Lipomatous hemangiopericytoma (LHPC) is a very rare tumor with uncertain biologic behavior. This report handles with the clinical, radiological and histological features of the 1st case to date of LHPC, occurring in the thoracic inlet and the surgical technique is described.A 18-year-old, non smoking, male, patient, with no significant medical history, was referred with a 6-month history of right-sided chest pain. On physical exam, an oval-shaped mass was palpated in the right supraclavicular area. Imaging findings revealed a well-circumscribed, inhomogeneous tumor in the posterior compartment of the right thoracic inlet, with dimensions of 5.3 X 8.9 X 10.8 cm. The tumor seemed to compress the adjacent structures, but not clearly to infiltrate them, while it’s lower limit reaching the 4th intercostal space.An incisional biopsy was performed, resulting in the early diagnosis of LHPC. A right upraclavicular incision was performed at a second time to access the tumor. The clavicle was transected at its middle in order to enlarge the surgical field, allowing that way excision of the tumor with intact capsule, through a combination of sharp and blunt dissection. A lot of feeding vessels were clipped or ligated during dissection within the thoracic inlet. The patient had an uneventful short postoperative course. The diagnosis of LHPC was confirmed by two more laboratories. He underwent adjuvant radiation therapy in the right thoracic inlet and supraclavicular area, to reduce the risk of recurrence, because the tumor was violated during the incisional biopsy.The thoracic inlet is one more location for the very rare LHPC. The anatomic topography of the mass requires special maneuvers to achieve safe removal of tumor with intact capsule, such as transection of the clavicle. Adjuvant radiation should be discussed within the multidisciplinary oncology team, as the global experience with these rare tumors is limited.


Primary peritoneal carcinomas are rare, highly aggressive malignant neoplasms containing both sarcomatous and carcinomatous elements. Surgical debulking is the mainstay of treatment for primary peritoneal carcinomas. Systemic chemotherapy is advised in all cases because of the early spreading of these tumours. We report on a case of primary peritoneal carcinosarcoma occurring in a 22-year-old woman. Carcinosarcomas are highly aggressive biphasic neoplasms composed of carcinomatous and sarcomatous elements. Mostly occurring in the female genital tract in elderly postmenopausal women, they have also been described in head and neck, gastrointestinal tract, biliary tract and peritoneum . PrimaryA 22-year-old unmarried female presented with a two month history of non-colicky lower abdominal pain. Clinically, there was a 10 × 12 cm mobile mass arising from the pelvis reaching up to the umbilicus. Contrast enhanced computerised Tomography (CECT) abdomen revealed an 11 × 11 × 9 cm heterogeneously enhancing predominantly cystic mass in the lower abdomen superior to the bladder and uterus extending upwards to aortic bifurcation. It was abutting the anterior surface of the left psoas muscle and left external iliac artery, and anteriorly reaching up to the parietal wall. The ovaries, uterus, and rest of the viscera were normal. Serum tumour markers  were within normal limits. The patient underwent an exploratory laparotomy. Intra-operatively minimal hemorrhagic ascites were noted. A 12 × 10 cm heterogeneous solid cystic mass was noted in the sigmoid mesentery adherent to the psoas fascia posteriorly. Both ovaries and uterus were seen to be normal. The mass was excised completely with a segment of sigmoid colon.Gross pathology showed an intact 13 × 9 × 10 cm mass with a segment of sigmoid colon. Cut section showed grey white fleshy solid areas, interspersed areas of haemorrhage, along with multiple cystic spaces filled with thick fluid. Microscopic examination showed that the cystic areas were lined by tall columnar cells arranged in papillary and cribriform pattern and with pale oeosinophilic cytoplasm and pleomorphic nuclei. Solid areas showed sheets of spindly cells with moderate cytoplasm and elongated pleomorphic nuclei. There were five to six mitosis/10 HPF. Neoplasm was adherent to the serosa of the sigmoid colon. Adjacent colonic mucosa was normal. The epithelial cells were positive for cytokeratin (CK) and focally for carcinoembryonic antigen (CEA) and the spindly cells were positive for vimentin .Post operatively she was given six cycles of chemotherapy with ifosfamaide and cisplatin. CT scan taken after completion of chemotherapy showed a recurrent lesion within the abdomen measuring 6 × 5.8 × 4.3 cm size. In view of the progressive disease, carboplatin, and paclitaxel combination chemotherapy was started. CT scan taken after two cycles showed that the disease was progressing on treatment and hence, a cycle of Adriamycin with cyclophosphomide was given. There was no clinical response with the third line chemotherapy also and the patient expired 9 months after starting the treatment.in vitro studies, immunohistochemistry, and molecular studies have shown that they are derived from monoclonal cancer cells which exhibit sarcomatous metaplasia [Carcinosarcomas or malignant mixed Mullerian tumours are rare, highly aggressive biphasic neoplasms comprised of carcinomatous and sarcomatous components, which accounts for one to three per cent of all uterine malignancies  and eventaplasia  and the Literature search revealed 35 cases of primary peritoneal carcinosarcomas , 12–15. Owing to the rarity of primary peritoneal carcinosarcomas, limited data regarding the management exists. The mainstay of treatment is surgical debulking, but most cases of carcinosarcomas have wide spread metastasis at the time of presentation, making optimal tumour debulking difficult . Systemip = 0.016 and p = 0.040). There is no significant difference in survival between Group 2 and Group 3 (p = 0.696). This indicates that a patient with primary peritoneal carcinosarcoma with tumour originating from pelvic peritoneum or uterine serosa is having better survival than a patient with tumour originating from other peritoneal surface. The Kaplan–Meir survival graph is given in Literature review shows that all patients were managed by surgery followed by chemotherapy when possible –3. OveraPrimary peritoneal carcinosarcomas are rare tumours associated with a poor prognosis. Tumours arising from the bowel serosa, abdominal wall, omentum or retroperitoneum seem to carry a poorer prognosis than those arising from the pelvic peritoneum. Since these tumours are similar to uterine carcinosarcomas where recent evidence has shown that the tumour behaviour is dictated by the epithelial component, chemotherapy with platinum taxane combination may work better as first line therapy.


We report an unusual case of idiopathic unilateral adrenal haemorrhage (AH) in a 55-year-old patient. This rare case had two characteristics that made it worth of report. First, idiopathic adrenal haemorrhage is very uncommon, and second it was presented as a huge, 23 cm diameter and 2,123 gr weight, “silent” adrenal mass. It is important to distinguish a benign lesion like this from a neoplasm, although we were not able to identify it preoperatively and the diagnosis was only made after the excised specimen was examined by a group of experienced histopathologists. Only a few similar published cases, to our knowledge, are described in the worldwide literature and even fewer of this size. Adrenal haemorrhage (AH) is an uncommon condition more frequently presented in adolescents than in adults 7 : 1). Symptoms and physical findings are not specific and vary among patients. Idiopathic, unilateral AH is a rare entity that either may have an acute presentation  or may present as an asymptomatic adrenal mass, as has been reported by other series . Symptom. Most auThe patient was admitted to our department for the evaluation of a palpable mass in the right side of his abdomen. His past medical history and any medication he ever had were irrelevant with the current problem. Physical examination revealed a palpable mass, approximately 20 × 20 cm in size, occupying an area from the upper to the lower right abdominal quadrantus. No pain or tenderness around the mass was reported and the patient mentioned only some discomfort when he was lying at night. 9/L, without any hepatic, renal, or adrenal dysfunction from the blood or urine examinations he had. His vital signs were within the normal range and the laboratory data showed mild anaemia (haemoglobin: 10.9 g/dL), WBC 8190 × 10Moreover, his serum tumor markers and coagulation function were all within normal values. Contrast-enhanced computed tomography (CT) showed a huge, well-circumscribed, 23 × 20 cm, cystic lesion between liver and right kidney with the enhancement of the capsule around the tumor.  No liver metastasis or lymph node swelling was noted. The gadolinium-enhanced magnetic resonance imaging (MRI) that followed showed a huge multicystic lesion, in the region of the right adrenal gland, with a thick capsule containing several septations especially in the periphery of the lesion Figures , and 3.  The abnormality deviated the liver upwards, the right kidney downwards and posteriorly, the pancreas to the left, and the abdominal aorta and IVC were deviated to the left. The vascularity of the lesion was not clearly demonstrated, but there were large venous structures peripheral to the lesion draining into the IVC. The previously described findings were discussed in the MDT meeting and the decision for surgery was made.During the 3-hour exploratory laparotomy that followed the lesion was carefully dissected from the surrounding structures  and excised along with the surrounding fatty tissues. The lesion was firmly attached to the IVC and right renal vein, so a full isolation of both was necessary and successfully performed Figures  and 5. PThe histopathology report described a yellow-whitish lesion with traces of normal adrenal gland tissue and appearances of adrenal haemorrhage. They reported that the lesion was consisted entirely of blood clots, fibrinoid debris, and ghost outlines of proexisting vessels. Finally, they noted that it was covered by a fibrous pseudocapsule and no evidence of malignancy was noted. The final diagnosis for this patient was idiopathic adrenal haemorrhage.Adrenal haemorrhage is a rare yet potentially life-threatening event that occurs both in traumatic conditions and in a variety of nontraumatic conditions. To our knowledge, it is an autopsy finding in most of the reported cases . It may Clinical manifestations of adrenal haemorrhage can vary widely depending on the degree and rate of haemorrhage, as well as the amount of adrenal cortex compromised by haemorrhage, as similar cases have reported –13. AlthAccording to the underlying cause, clinical presentation varies and symptoms may include abdominal or flank pain, nausea, vomiting, weakness, weight loss, or mental confusion . Those are often accompanied with physical findings of fever, hypotension, abdominal tenderness or distension, and a palpable abdominal mass, as discussed elsewhere , 11.The laboratory findings also vary and may include a falling haematocrit level, leukocytosis, or electrolytes abnormalities , 16. SomImaging assists and complements the clinical and endocrine evaluation of adrenal masses. Various studies showed that, although ultrasonography is a fast, low-cost, and widely available method, it is usually limited to infants and children , 20. Com Some have proposed a descent approach to incidentally discovered adrenal mass, which includes assessing the mass size, determination of whether is solid or cystic, and excluding biochemically active tumors by all the appropriate laboratory studies , 23. TheAdrenal haemorrhage is a rare cause of adrenal mass and is generally associated with trauma, infection, or bleeding diathesis. Only a few isolated cases of similar pathology  exist in the literature and nearly all of them were postoperatively diagnosed as such. Though rare, signs of adrenal haemorrhage may include an asymptomatic adrenal mass and should be considered in the differential diagnosis of this problem.


