonstrated an advanced T7-8 epidural mass ventral to the spinal cord. The T2-weighted images revealed low signal intensity for the lesion .The mass was believed to be a metastatic tumor based on the patient's age and MRI findings . Chest and abdominal CT, gastroscopy, colonoscopy, bone scan, tumor marker levels, and mammography were used to find the primary tumor focus but were unsuccessful. She underwent surgery via the posterior, transpedicular approach. After the laminectomy, a firm, black, epidural neoplasm was encountered, which had invaded the left pedicle, encasing and distorting the spinal cord . The masMelanoma is a malignant neoplasm arising from melanocytes. Melanocytes are melanin-producing cells that arise from the neural crest during embryogenesis and migrate to the skin, mucous membranes, and CNS . In the Meningiomas, schwannomas, and angiomatous lesions, such as cavernous hemangiomas or hemangiopericytomas, are rarely purely extradural and may show intratumoral hemorrhaging ,7. MeninFor primary CNS melanocytic neoplasms, a complete tumor resection is preferred because it can cure well-differentiated and intermediate-grade melanocytomas (IMGs) and most melanomas ,11. RadiPrimary CNS melanomas are relatively uncommon lesions compared to metastatic melanomas in the CNS. Once metastases, including drop metastases from pigmented medulloblastomas, have been excluded, the differential diagnosis includes pigmented meningiomas and schwannomas (solitary or as part of the Carney complex), as well as other pigmented CNS tumors, such as ependymoma and pineoblastoma and systemic diseases, such as lymphomas -12.


The splenic artery is the most frequent site of visceral arterial aneurysms. Usually a splenic artery aneurysm occurs as a single event; rupture is frequent, sometimes occurring as the first symptom and is sometimes fatal. This article presents a case of ruptured multiple splenic artery aneurysms—the symptoms and signs, operative and perioperative management, as well as a literature review of this clinically important entity. Among visceral aneurysms, the splenic artery aneurysm (SAA) is the most common, representing 60% of such lesions. There ar35A 42-year-old mother of four and 5/12 postpartum, presented to the King Fahad Medical City emergency room with a history of sudden onset of left-sided abdominal pain lightheadedness and repeated vomiting and hematemesis during the last two episodes. She was not on any medication or contraceptives. Her initial vital signs were stable, but a few minutes later, she became hypotensive (BP = 65/50 mm Hg), tachycardiac (HR = 110 bpm), and acidotic (pH = 7.1).She was resuscitated and stabilized. The results of her initial investigations revealed normal plain X-rays. The complete blood count showed leucocytosis of 22,000 cells/dL3 and Hb = 6.8 g/dL. A spiral computed tomography (CT) of the chest and the abdomen revealed a 1.5-cm SAA about 1 cm from the origin of the splenic artery, a second 1 cm aneurysm in the middle of the artery, and a third larger aneurysm that measured about 4.5 cm at the hilum of the spleen . No intrA midline laparotomy was performed and supraceliac aortic access was obtained. Proximal control was achieved by double ligation and by the division of the splenic artery close to its origin. A splenectomy was then performed, after which the patient had an unremarkable recovery and was discharged in a stable condition.Pathological examination showed two intact proximal aneurysms and a ruptured distal aneurysm, the widest measuring 3.5 × 2.1 × 4.5 cm, with an intraluminal thrombus. Microscopic examination (chromatin stain) revealed a very thin wall of the aneurysms and a lack of elastin with a large infracted area. There was no evidence of vasculitis or neoplasm.Although the rupture of a splenic artery aneurysm is a rare cause of acute abdominal pain, it is a very important item in its list of the differential diagnosis, especially when it is associated with hypovolemic shock. This topic was identified by literature searches in Medline, Embase, and the Cochrane Library. The search string  for rupture splenic artery aneurysm consisted of a combination of acute pain services, abdominal pregnancies, and splenic artery. The searches were limited to English language journals.The splenic artery is the most frequent site of visceral arterial aneurysms.1112 The 111411713The cause of an SAA is unknown. However, local failure of the connective tissue of the arterial wall to maintain the integrity of the vessel could be playing a major role in the development of this phenomenon. This leads to fragmentation of elastic fibers and a loss of smooth muscle. It is