Gastric submucosal lipoma is an uncommon finding in the stomach. A benign fatty tumor that is asymptomatic majority of the time. However, symptomatic and bigger tumors can be difficult to manage surgically and require detailed surgical planning prior to proceeding with bariatric surgery.58 year old female who presented for weight loss consultation. Underwent an esophagogastroduodenoscopy (EGD) that demonstrated a 3 cm mass near the incisura. Endoscopic ultrasound (EUS) and biopsy demonstrated a submucosal lipoma. Due to the lipoma's location, resection was critical, as it would have led to obstructive symptoms following sleeve gastrectomy. The patient underwent a simultaneous laparoscopic vertical gastrectomy, gastric lipoma excision, EGD, and laparoscopic cholecystectomyGastric lipomas are benign gastric submucosal tumors, representing less than 3% of all benign gastric neoplasms. Computer tomography and endoscopic ultrasound are important in establishing diagnosis. In current literature, excision with negative margins is standard of care, but small asymptomatic lesions can be followed without intervention.Gastric lipoma are a rare type of gastric submucosal tumors. Size is highly variable. Observation is a reasonable approach when small and asymptomatic, but multiple surgical modalities can be utilized to remove the tumor. Careful utilization of pre-operative imaging including EUS is critical in choosing the appropriate surgery if simultaneous bariatric management is undertaken When GST are found on pre-surgical endoscopy, definitive diagnosis can be challenging and affects surgical planning, requiring consideration of tumor location, type and consequent optimization of surgical modality. Preoperative diagnosis is imperative, as exclusion of atypical/malignant gastrointestinal tumors including gastrointestinal stromal tumor (GIST) is required for appropriate surgical planning.This case report was reported in accordance with the SCARE criteria 2A 58 year-old female with a past medical history of diabetes mellitus and thyroid disease presented for weight loss consultation. There was no other pertinent surgical history. Prior to her endoscopy, her only subjective finding was mild occasional gastrointestinal reflux disease (GERD).The patient underwent a pre-operative esophago-gastro-duodenoscopy (EGD) and was found to have a 3 cm submucosal mass along the anterior aspect of the gastric body, near the incisura angularis; biopsy at that time was indeterminate. As such, the patient was referred to gastroenterology for repeat EGD and endoscopic ultrasound (EUS).Subsequent EGD re-demonstrated a 3 cm mass near the incisura. This had a positive pillow sign (pillowing of the surface of the lesion when prodded). Stack biopsies were obtained, with apparent fat within the biopsy, consistent with lipoma . EUS wasDue to the lipoma's location, resection was critical, as it would have led to obstructive symptoms following sleeve gastrectomy. The patient underwent a simultaneous laparoscopic vertical gastrectomy, gastric lipoma excision, EGD, and laparoscopic cholecystectomy. A longitudinal gastrotomy was made on the greater curvature. Exposure was achieved using retraction sutures. Following this, a laparoscopic submucosal resection of the lipoma was performed. We then used electrocautery to open the mucosa overlying the lipoma. Blunt and ultrasonic dissection were used to mobilize the lipoma. We then identified the vascular pedicle and divided this using the harmonic scalpel. A primary closure of the mucosa and greater curvature followed. The gastrocolic and gastrocolic omentum was taken down using the harmonic scalpel. We then used the linear cutting staple to perform the sleeve gastrectomy. Concurrent endoscopy was performed during the first two stapler firings to ensure that the mucosal suture line was not crossed by the stapler. We then inserted a 40 French Bougie and completed the sleeve in standard fashion. At 3 month