AbstractSinusoidal hemangioma is a rare type of cavernous hemangioma with different clinico-pathological aspects. They are usually localized in the extremities with interest in the subcutaneous layer. The new imaging techniques play an important role in diagnosis, evaluation and follow-up of these types of tumors.We describe the case of a 21-year-old patient, four times operated for a recurrent soft tissue tumor, located intramuscularly in the distal third of the upper limb. Plain X-ray and computer tomography (CT) showed a nonspecific mass with calcification. The MRI (magnetic resonance imaging) exam demonstrated a lobulated heterogeneous signal tumor mass in the biceps brachial muscles, with high signal intensity on T2 weighted images and intermediate signal on T1 weighted images. MRI accurately assessed the extent of the tumor and evaluated the recurrence. MRI imagings combined with contrast-enhanced sequences were used to classify the lesions in low flow vascular disorders. CT angiography with multiplanar reconstructions (MPR), maximum intensity projections (MIP) and volume-rendered reconstructions (VR) was useful in confirming the venous origin of the tumor. Hemangiomas are histopathologically divided in capillary, cavernous, arteriovenous, venous, and mixed variations [2]. Sinusoidal hemangioma represents a rare subtype of cavernous hemangiomas, which presents with different clinico-pathological aspects. Magnetic resonance imaging (MRI) plays an important role in the assessment of these lesions, concerning the diagnosis the type of lesions and evaluation of the extent, for proper treatment guidance. Also MRI has emerged as a noninvasive diagnostic tool in postsurgical and recurrence evaluation of hemangiomas [3].Hemangiomas are the most common benign tumors of soft tissues and the intramuscular types comprise 0.8% of all cases .Recently angiography has probably been replaced by MR or CT angiography in providing anatomical vascular information, the main assignment for this imaging technique being the embolization prior to resection [In this case, overlapped by numerous recurrence and multifocality, the therapeutical approach will probably take under consideration in the future other methods such as cryotherapy, electrocoagulation or laser therapy.The particularities of our case are represented by topography and histopathology of a rare type of cavernous malformation. The imaging techniques with the new post processing software along with the clinical data play an important role for diagnosis and evaluation of these lesions in order to avoid diagnostic pitfalls and also to correctly guide the treatment.


A 12 years old boy, with no past medical history, presented to the neurology department with a 7 years clinical course of untreated limping, progressive kyphosis, repeated shoulder dislocation, and, recently, progressive limbs weakness. On examination, the patient was short trunked with fixed cervical hyperextension and thoracolumbar kyphosis. Also noted were genu valgum and ligamentous laxity. Neurological examination showed a spastic quadriparesis (3/5), exaggerated reflexes and bilateral Babinsky sign. Sensory function was normal, so as intelligence. Axial and appendicular X-ray radiographs demonstrated odontoid hypoplasia and possible occipito-cervical dislocation, a platyspondyly , acetabular dysplasia  and metacarpal irregular proximal ends . MRI of the craniocervical junction revealed severe spinal cord compression at C1-C2 level with marked instability and dislocation , odontoid hypoplasia associated with a soft tissue mass around . All were suggestive of the mucopolysaccharidoses? origin. Laboratory investigations showed an over excretion of the keratane sulfate which comforted the diagnosis. The differential diagnosis of the image can be made with the atlanto-axial subluxation in rheumatoid arthritis, ankylosing spondylitis, and pseudo-gout (CPPD). The patient was proposed for a cervical posterior decompression with occiput-to C2 fusion, which was declined by his family when surgical procedure risks were explained.


Lymphangioma is an uncommon benign lesion that usually occurs during childhood. Its occurrence in adults is rare. Its presentation in the abdomen is even rare. This case report describes a case of omental lymphangioma presented as retroperitoneal lump. Subsequent imaging, operative, and histological findings revealed omental lymphangioma. Laparotomy done under general anesthesia, a 10 × 12 cm cystic swelling arising from omentum, identified complete excision of the cyst done and send the specimen for histopathological examination. Biopsy report came as omental lymphangioma. Complete surgical excision is the treatment of choice. Prognosis is excellent and recurrence rate is very low if resection is complete. During two years of followup no recurrence was detected. Omental lymphangioma is very rare presentation among abdominal lymphangiomas specifically in adults. Complete excision is the treatment of choice. Long-term followup is required to detect recurrence. Although rare abdominal lymphangiomas are more common in boys than in girls, they usually occur during childhood. They are reported to occur most commonly in the mesentery, followed by omentum, mesocolon, and retroperitoneum. They arise in all age groups and have variable presentation. But they are extremely rare in adult population [Lymphangioma is an uncommon benign lesion that is due to result from a developmental failure of lymphatic system/inflammation of lymhatics causing obstruction. Lymphangiomas occur in many anatomical locations. Most cases (95%) are found in the neck and axillary regions, whereas other sites, such as the mesentery, pulation . LymphanA 35-year-old female patient presented with a history of pain abdomen. On examination abdominal lump of size 10 × 12 cm, occupying the right lumbar region extending to umbilical region smooth surface, soft in consistency, as retroperitoneal in position, was identified. Routine blood investigations show no obvious abnormality. As ultrasound abdomen shows 10 × 12 cm cystic anechoic lesion with multiple septations seen suggestive of lymhangioma. CECT abdomen was done showing multiseptate cystic mass with contrast enhancement of cyst walls suggestive of lymphangioma arising from omentum. Laparotomy done, intra operatively a 10 × 12 cm cystic mass occupying umbilical, right lumbar regions extended up to sub hepatic region identified. Complete excision was done. Postoperative period was uneventful. During follow-up period no recurrence was found .The etiology of lymphangiomas remains unclear. Because lymphangiomas occur mainly in children, the majority of cases are thought to derive from a congenital abnormality of lymphatic system. Clinical presentation can be variable and nonspecific. Acute symptoms include acute abdomen, distension, vomiting, and fever. Chronic symptoms include progressive abdominal distension and pain. Plain radiographs may show noncalcified soft tissue mass, displacement of intestinal loops and small bowel obstruction. Ultrasonography and CECT. are highly sensitive tests that can be used in diagnosis . SonograHowever there can be no specific radiological features to differentiate between these options—histological evaluation may be necessary. Ascites and lymphangioma can also be difficult to differentiate . The preOmental lymphangioma is very rare presentation among abdominal lymphangiomas specifically in adults. Complete excision is the treatment of choice. Long-term followup is required to detect recurrence.


Temporal triangular alopecia (TTA), also known as congenital triangular alopecia, is a nonscarring, noninflammatroy, circumscribed form of alopecia. TTA has been associated with several disorders, such as Phakomatosis Pigmentovascularis. Hair restoration surgery using follicular unit transplantation has been a successful treatment modality for TTA. Herein we report such a success that was sustained for over six years. Temporal triangular alopecia (TTA), also known as congenital triangular alopecia, is a nonscarring, noninflammatory, circumscribed form of alopecia. The alopecic lesion is usually asymptomatic and present at birth or during the first nine years of life. Lesions are stable and mostly presents with roughly triangular, oval, or lancet-shaped patches in the frontotemporal region that are characterized by normal hair density of vellous hair and normal epidermis. The condition mostly occurs unilaterally, but bilateral cases 13.5–20%) can occur [3.5–20% cTTA has been associated with several disorders , one of Hair restoration surgery using follicular unit transplantation has been reported to be a successful treatment for temporal triangular alopecia, where followup for 16 and 24 months postoperatively showed maintenance of the cosmetic end result [A 25-year-old Caucasian male presented to our practice in 2003 complaining of an alopecic scalp lesion in both his temples, which was present since his childhood. The lesions did not increase in size nor did it produce any symptoms.Scalp examination revealed an oval, slightly lancet-shaped hypotrichotic patches measuring approximately 8 × 5 cm and 8 × 4 cm on the left and right temples, respectively, located supra-aurically Figures  and 2. T2. Patient had no postoperative complications after either procedures. Sutures from the donor area were removed seven and ten days after the first and second sessions, respectively. Figures Hair restoration surgery with follicular unit transplantation was offered to the patient as a possible treatment which he accepted. The preoperative workout was unremarkable. Patient underwent two sessions one year apart where a 0.7 cm × 5 cm strip was harvested from the occipital donor region, the right half on the first session and the left half on the second session. In total, 1449 follicular units were obtained from both surgeries using stereomicroscopic dissection. The grafts were placed in the TTA patches with a final density of approximately 35 FU/cmReview of the literature shows, including our case, that hair restoration surgery using follicular unit transplantation is an effective and successful treatment modality for temporal triangular alopecia. In our case report, there was, as well, a consistent result over a six-year followup. We recommend that hair restoration surgery should be offered as a first-line treatment option for this particular type of alopecia.