be19131013Arteriography is the gold standard for diagnosis in suspected unruptured aneurysms.23 Howeve102Treatment of splenic artery aneurysms is recommended in patients with symptomatic or expanding aneurysms, patients with aneurysms > 2 cm, women of childbearing age, and liver transplantation candidates.141521 Se141531In patients with hilar aneurysms such as the one presented here, coil/Gelfoam embolization is a viable option. If selective catheterization of the aneurysm cannot be performed, embolization of the entire splenic artery is an alternative option. Patients should be alerted to the potential for severe pain from splenic infarction. Our patient underwent embolization of the distal ruptured aneurysm to stabilize her hemodynamics. Unfortunately, she continued to be hypotensive despite aggressive resuscitation. She underwent an open excision of the splenic artery with splenectomy.Endovascular treatment is an emerging therapy for SAA and other visceral aneurysms with constantly improving results. These minimally invasive endoluminal techniques may offer a distinct advantage to conventional repair. However, because of the relative paucity of visceral artery aneurysms, long-term results are yet to be reported. It offers the potential benefit of maintaining splenic perfusion while excluding the aneurysm, thereby eliminating the risk of rupture or infarction.34 The naA diagnosis of ruptured SAA should be considered in any woman who presents with severe left upper abdominal pain or hypovolemic shock. Successful treatment of SAA requires a high index of suspicion, early recognition, and prompt management. Treatment usually is challenging. However, safe surgical treatment can be applied by a carefully planned approach based on adequate exposure and proximal and distal arterial control.


Adrenal oncocytoma is a rare entity, with 20 cases reported in literature. A functional oncocytoma is extremely rare. We present a case of adrenal oncocytoma in a hypertensive male who had elevated catecholamine levels, which improved after adrenalectomy with decrease in daily antihypertensive requirement. Adrenal oncocytoma is a rare entity, with around 20 cases reported in world literature after surgical extirpation. Though it is a benign and nonfunctional tumor, rarely, we come across a functional adrenocortical neoplasm. We present an adrenal oncocytoma which mimicked a functional adrenal tumor.A 48-year-old male, a known diabetic and hypertensive for the last six years underwent a routine health checkup. Ultrasonography abdomen revealed a well-circumscribed, hypoechoic left suprarenal mass lesion measuring approximately 5 × 4.8 cm. His diabetes was well controlled, but the blood pressure (BP) was around 168/94 mmHg on regular treatment with three drugs . Though occasional palpitations were present, there was no history of headache, seizures, flushing, diaphoresis, weakness of limbs, abdominal striae, petechie or cushingoid features. Abdominal examination did not reveal any palpable mass, striae or bruit. All examinations, cardiac, ophthalmic, neurological were within normal limits.The 24h urine estimation of Vinyl mandellic acid was 7.3 mg/vol  and catecholamines were 236 microgm/vol . Contrast enhanced computerized tomography (CECT) abdomen revealed a well-circumscribed nonenhancing 5.3 × 4.9 cm left suprarenal mass lesion . In viewGrossly, the specimen weighed 40g and measured 5.2 × 5.0 × 3 cm. The external surface was smooth with an intact capsule. Cut section showed yellow to brown areas. There was no evidence of hemorrhage or necrosis. Normal adrenal gland was identified compressed at the periphery. Multiple sections were taken from different areas for histopathological examination.Microscopically, the tumor showed nests and diffuse sheets of polygonal cells with abundant granular eosinophilic cytoplasm, pleomorphic nuclei with central nucleoli. Few multinucleate giant cells were also evident. The nests of cells were surrounded by capillary network. There was no evidence of mitosis, necrosis, capsular or vascular invasion. Normal adrenal tissue was seen at the periphery . The tumThe postoperative period was uneventful and the daily quantum of antihypertensives required reduced significantly. His BP could be controlled only with Tab. Atenolol 25 mg OD. After six months, repeat CECT abdomen was normal. The BP was well under control (average 146/ 84 mm Hg), with reduction in 24h urine excretion of catecholamines to 64 microgm/vol. The patient is on regular follow-up.Neoplasms composed entirely of oncocytes are well described in the kidney, thyroid, salivary glands and other rare sites like the pituitary, parathyroid, lacrimal gland, respiratory tract and choroid plexus. Oncocyti2Adrenal oncocytomas usually occur in the age group of 27-72 years with more cases documented in females. These tumors vary in size from 3-15 cm and weigh between 3-865g. Fibrous encapsulation is characteristically seen in all these tumors and on cut section tend to be mahogany to tan brown. Microscopically, these tumor cells are arranged in nests, solid, trabecular, papillary or tubular patterns. On electron microscopy, tumor cells show densely packed mitochondria.[