follow up the patient was doing well.3Gastric lipomas are benign gastric submucosal tumors, representing less than 3% of all benign gastric neoplasms. They are a proliferation of mature adipocytes occurring primarily in the submucosal layer, with 5–10% being subserosal. A majority are found in the fifth or sixth decade of life. The colon is the most common gastrointestinal location . Most arThe differential diagnosis includes a number of benign and malignant mural neoplasms, including GIST, carcinoid, neural tumor, and metastatic deposit. Lipomas can be difficult to differentiate from other submucosal lesions. Since the lesion occurs in the submucosal plane, endoscopic biopsy is unlikely to be diagnostic. Some endoscopic signs that can guide diagnosis include a ‘tenting sign’ which occurs when the normal mucosa overlying the lipoma is retracted easily away from the mass using biopsy forceps, pillow sign occurs when the forceps produces a soft, cushioning indentation when applied to the lipoma, and a ‘naked fat’ sign, which refers to exposed lipoma that protrudes through the normal overlying mucosa after multiple mucosal biopsies are performed . PreoperComputed tomography (CT) allows for a definitive diagnosis of gastric lipomas because of their uniform fat attenuation measuring from -80 to -120 Hounsfield units. Gastric lipomas on CT usually are a well-circumscribed, smoothly marginated, ovoid or spherical masses of homogenous low attenuation . GastricIn pathology, lipoma, a benign fatty tumor, can arise in any location in which fat is normally present . Most arGiven the size and asymptomatic nature of the lipoma in our patient, we considered non-operative management, however, the location of the lipoma would have interfered with our staple line for the sleeve gastrectomy. Furthermore, given its location at the incisura angularis, there was a risk for gastric obstruction at this level following sleeve gastrectomy.In current literature, excision with negative margins is standard of care, but small asymptomatic lesions can be followed without intervention. In contradistinction, when greater than 4 cm, some surgeons advise resection given a greater likelihood of associated complications. Asymptomatic cases can be alternatively managed without treatment as long as their diagnosis is confirmed. Though generally treated with partial gastrectomy a number of reports document the successful use of endoscopic submucosal dissection (ESD) for resection. Ichinose et al., demonstrated a successful endoscopic resection for a 50 year -old male [Kang et al.  demonstrOur case was unique with only one other published case report that involves simultaneous bariatric surgery and resection of a gastric submucosal lipoma. In that case Hashimoto et al., performed a laparoscopic roux-Y gastric bypass along with a laparoscopic intragastric surgery in a 43 year-old morbidly obese female .There are multiple surgical approaches to excise a lipoma, however, in the setting of simultaneous bariatric surgery, careful pre-operative imaging and planning are critical.4Gastric lipoma are a rare type of gastric submucosal tumors. Size is highly variable. Observation is a reasonable approach when small and asymptomatic, but multiple surgical modalities can be utilized to remove the tumor. Careful utilization of pre-operative imaging including EUS is critical in choosing the appropriate surgery if simultaneous bariatric management is undertaken.There are no conflicts of interest.Ethical approval is exempted as this is a case report.Written/Verbal informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.No source of funding.Dr. Agafonoff did the bulk of the paper.Dr. Braverman contributed to the pathology section of the paper.Dr. Linobel contributed to the radiology section of the paper.Dr. Pitt/Dr. Udelhofen/Dr. Max contributed to the writing of the paper.Not applicable.Guarantor is Dr. Slava Agafonoff.Not commissioned, externally peer-reviewed.