This case report describes the endodontic treatment of a large cyst-like periradicular lesion a 29-year-old female with a large chronic periapical abscess in the region of right maxillary sinus presented into private practice, accompanied with non-vital first upper molar and poorly root treated second upper molar. Conservative root canal treatment was carried out for both of the involved teeth in a single appointment. Post operative examination after two weeks revealed complete resolution of the sinus tract. The clinical and radiographic examination after 9 months revealed complete periapical healing. The appropriate diagnosis of periradicular lesion and the treatment of the infected root canal system allowed complete healing of these large lesions without endodontic surgery. Pulpal tissue can become infected through various ways such as caries or trauma, making the pulpal tissue necrotic. The microbial aggregation or its by-products can infiltrate into periradicular tissues and stimulate the host defense system, resulting in periapical/ periradicular tissue destruction. Although this defensive lesion may be helpful to prevent further progress of the microbial infection, it is not self-healing and results in various types of lesions . The genA 29-year-old female attended a private endodontic clinic; her chief complaint was the presence of mild pain in right maxillary sinus area. The patient had no significant medical history. Right maxillary first molar was not previously root treated, was not carious and had no history of trauma. The adjacent second molar had poor endodontic root treatment with an incomplete obturation .Extra oral examination revealed no sign or symptom. Intraoral examination revealed a minor firm swelling of the vestibule above the molars and an associated sinus tract on the buccal alveolar process. Palpation produced purulent exudates and the mucosa in the region was inflamed. Teeth were not tender to percussion and were not mobile. Electric pulp test and cavity test exhibited negative results for right maxillary first molar.Gutta-percha cone was used to trace the path of sinus tract by periapical x-ray ; howeverThe patient´s clinical and radiographic findings seemed to suggest a large cyst-like peri-radicular lesion, most likely to be an infected radicular cyst of endodontic origin.One visit endodontic treatment was performed for the right maxillary first and second molars, in one session. After access cavity preparation, treatment was continued with a rubber dam in place. There were no exudates from the canals. Instrumentation was performed by Flexo-File  #15-40, using step-back technique, accompanying with copious irrigation with sterile normal saline between instruments. The working length was determined on the basis of radiographs. Obturation was performed with gutta-percha  and sealer  by lateral condensation technique .After two weeks of treatment, teeth were permanently restored with amalgam . The patient was recalled after one day, two weeks, 9 and 12 months. The signs and symptoms, including the sinus tract, had disappeared after two weeks of treatment.On 9 month and one year recalls, the patient had no sign and symptom; panoramic and periapical radiographic evaluation demonstrate-ed complete bony regression of the lesion  and Fig. ClinicaThis case illustrated a cyst-like periradicular lesion, most probably a radicular cyst. The exact diagnosis can be made by microscopic examination. However, the clinical diagnosis of a radicular cyst seemed rational because the lesion accompanied nonvital teeth, was more than 1.6 mm in diameter, and was bordered with a radiopaque line resembling cystic lesions . As mentioned in previous studies, in the cases of periradicular radiolucent lesions, sufficient biomechanical cleaning of the root canal system is the most critical factor for healing. It has been demonstrated that in these cases, non-surgical root canal therapy should be the first line of treatment  and apprPermanent restoration within two weeks of RCT also contributed to periradicular healing, as several studies have shown that an adequate coronal restoration-placed as soon as possible after RCT-plays an important role in the outcome of endodontic therapy -11.This patient was a young healthy subject and these factors will contribute to successful radiographical and clinical healing; previous studies have showed that the patient’s general health may have an influence on the healing process in periradicular lesions . AlthougRadiographic changes such as the increase in density of the lesion and trabecular regeneration, confirmed healing in addition to the absence of signs and symptoms. However it is difficult to be sure of complete healing with conventional radiographic techniques.In the present case, single visit root canal therapy without any intracanal medicament, proved successful in promoting healing of a large cyst-like periradicular lesion. The result confirms previous reports demonstrating that even large periradicular lesions can respond successfully to non-surgical single-visit endodontic treatment.


Adrenocortical carcinoma (ACC) is a malignant tumour arising from the adrenal cortex, while pheochromocytoma is a catecholamine secreting tumour of the adrenal medulla or extra adrenal sites. Both conditions are very rare, with incidence of approximately 1–2 cases per million adults annually. Most adrenocortical tumours are functioning. ACC can be associated with clinical Cushing syndrome and virilisation due to excessive production of cortisol and androgens, respectively. However, it is rare for ACC to present clinically as pheochromocytoma. We report a case of a 28-year-old lady who presented with paroxysmal hypertension and palpitations associated with raised urinary vanillyl mandelic acid. On examination, there was postural hypotension and ballotable mass in right lumbar region with no obvious features suggestive of Cushing syndrome or virilisation. A huge right suprarenal mass with areas of necrosis and calcification was noted on the abdomen CT. A right adrenalectomy was done. The histology was consistent with ACC. There are reported cases of ACC presenting with clinical features of pheochromocytoma but limited in number, accounting for barely a dozen cases in the literature. This pseudopheochromocytoma may be due to the presence of neuroendocrine features in ACC. BPBlood pressureCTComputed tomographyCXRChest X-rayECGElectrocardiogramMRIMagnetic resonance imagingVMAVanillyl mandelic acidACCAdrenocortical carcinomaPETPositron emission tomographyThough benign adreno-cortical tumours are common, adrenocortical carcinoma (ACC) is a relatively rare condition with diverse clinical manifestations due to excessive hormone production by the tumour, with Cushing syndrome and virilisation being the commonest clinical features . PheochrA 28-year-old single lady presented with palpitations on and off for one and half years, which was episodic with no obvious specific precipitating factors. These attacks were accompanied by excessive sweating, flushing, and cold extremities. She also experienced dizziness during change of posture particularly standing up from sitting. However, she denied fear of impending death or syncopal attacks. She was also diagnosed with hypertension one and a half years back at her first presentation of palpitation. She took prescribed antihypertensive medications irregularly. Her highest BP was 220/110 mmHg, and sometimes BP was stable without any medication. She noticed some degree of weight loss during these years.On physical examination, there were no features of virilisation or Cushing syndrome. Pulse rate was 120, regular with no special character or radiofemoral delay. Postural hypotension was noted with lying BP 140/100 mmHg and standing BP 120/90 mmHg. Precordium and respiratory examination were normal. There was a mass palpable in the right lumbar region, 5 × 5 cm, firm in consistency and ballotable. No tenderness or renal bruit noted. Left kidney was not ballotable. Grade II hypertensive retinopathy was seen on fundoscopy.Her ECG showed T wave inversion in leads I, aVL, V4-V6 with no evidence of left ventricular hypertrophy. CXR and serum electrolytes were normal. Ultrasonography of abdomen revealed right supra-renal mass of mixed echogenicity measuring 13.1 cm × 7.7 cm. On CT abdomen, there was a huge well-circumscribed soft tissue mass of 16.5 × 6.5 × 8.7 cm in right retroperitoneal region with break down areas and calcifications  and 2. AThe operation was performed by the urosurgical team after stabilization, which revealed a tumour mass of 17.8 × 12.7 × 7.6 cm in the right adrenal gland with adhesions to the right lobe of the liver and peritoneal wall. A right adrenalectomy was done. The histopathology reported an encapsulated adrenal tumour arising from the cortex, predominantly yellow on the cut surface with areas of haemorrhage, calcification, and necrosis. A moderate degree of atypia was noted, consisting of giant cells and spindle cells. However, there was no invasion into the adrenal vein or lymphatics. Because of limited resources, immunohistochemical staining cannot be performed. The impression of biopsy result was adreno-cortical carcinoma.Recheck urinary VMA 2 weeks after surgery was 5.4 mg/24 h, which was normal. Her BP was controlled with labetalol. The post-operative period was uneventful with improvement of symptoms.Adrenocortical carcinoma (ACC) is a very rare condition, accounting for 1–2 cases per million per year in adults [About one quarter to three quarters of ACC is functioning with excess hormonal production . VariousPheochromocytoma is a catecholamine secreting tumour which arises from adrenal medulla. It is also a rare neoplasm, with an estimated incidence of 2–8 cases per million per year . It accoThe size and appearance on imaging studies such as CT or MRI (magnetic resonance imaging) are useful in distinguishing benign adrenal tumours from ACC. The mean tumour size of ACC at the diagnosis was 11.5 ± 4.7 cm (range 3–28 cm) . The sizPathologically malignant adrenal tumours are differentiated from benign lesions by gross appearance of tumour weight, haemorrhages, capsular involvement as well as microscopic diagnostic score of Weiss. Moreover, broad fibrous bands, nuclear atypia, and necrosis are suggestive of malignancy . The preIt is extremely uncommon for ACC to present as pheochromocytoma. The initial presentation combined with biochemical evidence of excessive urinary VMA level lead to preoperative diagnosis of pheochromocytoma in our case. In a case report dated back to 1962, a 44-year-old man presented clinically with paroxysmal hypertension and sweating with raised catecholamines but histology revealed ACC . AnotherIn the present case, it was not the concurrence of both tumours but the functioning carcinoma of the adrenal cortex presenting with clinical features of pheochromocytoma which is unique and rare. To the best of our knowledge, this is the first reported case of ACC presenting with pheochromocytoma features from Myanmar.


We report a scrotal epidermal inclusion cyst located outside the median raphe which a rare entity in the absence of trauma and few cases have been reported. 47 year old male presents with a complaint of right sided testicular swelling and discomfort. On examination a 3 cm mass was palpated between the scrotum and the medial thigh on the subcutaneous tissue with a positive slip sign. Complete surgical excision of the cyst was performed. Histopathology confirmed epidermal inclusion cyst with no evidence of malignancy. Epidermoid inclusion cysts (EIC) are rare, benign cysts which occur from implantation of epidermal tissue into the dermis or subcutaneous tissues or in the testicle . This abA healthy, 47-year-old male presented with testicular discomfort and a palpable nodule in the upper scrotum just medial to the thigh. He reported that the nodule had been present for nearly seven years and had been slowly enlarging over the last few years. The mass had been initially asymptomatic, until recently when he began to experience some discomfort during exercise along with other activities. He denied any history of trauma or any prior surgeries to the area.On physical exam, a palpable 3 cm soft structure was present in the right scrotum as shown in  Given the low malignant potential, we did not send testicular markers and proceeded with excision of the mass. The cyst was found to be superior and lateral to the right testicle, but not originating from it. It was excised without difficulty. Intraoperative findings revealed the mass to be covered with a thin sac that contained necrotic, proteinaceous material. Pathological findings revealed benign epidermal inclusion cyst as shown in  EIC are benign tumors consisting of a sac lined by stratified squamous epithelium filled with laminated keratin, cholesterol crystals, and debris . The cys Ultrasound has become the primary imaging modality of intrascrotal lesions. On ultrasound, epidermoid inclusion cysts appear as well-circumscribed round or oval hypoechoic lesions with scattered echogenic reflectors and no evidence of internal blood flow on doppler. Lee et al. proposed five characteristic sonographic patterns of EIC after reviewing 24 patients with EIC in a variety of anatomical locations . They we In addition to ultrasound, magnetic resonance imaging (MRI) has become a popular tool in the evaluation of scrotal masses. On MRI, EICs are described as high intensity well defined solid masses surrounded by a low signal capsule on T2-weighted imaging . With th Although, scrotal extra-testicular lesions are an uncommon occurrence and benign in nature, the clinician should be cautious when it comes to the evaluation and management of these masses. Malignant extra-testicular lesions are uncommon but have been reported and include lymphoma, liposarcoma, fibrosarcoma or metastatic disease. The clinical debate has always been how to differentiate benign from malignant lesions. Even with the advancements of imaging modalities and characteristic features of epidermoid cysts ultrasound and MRI cannot entirely exclude the possibility of malignancy.  We share a case of an epidermoid inclusion cyst of the testicle found in an atypical location. The mass presented with benign features on physical exam and on ultrasound imaging. Because of the increasing discomfort along with the atypical location, we elected to proceed with excision of the mass. Epidermoid inclusion cysts of the testicle are very rare, benign masses, with low malignant potential. When physical and sonographic features are suggestive of epidermoid inclusion cyst, the management should be scrotal exploration with testicular sparing resection as to prevent infection or rupture complications and to avoid missing a possible malignancy.