Primary Ewing's sarcoma or primitive neuroectodermal tumor of the genital tract of women is uncommon. Rarer still is its occurrence in the vagina, with only five cases described so far. Out of these, only one case was confirmed using molecular analysis.We present an extremely rare case of Ewing's sarcoma or primitive neuroectodermal tumor in a 17-year-old Indian girl. She presented with a vaginal mass that was initially diagnosed as a malignant round cell tumor. Immunohistochemistry showed diffuse positivity for vimentin, membranous positivity for MIC2, and positivity for BCL2 and FLI-1. On the other hand, she was negative for cytokeratin, epithelial membrane antigen, desmin, Myo D-1, myogenin and smooth muscle actin. A diagnosis of primitive neuroectodermal tumor was thus offered. Furthermore, a molecular analysis of our patient using reverse transcription-polymerase chain reaction technique showed positivity for t  (EWSR1-FLI1), thus confirming the diagnosis of a Ewing's sarcoma/primitive neuroectodermal tumor. Our patient was offered chemotherapy on Institutional protocol EFT 2001.This is a rare case of primary vaginal Ewing's sarcoma or primitive neuroectodermal tumor, which was confirmed with molecular analysis, in the youngest patient known so far. This study reinforces the value of integrating morphological features with membranous MIC2 positivity, along with application of molecular techniques in objective identification of an Ewing's sarcoma or primitive neuroectodermal tumor at uncommon sites. Primary extraosseous Ewing's sarcoma, also called primitive neuroectodermal tumor (PNET) is uncommonly documented as occurring in the genital tract of women, such as the uterine corpus, the cervix and the ovary -3. The vin situ and an enlarged uterus with pyometra. She underwent routine laboratory, radiological, and pathological investigations.A 17-year-old girl from central India presented with complaints of whitish, foul-smelling vaginal discharge with fragmented bits that she had been experiencing for a year prior to presentation. She had no history of any associated vaginal bleeding. Prior to presentation, she underwent alternative treatment but the symptoms had persisted. A month before she presented to our hospital, she was referred to us with acute urinary retention. She was catheterized. Her clinical examination showed that her general condition was fair. She had a Foley's catheter Her laboratory findings are as follows: low haemoglobin (Hb) level at 9 g/dl, low red blood cell count (RBC) at 3.32 ; low haemoatocrit at 26.8% ; low mean corpuscular volume (MCV) at 80.0 . Meanwhile, her C-reactive protein value was high at 8.54 mg/dl . She also had high serum lactate dehydrogenase (242 U/L) and alkaline phosphatase levels.Our patient also underwent radiological investigations. Her magnetic resonance imaging (MRI) showed a soft tissue mass involving her anterior and posterior vaginal walls and her anterior sacrum. Her urinary bladder was displaced anteriorly. The mass was hyperintense on T2-weighted and hypointense on T1-weighted imaging. She had no pelvic lymphadenopathy. Her ureters were also identified along with her visualized bones. Her chest X-ray revealed no abnormality.A computed tomography (CT) of her thorax showed a sub-centimeter-sized mediastinal node and multiple nodules in her bilateral lung parenchyma and subpleural location. Her pleural spaces, heart, great vessels, and bilateral bronchi were normal.Her abdominal CT showed bilateral kidneys with pressure changes and with dilated ureters. Her other organs and biliary tree were normal. No lymphadenopathy was identified.Her pelvic CT scan revealed a heterogeneously enhanced mass measuring 10 × 9.8 × 9 cm that involved her vagina and caused a narrowing of her cervix. Her cervix was also dilated with fluid and air. The mass was seen as extending up to her introitus and involving her perineum. As a result, her urinary bladder was pushed anteriorly towards the right side. Bilateral adnexae were noted with no ascites or any associated lymphadenopathy  isolation and reverse transcription polymerase chain reaction (RT-PCR). Her total RNA was