Familial adenomatous polyposis syndrome is a rare condition characterized by the presence of numerous adenomatous polyps in the gastrointestinal tract and associated with risk for colorectal cancer. The disease is scarcely reported in Nigeria and this is the index report in Ilorin. Two cases were clinically diagnosed in our facility. They both presented with gastrointestinal bleeding and numerous rectal and colonic polyps were identified at colonoscopy. Histological examination of the polyps in both cases revealed features in keeping with adenomatous polyps. This report highlights the occurrence of this precancerous condition. Familial adenomatous polyposis syndrome (FAPS) is a rare genetic disease that is characterized by the presence of hundreds to thousands of polyps in the colon and rectum. Untreated, it inevitably leads to cancer . About 1Case 1: A 46- year old Nigerian male cleric presented to the Endoscopy suite of the University of Ilorin Teaching Hospital having been referred on account of an 8- month history of dyspepsia, progressive weight loss, diarrhea alternating with constipation, occasional haematochezia and painful peri-umbilical swellings. He had no fever, cough or drenching night sweats. No history of cigarette smoking or alcohol ingestion. He had a history of surgical removal of a mass lesion from his scalp 5 years earlier with a histological diagnosis of inclusion cyst. He had no known family history of either cancer or polyposis syndromes. He had a twin brother who died of an unknown cause in childhood. His other siblings were in good health. At presentation he was pale, had discrete non-tender right inguinal lymphadenopathy and Sister Mary Joseph (SMJ) nodules. Rectal examination revealed melaena. Gastroscopy revealed multiple sessile polyps in the antrum and a solitary polyp in the duodenal bulb. Histology of the gastric polyps revealed features of dysplasia. Colonoscopy performed using an Olympus CF-180 Evis Exera II forward-viewing colonoscope revealed hundreds of polyps of varying sizes extending from the rectum to the ascending colon. Two large polyps were seen at approximately 15cm and 90cm from the anal verge. Biopsies were taken from these, and one of the pedunculated polyps seen at 45 cm from the anal verge was snared , was referred for colonoscopy on account of a 2-month history of colicky left lower abdominal pain, hematochezia and progressive generalized body weakness. He had no history of weight loss, passage of pellet like stool, spurious diarrhoea nor abdominal swelling. He had laparotomy done 24 years earlier on account of an acute abdominal condition. The details of the surgical findings were not known. He was neither hypertensive nor diabetic. He neither smoked cigarette nor drank alcohol. On examination he was pale and dehydrated. Pulse rate was 120 beats per minute and blood pressure was 120/70mmHg supine. No postural hypotension. Abdominal examination revealed hypertrophied right paramedian scar. His packed cell volume (PCV) was 20%, and he was transfused with two pints of fresh whole blood prior to colonoscopy with an Olympus CF-180 Evis Exera II forward-viewing colonoscope. Numerous sessile and pedunculated polyps of varying sizes were seen in the rectum and colon (nd colon . SurgicaThe diagnosis of FAP is mainly by colonoscopy and the presence of 100 or more polyps that are later confirmed adenomas is clear indication of the diagnosis . Double The second case was most likely a case of attenuated FAPS because the number of polyps viewed at colonoscopy was less than 100. The two patients presented with rectal bleeding which is the most common presentation in FAPS , 10. FirFAPS is rare in Africa where the incidence of colorectal cancer is reportedly low. These two case reports on two clinical variants of FAPS highlight the importance of this disease in our sub-region calling for a high index of suspicion to ensure early diagnosis and prevent the development of CRC.All authors declare that they have no competing interests related to this work.


Morgagni’s hernia (MH) is a rare type of congenital diaphragmatic hernia with limited available literature. Late presentations are infrequent and the ones complicated due to gastric volvulus are even rarer. Another uncommon association of MH is with small bowel diverticulosis. We herein discussed a case of gastric volvulus as the content of MH, and small bowel diverticulosis present in a patient concomitantly.A 30 year old woman, who presented with a one year history of epigastric burning and indigestion, occasionally associated with pain and vomiting. On clinical examination, no clue to the diagnosis could be ascertained. Her chest and abdominal x-ray indicated an abnormal air-fluid level at right hemithorax, which prompted a Computed Tomography (CT) scan, showing organo-axial gastric volvulus. MH with gastric volvulus was observed during laparotomy and trans-thoracic reduction of the contents was performed, along with repair of the defect. Multiple intestinal diverticuli