Castleman’s disease is a rare disease characterized by lymph node hyperplasia. Its occurrence in the retroperitoneal space has rarely been reported, making its preoperative diagnosis difficult. Here, we report a case of retroperitoneal Castleman’s disease, which radiologically resembled paraspinal schwannoma.d-glucose in the tumor at a standard uptake value of 4.7, whereas no other abnormal uptake suggestive of metastatic lesion was noted. On the basis of imaging studies, we mostly suspected paraspinal schwannoma, although malignancy was not completely excluded. Angiography showed feeding vessels from the right lumbar arteries, which were embolized with porous gelatin particles in order to reduce intraoperative bleeding. Surgical resection was performed using a retroperitoneal approach, which revealed the tumor in the swollen psoas muscle. Intraoperative pathological examination of a frozen section revealed no evidence of malignancy; thus, marginal excision of the tumor was performed. The tumor adhered tightly to surrounding muscle tissues, resulting in 940 g of intraoperative blood loss. The pathological examination demonstrated infiltration of lymphocytes surrounding small germinal centers with extensive capillary proliferation. Immunostaining revealed that proliferated lymphocytes were CD3-negative and CD79a-positive.A 33-year-old Japanese man with epigastric discomfort underwent abdominal ultrasonic examination revealing a solid mass next to the right kidney. Computed tomography demonstrated a well-circumscribed mass with central calcification in the right psoas muscle. Because the mass presented a dumbbell-like shape extending to the intervertebral foramen, neurogenic tumor was suspected. Both iodine-123 metaiodobenzylguanidine and gallium-67 scintigraphies were negative in the mass, whereas thallium-201 mildly accumulated in the tumor, suggesting blood flow to the tumor. Positron emission tomography revealed accumulation of fluorine-18-2-fluoro-2-deoxy-Although a dumbbell-shaped mass in a paraspinal region is indicative of a schwannoma for orthopedic surgeons, the possibility of Castleman’s disease should be considered if a central low-signal area in fissured and a radial pattern is detected on computed tomography or magnetic resonance imaging. Appropriate preparation for massive bleeding during the treatment of Castleman’s disease, including angiography and embolization, would be helpful for performing surgical procedures safely. Castleman’s disease (CD) is a rare disease characterized by lymph node hyperplasia with polyclonal proliferation, first reported by Castleman in 1954 ,3. Althod-glucose in the tumor at a standard uptake value (SUV) of 4.7, whereas no other abnormal uptake suggestive of a metastatic lesion was demonstrated (Figure A 33-year-old man with epigastric discomfort underwent abdominal ultrasonic examination, revealing a solid mass next to the right kidney. Abdominal radiography revealed striated calcification on the right side of the first and second lumbar vertebrates Figure A,B. Compth rib (Figure Surgical resection was performed with the retroperitoneal approach from the right side by resecting the 11b Figure A. The tub Figure B. Beforeb Figure C. The cub Figure D.Pathological examination demonstrated infiltration of lymphocytes forming lymphoid follicles accompanied with capillary proliferation Figure A. CapillA recent meta-analysis of retroperitoneal tumors showed that sarcoma comprise a third of retroperitoneal tumors . A varieBecause the findings by CT and MRI did not lead to a definite preoperative diagnosis, radionuclide studies, including PET, were performed. Negativity of gallium scintigraphy suggested malignant lymphoma as a less suspected diagnosis. It is reasonable that CD could be positive for gallium scintigraphy because of its nature of lymphatic proliferative disease . HoweverHV-UCD is the most common variant of CD and accounts for 72% of all CD variants . Plasma Surgeons should be prepared for massive bleeding during the resection of CD, especially in the deep portion of the body close to major vessels. Preoperative angiography clearly visualized the feeding vessels that aided intraoperative detection and ligation of the feeding lumbar artery. If such an imaging study is not performed and the preoperative diagnosis does not include such hemorrhagic tumors, massive bleeding may obstruct surgical procedures and patients may be at risk of fatal blood loss. Although we chose the retroperitoneal approach in the decubitus position in the present case, the transperitoneal approach may have been an alternative option for tumor resection. One of the reasons for us choosing this is approach is familiarity with the retroperitoneal approach for spinal surgery. Another reason is that exploration of the intervertebral foramen may have been required because schwannoma was the most suspected preoperative diagnosis. If UCD is preoperatively suspected and the surgeons are familiar with the transperitoneal approach, the wide operative field obtained with that approach may be helpful to reduce intraoperative bleeding.The paraspinal mass that resembled a schwannoma proved to be a rare case of UCD. Careful preoperative assessment of the imaging study enables proper planning of the surgery. More importantly, central low areas in fissured and radial pattern on CT or MRI would be helpful for making the correct diagnosis of CD.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 Series Editor of this journal.CD: Castleman’s disease; UCD: Unicentric CD; MCD: Multicentric CD; HV-UCD: Hyaline-vascular type UCD; PC-UCD: Plasma cell type CD; SUV: Standard uptake value; T1W1: T1-weighted image; T2W1: T2-weighted imageThe authors declare that they have no competing interests.SN, MY, and TY participated in the surgical treatment and follow-up of the patient. HK and SK helped draft and finalize the manuscript. TH, YT, and SY performed the pathological examination. All authors read and approved the manuscript.


Objective. 1H MR spectroscopy (MRS) is widely performed for assessment of brain tumours and is considered a highly sensitive technique capable of differentiating benign from malignant conditions and tumour grading. Method. We present a case of a 69 year old woman who was suspected to have gliomatosis on MRI. Results. MRS performed using single voxel and chemical shift/multivoxel techniques was within normal limits. A repeat scan 6 months later showed progressive disease, and biopsy was performed that proved the diagnosis of glioblastoma. Conclusion. Normal MRS in a patient with suspicion of gliomatosis on MRI should not reassure clinicians into assuming a benign aetiology or a good prognosis in short term. MRS is widely performed as a research and diagnostic tool for assessment of brain tumours. Establishing a type and grade of intracranial tumour is often difficult with conventional magnetic resonance imaging (MRI), which is a principal imaging modality for its evaluation. MRS is a noninvasive method of acquiring information about metabolic activity of the brain tissue. It can be routinely obtained at the time of diagnostic MRI. There are several reports discussing the role of MRS in differentiating benign from malignant gliomas and radiation necrosis from tumour recurrence. This heavily relies on the high sensitivity of MRS in detecting metabolic abnormalities in brain tumours, particularly malignant ones. However, there is not much literature regarding negative predictive value or sensitivity of MRS in suspected brain tumours or gliomatosis cerebri.We report a case of an intra-axial lesion, which had MRI appearances consistent with a low-grade diffuse glioma or gliomatosis, but MRS findings were entirely within limits for a normal brain tissue. A few months later, the histological analysis of the lesion, following biopsy, confirmed the diagnosis of glioblastoma multiforme (GBM).A 69-year-old woman presented with a 14-month history of intermittent fornication in the distribution of the left mandibular V3) division of the trigeminal nerve. MRI scan identified a presence of an ill-defined, diffuse lesion, involving cortex and subcortical white matter of the left occipital and parietal lobes extending into the frontal lobe, with only slight mass effect and relative preservation of underlying anatomy with no postcontrast enhancement. The MRI features were suggestive of low-grade diffuse glioma or gliomatosis cerebri. After five months time follow-up MR scan with MRS (1.5 T Siemens Avanto) using 2D CSI and single voxel (SV) technique  was performed. The analysis of MRS findings was done using the standard software provided by the manufacturer. The MRI scan again confirmed the presence of extensive signal abnormality, which unchanged in size from the previous scan . The MRS divisionThe patient presented six months later with 2 weeks history of partial seizures, involving the right upper and lower limbs. Repeat cranial MRI scan revealed significant progression of the lesion into the occipital region and an increase in the mass effect. An area of enhancement in the left parietal lobe was also present, measuring 19 × 18 mm . The patEstablished MRS applications in cerebral tumours include differentiation between brain abscess and cystic tumours, low-grade and high-grade gliomas, gliomatosis cerebri, oedema and tumour infiltration, recurrent tumour, and radionecrosis and used in a stereotactic biopsy planning of brain lesions . MRS litThe most frequently assessed chemical ratios in diagnosis of intracranial tumours is Choline/Creatine (Cho/Cr), Choline/N-acetyl aspartate (Cho/NAA), and Lactate/Creatine (Lac/Cr) ratios. As compared to the normal brain tissue, tumours characteristically present a decreased NAA and increased Cho levels. NAA is normally present in neurons and neuronal injury and replacement of neurons by tumour cells cause reduction in NAA levels. Increased Cho levels are due to breakdown of membrane phospholipids caused by a rapid cell turnover in neoplastic lesions. The usual finding in neoplastic lesions is a Cho/NAA ratio greater than one . The ChoIt is widely believed that adding MRS to conventional MRI protocols improves accuracy of diagnosis of intra-axial tumours. In 2004, Lukas et al. reported the accuracy increased to 94% using long echo time (TE) and to 96% using short TE , 4. GalaDue to high sensitivity of MRS, it has also been shown to be able to differentiate benign from malignant conditions. Vuori et al. proved that loss of NAA and increase of Cho were more pronounced in low-grade gliomas than in focal cortical developmental malformations . The value of MRS in gliomatosis cerebri is more controversial. MRI findings have been reported to be nonspecific and can underestimate the extent of the lesion . The difWe have no clear explanation why MRS was normal in our patient. However, given the paucity of reported MRS findings in gliomatosis cerebri in the literature compared to other gliomas, we wonder if it can be to the extent of “within normal limits” as in our patient. Our findings raise questions about the sensitivity and negative predictive value of MRS in gliomatosis cerebri and its ability to differentiate gliomatosis cerebri from other benign cerebral conditions. We do not have histology results at the time of MRS and no MRS data at the time of biopsy, as these were 6 months apart. However, it is likely that the abnormality represented a low grade glioma that dedifferentiated into high-grade glioma a few months later. Hence, it can be reasonably assumed that the MRS data represents low-grade glioma that lasted for a while and was seen as abnormal appearance on MRI, but the MRS findings have been unhelpful in predicting how soon this may occur.While it is widely believed that MRS is highly sensitive in gliomas, the current case illustrates that MRS may show normal spectrum in a low-grade glioma or gliomatosis cerebri that can potentially change into a GBM in just a few months time. Therefore, a normal spectrum should not reassure clinicians into assuming a benign aetiology and patients should still be followed up at usual intervals with equal emphasis on other imaging and clinical findings.