isolated from formalin-fixed, paraffin-embedded tissue sections (FFPE tissue) using a Recover All Total Nucleic Acid Isolation kit . The extracted RNA was then treated with RNase-free DNase I before cDNA preparation. Her RNA was then reverse transcribed into cDNA using the Superscript First strand synthesis system (Invitrogen). A total of 500 nanograms of her total RNA was briefly reverse transcribed into cDNA using random hexamers at 42°C for 50 min, which was then followed by 70°C for 15 min.The synthesized cDNA from our patient was then treated with RNase H for 20 min at 37°C to remove the RNA-DNA hybrids. A total of 2 μl from the reaction was PCR amplified using EWS 22.3 forward primer (5'-TCC TAC AGC CAA GCT CCA AGT C-3') and FLI1 11.3 reverse primer (5'-ACT CCC CGT TGG TCC CCT CC-3') in a 20-μl reaction volume containing 10 pmol each of the forward and reverse primers, for a total of 20 μl PCR master mix . PCR conditions from her samples were as follows: 35 cycles of 94°C for 30 s, 65°C for 1 min, and 72°C for 1 min. Her amplified PCR products were checked in 10% polyacrylamide gel and stained with silver nitrate. Two positive controls (EWS-FLI1 type-I and type-II PCR product cloned into pTZ57R/T vector) and one water-only (no cDNA) negative control were included in each run. To check the quality and integrity of the cDNA, FOXO1A was amplified as a housekeeping gene .Meanwhile, our patient's pathological findings were achieved through conventional H&E. The stained sections showed a malignant round cell tumor, with undifferentiated cells arranged in a diffuse pattern and exhibiting focal rosettes. The cells were found to be small with scanty cytoplasm and uniform chromatin. On IHC, her tumor cells were diffusely positive for vimentin and showed membranous positivity for MIC2, along with areas of BCL2 and FLI-1 positivity. On the other hand, her tests for CK, EMA, desmin, Myo D-1, Myogenin and SMA were negative. Figures .We then offered our patient a diagnosis of PNET, although we also considered a close differential diagnosis of synovial sarcoma. In view of the uncommon location of her mass, molecular studies were recommended.On molecular analysis using the RT-PCR technique, t   (EWSR1-FLI1) turned out positive, thus confirming the diagnosis of primary Ewing's sarcoma or PNET of the vagina. Figure . Her SYT2), ifosamide (2.0 g/m2), etoposide (100 mg/m2), cyclophosphamide (600 mg/m2), doxorubicin (60 mg/m2), and actinomycin (1.0 mg/m2). The induction phase was followed by local treatment with radiotherapy (RT), during which our patient was found to be clinicoradiologically free of the disease. Following this, she was put on maintenance chemotherapy, including the aforementioned drugs. She was also put on regular follow-up examinations for the next seven months.Following our diagnosis of primary Ewing's sarcoma or PNET of the vagina, our patient was subjected to combination chemotherapy for 9 weeks based on institutional protocol for Ewing's family of tumors (EFT) 2001, including drugs such as vincristine  are defined as round cell sarcomas that show varying degrees of neuroectodermal differentiation and are identified by light microscopy, IHC and electron microscopy. While Ewing's sarcoma lacks neuroectodermal differentiation, a PNET possesses it . Both, hPrimary extraosseous Ewing's sarcoma/PNET is rarely diagnosed in the genital tract of women. It rarely occurs in the vagina, as in the case we describe in this report. Previously documented cases involved the presence of mass and associated pain -8 Table.Our patient's biopsy revealed a malignant round cell tumor below an unremarkable mucosa. In view of this location, differential diagnoses included poorly differentiated carcinoma such as a small cell carcinoma and round cell sarcomas like a Ewing's sarcoma/PNET, monophasic synovial, and rhabdomyosarcoma. The presence of uniform cells with focal rosettes, however, made a diagnosis of rhabdomyosarcoma less likely. On IHC, diffuse positivity with vimentin and lack of CK, EMA, CK7 ruled out a carcinoma. Diffuse membranous MIC2 positivity was more in keeping with a Ewing's sarcoma/PNET than neuroendocrine carcinoma and a synovial sarcoma that also shows MIC2 and cytoplasmic expression.Meanwhile, MIC2 positivity is also identified in rhabdomyosarcomas and lymphomas. However, the lack of desmin, myogenin and MyoD-1 ruled out rhabdomyosarcoma, while LCA negativity ruled out a diagnosis of lymphoma. In addition, FLI-1 positivity was also helpful even though its expression is noted in lymphomas, rhabdomyosarcomas and synovial sarcomas . While tThe value of identifying Ewing's sarcoma/PNET as the correct diagnosis is necessary