The majority of gastrinomas causing Zollinger-Ellison syndrome (ZES) are located in the duodenum or the pancreas. Primary hepatic gastrinomas (PHG) are extremely rare and difficult to diagnose because the liver is the commonest site of metastatic disease and gastrinomas can be very small. Furthermore, gastrinomas are typically slow-growing thus a missed, occult primary tumour may not become evident for many years. The diagnosis of PHG is therefore dependent on a careful search for a primary and long-term biochemical follow-up following curative hepatic resection. We report a case of a 7 cm PHG in a 48 year old man with ZES. Preoperatively, both a basal and stimulated gastrin levels were elevated. Surgical exploration including intraoperative ultrasound and duodenotomy, failed to reveal a primary. Patient underwent a right hepatectomy. Yearly, gastrin and secretin stimulation tests remain normal 6 years following surgery. He remains symptom free off all medication. An additional 26 cases of PHG were found. Including this case, 21 had at least 1 year follow-up, however only eight had greater than 5 years (median 24 months). Post-op gastrin levels were reported in 25, however provocative testing was done in only 10. Persistence and recurrence occurred in one and four, respectively. PHG causing ZES is extremely rare. Although the current literature claims to include 26 additional cases of PHG, without a thorough search for the primary and long-term follow-up data including provocative testing, this diagnosis remains a challenge. Gastrinomas account for 20% of functioning neuroendocrine tumors (NET) of the pancreas . ExcessiPrimary gastrinomas are typically located in the duodenum or pancreas. More than 90% of primary tumors are found in the so-called “gastrinoma triangle” an area bounded by the neck of the pancreas, the confluence of the cystic and common bile ducts and the junction of the second and third portions of the duodenum . A smallAs is the case with primary lymph node gastrinoma, primary hepatic gastrinomas (PHG) are extremely rare and difficult to diagnose because the liver is also a common site of metastatic disease. To date only 26 cases of apparently PHG have been reported in the English language literature  3,4,6,7,25,26,27In this paper, we report a case of primary hepatic gastrinoma that underwent curative hepatic resection with no evidence of clinical, biochemical or radiological recurrence at the 6th year follow-up. We carefully scrutinized the work-up, treatment and follow-up of the additional 26 cases reported in the literature and challenge the diagnosis of PHG in some of these.A 48-year-old male presented with a 2-year history of intermittent diarrhea and vague abdominal pain. The patient described a long-standing problem with significant heartburn and reflux. His symptoms had become more frequent and severe in the few months prior to presentation. He described a weight loss of approximately 10 kilograms over the same time period. Physical exam reveal no abnormality. A colonoscopy, barium enema and small bowel follow-through all were negative. He underwent an upper GI endoscopy, which demonstrated significant gastritis and multiple duodenal ulcers. His fasting serum gastrin (FSG) level after stopping all anti-secretory medications was moderately elevated at 288 pg/mL  and Chromogranin A level was also elevated at 340 ng/mL . Both enhanced computerized tomography (CT) and magnetic resonate imaging (MRI) scans demonstrated a solitary 7.0 × 5.7 cm lesion within the right lobe of the liver .An image-guided core biopsy was consistent with a neuroendocrine tumour. Whole body octreotide scan and SPECT showed a solitary liver mass with no evidence of disease outside the liver .At laparotomy, the pancreas was fully mobilized to allow for careful palpation and intra-operative ultrasound (IOUS) assessment. A duodenotomy was performed and the duodenal mucosa was carefully palpated from the pylorus to its fourth portion. A regional lymph node dissection failed to identify any primary or regional disease. An extended right hepatic lobectomy and cholecystectomy was performed. The patient’s post-operative course was unremarkable and he was discharge on day seven. Pathology revealed a 6.5 × 6.0 × 5.0 cm neuroendocrine tumour that stain positive for gastrin, Chromogranin A and synaptophysin. All of the removed lymph nodes were negative for tumour.Three months post-operatively, a FSG level, provocative secretin-stimulation test and a serum Chromgranin A were all within normal limits. Despite this, to the patient was closely followed with the potential expectation of an occult primary tumour becoming evident as time progressed. Six years following his hepatic resection the patient remains symptom free and off all medication. Both basal fasting gastrin and secretin-stimulation tests remain normal, as does his cross-sectional imaging .The diagnosis of ZES is based on the clinical endocrinopathy and confirmed by an elevated FSG level in the presence of raised gastric acid secretion. When the results of FSG and acid secretory studies are borderline or non-diagnostic, provocative testing with either intravenous secretin (2 mg/kg) or calcium or both is indicated . Our patApproximately 90% of gastrinomas are located in the Gastrinoma Triangle mainly in the duodenum or pancreas. Pancreatic gastrinomas tend to be relatively large, with a mean reported diameter of between 2.7 and 3.2 cm . In contet al. noted that the rate of biochemical cure was significantly higher when a duodentomy was performed compared to those that did not (90% vs. 50%) [et al. described the use of intra-operative gastrin measurement to assess completeness of resection following exploration on 20 patients with gastrinomas [Ultimately, preoperative studies will fail to localize the tumor in a significant percentage of patients, leaving the surgical exploration to be the localizing procedure. The issue of what comprises an adequate operative exploration for sporadic gastrinoma has been a subject of some debate in the literature. As the vast majority of tumours are known to be located in the duodenum and pancreas, a careful examination of these sites is mandatory. Examination of the pancreas is facilitated by mobilization of the pancreatic head (via the Kocher maneuver), body and tail to allow for bimanual palpation. The use of intraoperative ultrasound has been shown to improve the ability to locate these tumors . The duovs. 50%) . Proye etrinomas . This teOur patient underwent a thorough operative exploration including mobilization of the pancreas and duodenum, bimanual palpation, intra-operative ultrasound and a duodenotomy. Of the additional 26 cases reported in the literature, 21 described an intra-operative approach to attempt localization of a primary within the gastrinoma triangle involving various combinations of intra-operative ultrasound, palpation, lymph node biopsies and endoscopic duodenal trans-illumination . No caseGastrinoma is typically a slow-growing tumour. Consequently; a missed, occult primary tumour may not become evident for many years after surgical exploration. Given the rarity of PHG insight into the natural history must be taken from a similar clinical scenario, that of potential primary lymph node gastrinomas. In this literature, a potential lymph node primary tumour is defined when no additional site of tumour can be found at exploration and the operation results in resolution of clinical symptoms and normalization of biochemical indices. In a series of apparently primary lymph node gastrinoma, eight of the 26 presumed primary lymph node gastrinomas showed biochemical or clinical evidence of recurrence . Four ofet al. found that combination of both these tests at 2 years following surgical resection was 100% predictive disease-free status at 3 years [Twenty-three of the 26 previously reported cases had initial clinical and biochemical evidence of cure. Median follow-up in these cases was 24 months (range 0–240). Only seven cases out of 26 reported follow-up of more than 5 years ,18,20,27 3 years . ProvocaCommonly reported cure rates for gastrinoma are around 26% for sporadic disease and typically much lower in gastrinomas associated with MEN 1 . It is lPrimary hepatic gastrinoma causing ZES is extremely rare, yet our case illustrates that it does occur. A firm diagnosis of this rare clinical finding is complicated by the commonality of small occult duodenal primary tumors and their slow growing nature. Strong support for this rare diagnosis requires: (1) clear clinical and biochemical evidence of ZES; (2) an appropriate pre and intra-operative search for an occult primary tumor, especially the duodenum; and (3) long-term clinical, biochemical and radiologic follow-up. Although the current literature claims to include an additional 26 cases of PHG, without a thorough search for the primary and long-term follow-up data including provocative testing, we challenge several of the cases claiming such a diagnosis.


Background. Small bowel ischemia due to superior mesenteric vein thrombosis (MVT) is rare during pregnancy. However, additional precipitating factors should usually be identified. Case. A 31-year-old woman, pregnant at 34 weeks, was sent to the emergency department because of acute peritonitis. An emergency exploration revealed a segmental gangrene of the small intestine without any mechanical obstruction. Together with the termination of pregnancy, resection of the damaged small bowel was performed, and an end-to-end enterostomy was followed. Based on the operative and pathological findings, small bowel ischemia might be attributed to superior mesenteric vein thrombosis. Conclusion. Hypercoagulation state normally found in pregnant women is believed to lead to this catastrophic condition without other precipitating factors. Small bowel ischemia is a relatively uncommon disorder; the primary causes are diverse and can be grouped into five major categories: (1) strangulation, (2) low-flow states , (3) embolus or thrombosis of superior mesenteric artery, (4) superior mesenteric vein thrombosis (MVT), and (5) miscellaneous. Due to vague clinical presentation and the lack of specific diagnostic tests, early diagnosis for intestinal ischemia is difficult, resulting in significant morbidity and mortality. We herein report a case of a 31-year-old woman who was presented with the signs and symptoms of peritonitis at 34-week gestation. Surgical exploration revealed gangrene in the small intestine without mechanical obstruction. Hypercoagulable state is normally found in pregnant women, which was believed to result in superior MVT and then intestinal ischemia. To the best of our knowledge, this is the second report to describe such a rare disease developing during pregnancy, without evidence of other precipitating factors.At 34 weeks of gestation, a 31-year-old gravida 3 para 1 woman was referred to our emergency department from a local clinic owing to progressive epigastric pain associated with nausea and vomiting for 3 days. She had no significant medical or surgical history and no history suggestive of thromboembolism. She had never used any oral contraceptives or other hormonal preparations. Upon arrival, her vital signs were stable without fever . The hematologic examination revealed marked leukocytosis  and relative intravascular depletion (hematocrit 44.5%). Coagulation profile and biological tests were within normal limits. Obstetric ultrasound showed a normal male fetus compatible with his gestational age. In addition, a mild fatty change in the liver and a moderate amount of ascites were also noted. The fetal monitor showed that the uterus contracts every 10 minutes. She was initially kept for conservative treatment  and absolute bed rest under the suspicion of acute gastroenteritis and preterm labor. However, signs of acute peritonitis gradually developed within 6 hours after admission. Thus, an emergency exploration was performed to find a segmental gangrene of the small intestine with 1,500 mL of serosanguineous peritoneal fluid, but no frank obstruction and perforation was identified . The preSmall bowel ischemia occurs very infrequently during pregnancy or puerperium. After searching the MEDLINE, only 16 cases of small bowel ischemia are reported to demonstrate the association of its occurrence with pregnancy to date. Among them, 5 cases (31.2%) were caused by the volvulus of the bowel , and theEarly diagnosis of superior MVT is very difficult due to the nonspecific signs, symptoms, and laboratory results. Almost all patients present with abdominal pain, nausea and vomiting, and leukocytosis with elevated hematocrit, but these are not helpful for diagnosis . Today, Once the diagnosis of superior MVT is made, the immediate use of anticoagulation is indicated due to the high recurrence rate. In patients with inherited hypercoagulable disorders , life-long anticoagulation is warranted. For patients with reversible predisposing causes, at least 6 months of anticoagulation is recommended . Though In conclusion, it is suggested that in a pregnant woman with a clinical presentation of acute abdomen, even with the absence of the other known risk factors, superior MVT should be included in the differential diagnosis because the pregnancy itself should be considered as an important background etiological factor.


Schwannomas are benign encapsulated tumors arising from Schwann cells in the peripheral nervous system. Between 25 and 45% of schwannomas occur in the head and neck region. Schwannomas of the larynx are extremely rare with an incidence of 0.1–1.5% in all benign laryngeal tumors. Laryngeal schwannomas usually occur in females aged between their 4th and 5th decades. The most successful curative method is surgical resection. When selecting the surgical method, the size and location of the tumor, as well as the requests of the patient, should be considered. The present case report describes a 29-year-old female patient with symptoms of hoarseness and dyspnea on exertion. Through endoscopic biopsy, histopathology revealed a schwannoma. Considering the symptoms, age and preferences of the patient, the method of trans-oral microlaryngoscopic excision without tracheotomy was used to excise the tumor located in the aryepiglottic fold. Results from a short-term follow-up showed the postoperative result to be satisfactory. Schwannomas are solitary, encapsulated, slow-growing, benign tumors arising from Schwann cells of the peripheral nervous system . BetweenA 29-year-old female consulted the Department of Otolaryngology, Head and Neck Surgery, Shanghai Jiao Tong University Affiliated Shanghai First People’s Hospital  with symptoms of hoarseness and dyspnea on exertion that had been present for 3 years. The patient had also experienced dysphagia for 1 month, which had recently worsened. In a previous hospital, 2 months prior to consultation, the tumor had been biopsied and the pathodiagnosis revealed a schwannoma. No other significant past medical illnesses were present and systemic examination was normal. No palpable lymph nodes or café au lait patches were present. Informed consent was obtained from the patient.Outpatient laryngoscopy revealed a large mass located within the left recessus piriformis. In addition, the glottis was obstructed and the vocal folds were not visible . MagnetiFollowing consideration of age and symptoms, the patient selected not to undergo surgery via an incision in the lateral neck. Therefore, a trans-oral resection using a microlaryngoscope was performed and postoperative tracheostomy was avoided. Treatment for the symptoms and nutritional support was administered, including anti-inflammatory agents, hemostatic methods and nasogastric nutrition. Microscopic examination showed compact cellular margins and prominent palisade formation, known as Antoni A patterns. A less cellular, loosely-textured pattern, known as the Antoni B pattern, was also observed. Immunochemical staining evaluation demonstrated the expression of S-100, resulting in a final diagnosis of a benign schwannoma .The patient was discharged on the fifth postoperative day and the nasal feeding tube was removed. Postoperative laryngoscopy showed that the left vocal cord was immobile and that the remaining tumor was present within the left recessus piriformis . It is uSchwannomas and neurofibromas are two types of neurogenic tumor. Neurofibroma is the main differential diagnosis of laryngeal schwannoma. Other differential diagnoses include chondroma, adenoma, laryngeal cyst and laryngocele . SchwannSchwannomas most commonly occur in females between the ages of their 4th and 5th decades. In total, 80% of laryngeal schwannomas are found in the aryepiglottic folds, while the remaining 20% are found in the false and true cords . In the Clinical symptoms are related to the size and location of the tumor . The patLaryngoscopic evaluation reveals a round submucosal mass originating from the aryepiglottic fold and/or true and/or false vocal cords.With CT scans, a small schwannoma is regarded as a homogenous, enhanced mass . When thWhen examined by MRI, T1-weighted images of schwannomas have a low signal intensity ranging between those of the brain and muscles, with a homogeneous or heterogeneous appearance . With T2Although CT and MRI scans reveal a well-defined submucosal mass without surrounding tissue destruction , the metet al (Laryngeal schwannoma is a rare benign tumor and the curative method is surgical resection . Howeveret al  demonstret al . The comet al . For the


A case of massive upper gastrointestinal bleeding in a 37-year-old female is presented showing a submucosal mass in the gastric body. At laparotomy a pedunculated submucosal mass was found located on the posterior wall at the junction of the body and antrum of the stomach, 8 cm from the pylorus. Pathology confirmed that it was a 4 cm benign gastric lipoma with a bleeding central ulcer. Gastric lipomas are rare, benign, typically submucosal tumors occurring in the gastric antrum. They are usually asymptomatic but can become symptomatic depending on size, location, and if there is ulceration of the lesion. These lesions may be mistaken as malignant tumors or present with upper GI bleeding or intussusception. The diagnosis can be made using a combination of upper endoscopy, endoscopic ultrasound, CT, and MRI with surgical excision being the definitive treatment of choice. We hope that this case highlights the fact that these lesions can present with massive upper GI haemorrhage and should be included in the diagnosis when appropriate. Gastric lipomas are rare and account for less than 1% of all tumors of the stomach and 5% of all gastrointestinal lipomas . They tyA 37-year-old female presented with a four-day history of epigastric pain and melaena associated with vomiting. The pain was sudden in onset, severe, and with no radiation. There were no aggravating factors and it was associated with three episodes of black, tarry, and foul-smelling stool. She had a history of weakness, dyspnoea, headaches, and palpitations (symptoms of anaemia) and used Ibuprofen for menstrual cramps on a monthly basis. There was no history of cigarette smoking, alcohol use, peptic ulcer disease, or reflux. Examination revealed pale mucous membranes and epigastric tenderness. The haemoglobin dropped to 5.9 g/dL and she was transfused 6 units of packed cells.An upper GI endoscopy revealed a normal oesophagus, cardia, and fundus with a submucosal mass with a 1 cm ulcerating area in the gastric body. The patient was prepared for a laparotomy and an anterior gastrotomy was performed on the basis of the current diagnosis of a stromal tumor. A pedunculated submucosal mass was found, located on the posterior wall at the junction of the body and antrum of the stomach, 8 cm from pylorus . PatholoGastrointestinal (GI) lipomas are benign tumors composed of mature adipose tissue covered with a fibrous capsule. Most GI lipomas are located in the colon, ileum, and jejunum and are predominantly asymptomatic. Gastric lipomas consist of less than 1 percent of all benign gastric tumors and 5 percent of all GI lipomas . They arMost lipomas are found in the submucosa (95%), the subserosal subtype being extremely rare. They are usually solitary and most commonly found in the antrum, with an incidence of 75% . AlthougUpper GI endoscopy will show a submucosal mass and three signs which help diagnosis . These aComputed tomography has been shown to be valuable for diagnosis, demonstrating a well-circumscribed, submucosal mass with uniform fat density and attenuation ranging between −70 and −120 Hz. CT scanning of large (>2 cm) submucosal gastric masses detected on endoscopy can obviate the need for biopsy. Magnetic resonance imaging may show high signal intensity on T1 weighted sequences typical of a lipoma. Endoscopic ultrasound is also quite useful and may show a hyperechoic and isodense mass as opposed to a fibrolipoma which is hyperechoic and heterogenous .The histology in this particular case showed a tumor composed of mature adipocyte proliferation, showing significant variation in cell size, associated with some lipoblasts. Some nuclei were large, slightly irregular but without hyperchromasia or mitosis. Grossly, a lipoma with ulceration, haemorrhage, acute, and chronic inflammation was seen . The diaSignificant complications of a symptomatic gastric lipoma include gastrointestinal obstruction, gastroduodenal intussusception, and severe massive GI haemorrhage. Very rarely, these tumors can become malignant, with a handful of cases being reported in the literature. Histologically, there are four types of liposarcomas including well-differentiated, myxoid, round cell, and pleomorphic. Well-differentiated liposarcomas account for 40% of all liposarcomas, having a peak incidence between the 5th and 7th decades and are further subdivided into adipocytic, sclerosing, inflammatory, and spindle cell subtypes .In conclusion gastric lipomas are benign, typically submucosal tumors occurring in the gastric antrum. They are usually asymptomatic but can become symptomatic depending on size, location, and if there is ulceration of the lesion. These lesions may be mistaken as malignant tumors or present with upper GI bleeding or intussusception. The diagnosis can be made using a combination of upper endoscopy, endoscopic ultrasound, CT, and MRI with surgical excision being the definitive treatment of choice. We hope that this case highlights the fact that these lesions can present with massive upper GI haemorrhage and should be included in the diagnosis when appropriate.