in coming up with specific treatment for patients. Despite its aggressive behavior at osseous sites, this tumor responds to specific chemotherapy. All the cases documented so far have been subjected to chemotherapy with options of surgery ranging from wide excision to total abdominal hysterectomy -8. AdjuvThe treatment outcome for patients with primary vaginal Ewing's sarcoma/PNET has been favorable in so far as documented cases are concerned. No metastasis or causalities have also been reported -7. In coOur case report describes a rare site of primary vaginal Ewing's sarcoma/PNET in the youngest patient known so far. It reinforces the value of IHC, including membranous MIC2 positivity and with molecular analysis, in the objective identification of this sarcoma at unusual sites like the vagina. Correctly identifying tumor type enables clinicians to identify specific CT procedures as treatment mainstay. The identification and documentation of more cases with a clinicopathological index of suspicion, the confirmation of diagnosis using ancillary techniques, and the management and follow-up results would greatly contribute to the existing knowledge of diagnosis and outcome of PNETs developing at unusual sites.Written informed consent was obtained from our patient for publication of this case report and any accompanying images. A copy of written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.BR was the diagnosing pathologist involved in patient care. He also provided the concept and design of the study, helped prepare the manuscript for publication, and contributed the artworks used in this case report. SQ contributed clinical details mentioned in the manuscript and helped in preparing the manuscript for publication. RB performed the molecular analysis of samples from our patient described in the manuscript. SBD analyzed and reported the molecular results. SM provided radiological inputs and was also involved in diagnosing our patient. PK and AM provided the clinical details cited in the manuscript. NAJ supervised the preparation and approval of the manuscript for publication. All authors read and approved the final manuscript.


Hepatic hydatid cysts may cause serious complications. Intraperitoneal rupture of hepatic hydatid cyst is rarely seen and the prognosis can be fatal. By experience, we know that it might be difficult to diagnose an unruptured cyst expulsed into the peritoneal cavity. In this report, we present the case of a 54-year-old man with an intraperitoneal cystic mass of 10 cm of diameter which had extruded out from the liver due to a blunt abdominal trauma. Intraperitoneal rupture of hydatid cyst of the liver is a rare situation and the prognosis is usually fatal. However, expulsion into the peritoneal cavity without perforation is a much more unusual complication of a hepatic hydatid cyst. We report the observation of a 54-year-old man who was diagnosed of biliary peritonitis due to an unruptured hepatic hydatid cyst after a blunt abdominal trauma.A 54-year-old man presented in the emergency unit (EU) of our hospital with severe abdominal pain, 2 days after falling down on his abdomen at home. Patient history was remarkable for previously diagnosed multiple hepatic hydatid cysts, and he had been on albendazole regimen for 1 month. He had no known disease other than a recent blunt abdominal trauma as he had fallen down on his abdomen 2 days prior to showing up in the EU. As he stated, he had never had a similar type of pain before this house accident, and the pain had started to ‘really hurt’ several hours after ‘the fall.’ Intensity of the pain was gradually increasing for 2 days.3, eosinophils: 0.43 × 10³/µl). The patient's overall clinical and laboratory findings clearly suggested the diagnosis of acute abdomen and immediate surgical intervention was considered.Initial physical examination revealed diffuse abdominal tenderness on palpation with epigastric predominance and positive rebound tenderness at each abdominal quadrant. As per initial vital signs, arterial blood pressure: 140/90 mm Hg, heart rate: 96/min and the body temperature (axillary) was 37.5°C. Chest X-ray and acute abdominal X-ray series were normal. Abdominal ultrasonography showed three intrahepatic and one extrahepatic cystic masses , approxiPatient was promptly prepared and taken into the operating room. Abdominal exploration revealed considerable amount of serous free fluid with bile leakage in the abdomen. Sample fluid was spared for laboratory and remaining was aspirated. Further exploration was interesting as we found an unruptured hydatid cyst of 10 cm in diameter with an intact germinative membrane, which was enucleated spontaneously out from the fifth hepatic segment. We made this assumption since there was an entirely empty cystic cavity in the fifth segment with the same dimensions as the wandering extrahepatic cyst. Bile leakage from the wall of this cavity was clearly seen . We usedEchinococcus granulosus and less frequently with Echinococcus multilocularis.