We will describe a case of a patient diagnosed with a rare identity of a benign lesion, “reactive nodular fibrous pseudotumor” (RNFP). It is a tumor which preoperatively can present as a malignant tumor and is only reported in 19 cases. According to the very limited amount of information on this tumor in the literature it is mostly seen after trauma or intraperitoneal inflammation. Our case is the second one of RNFP associated with endometriosis, which is a frequently seen intraperitoneal inflammation process in women. Knowledge that these large pseudotumoral lesions can occur is important to direct the management of these patients. A multitude of tumors can occur in the peritoneal cavity. Correct diagnosis is of paramount importance for proper treatment. We recently observed a patient with an intra-abdominal mass, diagnosed histopathologically as “reactive nodular fibrous pseudotumor” (RNFP). This is a benign lesion, often mimicking a malignant tumor preoperatively. The pathogenesis might be related to intraperitoneal “trauma” such as endometriosis. It is previously reported in only 19 cases.A 45-year-old woman was hospitalized in another hospital, because of intractable menometrorrhagia, pain, and gradual abdominal swelling. A vaginal hysterectomy was planned and performed but shortly had to be stopped because of bleeding. During the procedure intraperitoneal lesions were seen and biopsied. Pathology shows no signs of malignancy. Afterwards the patient was sent to our hospital for further diagnosis. Her medical history mentioned no other abdominal surgery, migraine headaches, or other major incidents. Her obstetrical history recorded an uncomplicated vaginal delivery. Clinical examination showed no particularities. Biochemically we found no abnormalities and serum CA125 levels were normal. Gynecological ultrasound  demonstrBecause of the age of the patient and because a malignant disease could not be excluded, it was decided to perform a total abdominal hysterectomy with bilateral salpingo-oophorectomy. Laparotomic peritoneal exploration showed normal female genital organs, but multiple fibrous plaques and nodules on the sigmoid. A sigmoid resection with end-to-end anastomosis was performed as the tumor was widespread on the sigmoid involving the vasculature. Frozen section analysis reported a benign lesion with low cellularity, chronic inflammation, and no atypia. Postoperative recovery was uneventful and our patient could leave the hospital within a week. Until now there are no signs of recurrence.The surgical specimen consisted of the uterus with both adnexa, a sigmoid segment, and the omentum. Gross examination revealed white, hard nodules with a smooth surface on the omentum, sigmoid, and right ovary. The largest nodule measured 7 × 6.3 × 3 cm and was located on the sigmoid. Histologically, there were multiple fibrocollagenous nodules on the omentum, pelvic wall, right ovary, sigmoid, and Douglas pouch . These nPathologically, the resected lesions are consistent with the so-called “reactive nodular fibrous pseudotumor” (RNFP), first reported by Yantiss et al.  in 2003.Since 2003, a total of 19 cases of this entity have been recognized. It is considered as a nonneoplastic (myo)fibroblastic proliferation, representing an inappropriate postinflammatory response to injury. All kinds of injury have been proposed: abdominal surgery (5x), peptic ulcers (2x), foreign body ingestion (2x), perforated duodenal diverticulitis, endometriosis in combination with the use of ergotamine, and chronic bowel obstruction. In our case we can only recognize endometriosis as a risk factor.All previously reported cases are from developed countries . Most patients are adults above 18 years; one case was a 1-year-old child. There is a clear male-to-female preponderance of 14 to 5. Cases with both solitary (11) and multiple (8) masses have been described [The fact that RNFP often presents with multiple intra-abdominal masses evidently causes clinical concern for malignancy . RNFP haAnother neoplastic entity to consider in the differential diagnosis is a GIST, which commonly has a more brown, fleshy, more variable gross appearance with hemorrhage and necrosis. Histologically these tumors show a more organoid architecture with cellular fascicles of plump spindle cells with eosinophilic cytoplasm separated by delicate fibrous septa and a hyalinized or edematous stroma –6.In the differential diagnosis of RNFP, a number of nonneoplastic lesions have to be considered, for example, the calcified fibrous pseudotumor which shares many features with RNFP. Like RNFP, it frequently occurs intra-abdominally and is a benign soft tissue lesion consisting of a hypocellular spindle cell proliferation within dense collagen. It is accompanied by dystrophic calcifications but is more cellular and contains a mixed inflammatory infiltrate . SclerosThis is the second case of RNFP with associated endometriosis . The oth


Desmoid tumors (DTs) are rare lesions that do not possess any metastatic potential. However, they have a strong tendency to invade locally and recur. They constitute 3% of all soft tissue tumors and 0.03% of all neoplasms. Abdominal DTs occur sporadically or are associated with certain familial syndromes, such as familial adenomatous polyposis (FAP). The single form of this neoplasm most frequently occurs in females of reproductive age and during pregnancy. A female patient with a DT of the abdominal wall who had no relevant family history was admitted to hospital. The patient, who presented with a painless mass in the left anterolateral abdomen, had no history of trauma, surgery or childbearing. According to the medical history, physical examination and CT report, the patient was diagnosed with DT. Radical resection of the affected abdominal wall musculature was performed, and the defect was replaced with a polypropylene mesh. The histological diagnosis was of DT. The patient remains in good health and complete remission without any other treatment following surgery. DTs exhibit aggressive growth and have a high rate of recurrence. Surgery is the optimal treatment, and subsequent radiotherapy may decrease the local recurrence rate. Further research into their aetiology is required combined with multicentre clinical trials of new treatments in order to improve management of this disease. This case report provides general knowledge of DT, and may be used as a guidance for diagnosis and treatment. Desmoid tumors (DTs), also known as aggressive fibromatoses, are benign myofibroblastic neoplasms originating from muscular aponeuroses that are also classified as deep fibromatoses . They coThis report presents the case of this rare fibromatosis in a 17-year-old female who had no history of trauma, abdominal surgery or childbearing. The appearance of the tumor was analyzed using computed tomography (CT). Informed consent was obtained from the patient prior to the study.A 17-year-old female was admitted to the Department of General Surgery, The Second Affiliated Hospital of Dalian Medical University  with a painless mass in the left anterolateral abdomen. During a physical examination, the mass was observed to be firm, lacking tenderness and fixed to the abdominal wall. The patient stated that the mass was gradually increasing in size. The patient had no relevant family history and no history of smoking, drinking alcohol or taking any medication. The analyzed blood parameters were within the normal range and the tumor marker results were negative. The patient had no history of trauma, surgery or childbearing.Pre-operative CT scans revealed a large mass (9.3×6.1 cm) with unclear borders of attenuation equal to that of muscle. The mass originated from the left rectus abdominis muscle and, following intravenous administration of contrast medium, demonstrated mild enhancement, even in the delayed images .Radical resection of the affected abdominal wall musculature was performed down to the peritoneum, and including a peripheral margin of 3 cm of healthy tissue. Following the resection of the DT, the abdominal wall defect was replaced with a Bard Composix EX Mesh . Macroscopically, the tumor had a firm texture. On the cut surface, it was pale and certain areas had the appearance of fish meat. The tumor has no confirmed capsule, and its margin was ill-defined . The hisDTs are benign deep fibromatoses that originate from fascia and muscular aponeuroses, with an infiltrating growth pattern . DTs areSeveral modern imaging methods, including abdominal ultrasonography, CT and magnetic resonance imaging (MRI), are used for the diagnosis of DTs . In ultrThe treatment of neoplasms, such as DTs, is guided by their clinical and evolutive characteristics. Radical therapy consists of wide tumor and adjoining tissue resections . SurgeryIn conclusion, the optimal treatment for DTs remains unclear. Surgery is the primary treatment option, although it carries a risk of local recurrence. A radical resection with clear margins remains the principal determinant of outcome. The repair of abdominal wall defects may be achieved with prosthetic mesh reconstruction, which leads to good functional results. Non-surgical treatments result in diverse and unpredictable outcomes, but are considered as an option for adjuvant therapy in patients with unresectable lesions.


Mature teratoma is the most common primary germ cell tumor in the mediastinum. On rare occasions, cystic teratomas rupture in adjacent structures, such as pleural space, pericardium, lung or tracheobronchial tree. We present a case of a mediastinal mature cystic teratoma in 16-year-old female with complex rupture into the lung, bronchus and skin. Mature mediastinal teratoma fistulized to the skin has not been previously described. Mature teratoma is the most common primary germ cell tumor in the mediastinum . It is cThese tumors rarely rupture into the adjacent structures, such as the pleural space, pericardium, lung parenchyma or tracheal tree. We report a case of mature mediastinal teratoma (MMT) with complex rupture into the lung, bronchus and skin with cysto-cutaneous fistula, which produced sebum and hair. To our knowledge, this observation has not previously been described.A 16-year-old female patient was admitted to the hospital with recurrent hemoptysis accompanied by chest pain for 5 months, and recent emergence of a left parasternal cutaneous fistula. She also complained of mild dyspnea on exertion and subfebrile body temperature.On admission, physical examination was normal except for the presence of a cutaneous fistula Figure  at the lA simple thoracic radiograph showed a left anterior mediastinal mass Figure . ContrasMMT with complex rupture into the lung, bronchus and skin was suspected. Surgical treatment performed by a left posterolateral thoracotomy, consisted of complete tumor resection combined with resection of the lingula and flattening of the fistulous tract to the chest wall. The tumor was difficult to resect, and adhesions were noted. The tumor was removed with extreme care to preserve vital structures such as the phrenic nerve, recurrent laryngeal nerve, aorta, pulmonary and subclavian vessels.The tumor contained yellowish sebaceous material as well as some hair and fat Figure . MicroscThe postoperative period was uneventful and the patient was discharged on the sixth day after surgery. She remained well at 2-year follow up.Mediastinal teratomas are asymptomatic in up to 53% of cases and are frequently discovered incidentally on chest radiography performed for other reasons . PerforaThe etiology of perforation of mediastinal teratomas is still controversial, although ischemia, infection and inflammation have been proposed . Tumor set al. [et al. [CT is the best modality to demonstrate tumor morphology and its complications. According to Choi et al. , in case [et al.  report tRupture of a mediastinal teratoma in an adjacent organ, including the lung, bronchus, pleura, and pericardium is classic, and is often reported as a clinical case. To our knowledge, complex rupture of a mediastinal teratoma simultaneously in the lung, bronchi and skin has never been described, this is the first case reported in the literature. In this case, the existence of a cutaneous fistula with flow of sebum containing hair, is like trichoptysis, pathognomonic of ruptured mediastinal teratoma.Prompt treatment of mediastinal teratoma is necessary to confirm the diagnosis by eliminating the presence of an immature component, and avoid complication due to its rupture causing hemoptysis, acute respiratory distress or cardiac tamponade . SurgeryIn mature mediastinal cystic teratoma, even if the fistula to the skin and lung is an unusual and anecdotal complication, reflecting its long history and its late diagnosis, this is further evidence of the benefits of performing surgery without delay, and confirming the diagnosis by eliminating the presence of immature or malignant components, and thus avoiding complications.Written informed consent was obtained from the patient’s parent for the study and publication of this case report and accompanying images. A copy of written consent is available for review from the Editor-in-Chief.CT: Computed tomography; MMT: Mature mediastinal teratoma.The authors declare that they have no competing interests.SM collected information and prepared the original draft. LM and GJ researched the relevant literature and revised the draft. OY helped with the literature research and preparing the manuscript. SM helped prepare the manuscript. All authors read and approved the final manuscript.


We report a rare male case of an undifferentiated carcinoma with osteoclast-like giant cells originating in an indeterminate mucin-producing cystic neoplasm of the pancreas. A 59-year-old Japanese man with diabetes visited our hospital, complaining of fullness in the upper abdomen. A laboratory analysis revealed anemia  and elevated C-reactive protein (3.01 mg/dl). Carbohydrate antigen 19-9 was 274 U/ml and Carcinoembryonic antigen was 29.6 ng/ml. A computed tomography scan of the abdomen revealed a 14-cm cystic mass in the upper left quadrant of the abdomen that appeared to originate from the pancreatic tail. The patient underwent distal pancreatectomy/splenectomy/total gastrectomy/cholecystectomy. The mass consisted of a multilocular cystic lesion. Microscopically, the cyst was lined by cuboidal or columnar epithelium, including mucinous epithelium. Sarcomatous mononuclear cells and multinucleated osteoclast-like giant cells were found in the stroma. Ovarian-type stroma was not seen. We made a diagnosis of osteoclast-like giant cell tumor originating in an indeterminate mucin-producing cystic neoplasm of the pancreas. All surgical margins were negative, however, two peripancreatic lymph nodes were positive. The patient recovered uneventfully. Two months after the operation, multiple metastases occurred in the liver. He died 4 months after the operation. Undifferentiated carcinoma (UC) with osteoclast-like giant cells (OGCs) is rare neoplasm of the pancreas. The tumor was first described by Rosai in 1968 , there hA 59 year-old man presented at our hospital with a complaint of fullness in the upper abdomen. A physical examination showed a palpable mass in the upper left abdomen. Laboratory tests showed anemia and inflammatory reactivity, hemoglobin (Hgb) was 9.7 g/dl and C-reactive protein (CRP) was 3.01 mg/dl. Carbohydrate antigen 19-9 (CA19-9) was 274 U/ml and carcinoembryonic antigen (CEA) was 29.6 ng/ml. A computed tomography scan revealed a large cystic mass in the upper left quadrant of the abdomen that appeared to originate from the pancreatic tail Figure . The cysSince Posen et al. reported the first case of an UC with OGCs of the pancreas associated with a mucus-secreting cystadenocarcinoma in 1981 , there hThe reports described 2 men and 10 women ranging in age from 25 to 77 years with a median age of 47 years, suggesting that this type of tumor tends to develop in middle age and predominantly in females. That spectrum was compatible with that of ordinary MCN. Patients showed symptoms such as abdominal pain or discomfort, anemia, and weight loss. The tumor arose from the head of the pancreas in 2, body in 1, tail in 6, and body and tail in 3 patients. The lesions were resected in all of the patients. The average tumor size was 12.5 cm at the largest diameter, ranging from 5 to 20 cm. Lymph node metastasis was seen in two cases. Invasion to another organ was seen only in our case, in which the tumor invaded to the stomach. With the exception of the two male cases, the patients had experienced favorable courses of their disease and were alive when papers were published. An ovarian-type stroma was seen in 6 cases, and 5 cases did not mention it. Our case did not display an ovarian-type stroma.Although some authors have stated that UC with OGCs of the pancreas is apt to present as a large mass with a slow metastatic spread and a much better prognosis than ordinary carcinoma ,15, the Zamboni et al. reported that 14% of MCNs of the pancreas did not demonstrate an ovarian-type stroma and that these tumors had a high tendency to invade compared to the tumors with ovarian-type stroma . FurtherUC with OGCs is a rare neoplasm of the pancreas. In most cases, UCs with OGCs originate in ductal adenocarcinoma, classified as a subtype of undifferentiated carcinoma in the WHO classification , and areIn conclusion, we have reported a male case of UC with OGCs originating in an indeterminate mucin-producing cystic neoplasm of the pancreas. Because the number of cases is too small to arrive at definitive conclusions, more studies are needed to establish the treatment strategy for this tumor.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.UC: undifferentiated carcinoma; OGC: Osteoclast-like giant cell; MCN: Mucinous cystic neoplasms; Hgb: Hemoglobin; CRP: C-reactive protein; CA19-9: Carcinoembryonic antigen; PLC: Pleomorphic large cell; PAS: Periodic acid-Schiff stain; IPMN: Intra- ductal papillary-mucinous neoplasms.The authors declare that they have no competing interests.TW and OI wrote the manuscript. OI have operated this case. TW, GO, NC, HI and YK did the assistant of the operation. WD diagnosed the pathology of this case. YK reviewed the manuscript. All authors read and approved the final manuscript.


Background. Development of neuroepithelial tumors from mature cystic teratoma is very rare. We present a case of oligodendroglioma developing inside mature cystic teratoma. Case. Eighteen-years-old female, right adnexal mass with solid and cystic areas was detected. Sections showed all three germ layers. Also, a tumoral lesion was observed in a glial fibrillary matrix. Tumor was composed of monotonous, uniform cells which have oval-round nucleus, perinuclear halo, and indistinct cytoplasm. GFAP, EGFR, P53 were positive. Conclusions. We diagnosed oligodendroglioma arising from mature cystic teratoma. There was no recurrence at the end of 13 months followup. The number of cases which have been reported in the literature is only a few. Mature cystic teratoma is the most common neoplasia of the ovary and it originates from all three germ layers . Ovarian teratomas account for 25% of all ovarian tumors . MalignaAn eighteen-year-old female was admitted to our hospital with abdominal pain. On physical examination, on the right side of the abdomen a palpable mass was detected. Ultrasonography showed right adnexal mass with solid and cystic areas inside. After laparotomy and oophorectomy, pathological examination was performed.Oophorectomy material had smooth surface and consisted of solid and cystic areas. On the cut surface mature adipose tissue, bone, cartilage, and hair and, in an area of about 6 cm soft, gray-pink, solid-microcystic lesion were observed.In many sections tissues belonging to all three germ layers  were seen Figures  and 2.Also, an area of about 6 cm tumoral lesion was observed in a glial fibrillary matrix. Tumor was composed of monotonous, uniform cells which have oval-round nucleus, perinuclear halo, and indistinct cytoplasm Figures , and 3.In this tumoral area, GFAP , EGFR, aSquamous cell carcinomas account for 80% of the malignancies arising in mature cystic teratomas . Apart fOligodendrogliomas are malignant tumors originating from oligodendrocytes and arising in the cerebral hemispheres in the young and middle ages. The macroscopic examination shows a soft, shiny, gray-pink colored, solid mass with focal cystic and calcific degenerations. The microscopic examination shows a monotonous population of round, uniform cells with a hyperchromatic nucleus and perinuclear halo. The perinuclear halo observed with H&E staining is a fixation artifact and represents a fried-egg appearance typical of oligodendrogliomas. In addition, anastomosis of fine capillary network is termed as a “chicken wire” appearance. Calcification is more frequent in such tumors.Immunohistochemistry has a limited place in the diagnosis of oligodendrogliomas, and the diagnosis is essentially based on the histomorphological findings and the exclusion of other diagnoses. GFAP, NF protein, S100, and Leu-7 can be useful in the diagnostic process. Dot-like EMA positivity and GFAP positivity particularly in the perivascular zone observed in ependymomas are helpful markers in differentiating oligodendrogliomas from clear cell ependymomas. In addition, synaptophysin immunoreactivity is a typical finding for central neurocytomas, which are also clear cell-type tumors.Surgical resection is a curative treatment in mature cystic teratomas. In the reported cases until today, surgery achieved up to four years of disease-free survival in patients with oligodendrogliomas arising in the teratoma. The current case remained recurrence-free at the end of 13 months of followup. However, long-term follow-up results are required to structure a treatment approach. Furthermore, the possibility of various malignancies in the teratoma should be kept in mind, and multiple samplings should be performed from different sites.