[Echinococcus species, adult tapeworms inhabit the small intestine of carnivorous definitive hosts , and echinococcal cyst stages occur in herbivorous intermediate hosts . In the typical life cycle, tapeworm eggs are passed in the feces of an infected dog and ingested by grazing sheep; hatch into embryos in the intestine, penetrate the intestinal wall, and then carried by blood to major filtering organs (liver and/or lungs). Embryos localize in a specific site and they develop into larval echinococcal cysts where new tapeworm heads (protoscolices) are produced by asexual reproduction. Dogs are infected by ingesting viscera of home-slaughtered livestock containing echinococcal cysts. Protoscolices attach to the dogs' intestinal wall and develop into mature adult tapeworms. In primary echinococcosis, metacestodes develop from oncospheres after peroral infection. In secondary echinococcosis, larval tissue proliferates after being spread from the primary site . Most patients have single organ involvement with a solitary cyst, and two-thirds of the patients experience liver echinococcosis. Lung is the second most commonly involved organ. Cysts are surrounded by the parasitic host tissue called pericyst where larval endocysts are located. Outermost layer of the cyst is a hyaline membrane which covers the multipotential germinal layer where protoscolices and brood capsules are produced. Cystic cavity is filled with clear fluid, brood capsules, protoscolices and also daughter cysts are frequently found in the cyst. Symptoms of the disease are variable and depend on the cyst localization, size, complications caused by rupture, bacterial infection of cysts, spread into bile ducts or blood vessels, immunologic reaction such as membranous nephropathy, asthma, and anaphylaxis. Cystic echinococcosis is rarely fatal and death occurs because of anaphylactic shock or cardiac tamponade in case of cardiac echinococcosis. The incidence of rupture of the hydatid cyst of the liver is about 15-40% of the cases.[2 diameter without rupture.[Hydatid disease is not an uncommon problem, especially in endemic countries. Human hyocularis.2 In the he cases. Trauma i rupture. In our c


A 21 day old male child presented with non bilious vomiting and abdominal mass.This case is reported because pyloroduodenal duplication cysts are an extremely rare congenital anomaly, whose clinical presentation often mimics those of hypertrophic pyloric stenosis. Ultrasound examination showed cystic mass at pyloric region and barium study was suggestive of extrinsic mass compressing the pyloric region. A laparotomy, a tense cystic mass was present at the pyloroduodenal junction (PDC) which was resected and end to end anastomosis was done. Patients followed an uneventful recovery and doing well.The clinical and radiological analysis can reveal configurational changes consistent with a large extrinsic mass rather than muscular hypertrophy and can lead to accurate preoperative diagnosis. Gastrointestinal duplications are observed in 1 of every 4500 autopsies, predominantly in white males. The small intestine is the most frequent site involved, whereas gastric, duodenal, rectal, and thoracoabdominal involvement is relatively rare. Pyloroduodenal duplication cyst is an extremely rare and only two cases have been reported in English literature one by Tihanaski . PylorodA 21 days old male neonate weight 3.2 kg, was presented to us with history of recurrent non bilious projectile vomiting after taking feeds. On physical examination a mobile mass of 3 cm × 2.5 cm was palpable in right upper abdomen. Ultrasonography of abdomen revealed a cystic mass in the pyloro-duodenal region measuring 2.6 × 2.3 cm  are an extremely rare congenital anomaly, presents with symptoms of gastric outlet obstruction and palpable mass which is clinically indistinguishable from IHPS. Enteric duplications can occur anywhere along the GI tract, but are most commonly found in the ileum and are perhaps rarest along the pyloric channel  and reprWritten informed consent was obtained from the parents of the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.SPS and PS operated the patient and reviewed the literature. VDU and RJ was the main writer of the manuscript. ANG and DKG moderated the manuscript.All authors read and approved the final manuscript