were also found and the largest diverticulum was excised.Gastric volvulus through MH is a rare but potentially life-threatening condition. Non-specific symptoms like epigastric pain and vomiting can delay the diagnosis and management, however, advanced imaging techniques like CT scan can speed up this process. After the diagnosis is made, surgical repair should be attempted regardless of symptoms.The online version of this article (10.1186/s12893-018-0399-y) contains supplementary material, which is available to authorized users. Morgagni’s hernia (MH) is an uncommon birth defect accounting for about 3% of all congenital diaphragmatic hernias, with substantial morbidity and mortality . It is aA 30-year-old female presented with complaints of epigastric burning and indigestion for 1 year, which was occasionally associated with pain and vomiting. On a previous oesophago-gastroduodenoscopy, multiple oesophageal ulcers were noted, located from 30 to 35 cm and the mucosa was seen to be circumferentially hyperaemic. Upon investigation, chest and abdominal X-ray showed abnormal air-fluid level at right hemithorax as shown in Fig. An elective laparotomy was performed through a midline incision. Stomach was not seen in the abdominal cavity, but pull on the gastrocolic ligament revealed the greater curvature of the stomach through foramen of Morgagni in the right hemi-diaphragm, with the defect measuring 4 × 5 cm Fig. . The herMH is the rarest form of congenital diaphragmatic hernia which occurs through a developmental defect as a result of failure of fusion between the sternal and costal portions of the muscle . UsuallyDiagnosis of MH is facilitated by chest x-ray, abdominal x-rays, barium studies or ultrasound, but currently CT scan is the diagnostic modality of choice . The scaLiterature revealed very few cases of gastric volvulus into the foramen of Morgagni, with presenting features in most cases being hematemesis, melena, abdominal bloating, and vomiting , 13. GasIn order to prevent recurrences of gastric volvulus, a temporary gastrostomy may be added to act as a diversion thereby protecting the high jejunal repair and to fix the stomach to the anterior abdominal wall, serving as a gastropexy. The location of gastrostomy tube is essential, as recurrence of volvulus has been reported after gastrostomy, acting as two fixed points of the axis . LiteratSBD occurs due to abnormal peristalsis resulting from motor dysfunction of the gut. It is more frequently confined to the proximal jejunum and distal ileum due to a wider diameter of vasa recta in these segments . ClinicaIn conclusion, MH along with gastric volvulus and small bowel diverticulosis is a rare occurrence, and may not be identified clinically; meticulous analysis of CT scan followed by surgical exploration allows definitive diagnosis. Hernia closure using non-absorbable sutures along with gastropexy and diverticulectomy yields good results with no major sequelae.Additional file 1:Timeline for Case Report. A flowchart depicting the events and plans of the case with respective dates from the time of presentation up to the latest follow up. (DOCX 38 kb)


Aspergillosis in patients with impaired immunity usually presents with invasive pulmonary infection and dissemination to a variety of organs via hematogenous spread. Aspergilloma in the retroperitoneal cavity is a rare disease with only a few cases reported in the literature. To the best of our knowledge, the present case of a retroperitoneal aspergilloma with no surgical history is only the second report in the literature.A 65 year-old man, who had been receiving immunosuppressive treatment for rheumatoid arthritis with vasculitis for 9 years, was referred to the Urology Department with a retroperitoneal mass. This was confirmed by computed tomography performed during treatment for pulmonary aspergilloma. Because it was not possible to rule out malignant disease , surgical exploration was performed. Pathological examination revealed aspergillus hyphae with fat necrosis, and retroperitoneal aspergilloma was diagnosed and appropriately treated. The tumor did not recur subsequently.Our present case emphasizes that pharmacological treatments for aspergilloma in the retroperitoneal cavity have poor drug transitivity, so the relative effectiveness of pharmacological response is not useful for differentiating retroperitoneal aspergilloma from malignant disease. Aspergillus colonizes a cyst or existing cavernous lesion and multiplies, forming a fungus ball. Long-term ingestion of antifungal agents is necessary to treat pulmonary aspergilloma and surgical resection may be indicated for isolated lesions when they are refractory to antifungal treatment.Aspergillosis, alongside candidiasis, is one of the most common deep-seated mycoses. The increased number of immunocompromised patient resulting from the use of immunosuppressive drugs for organ transplantation and other indications, as well as the effects of anticancer chemotherapy, has resulted in an increased incidence of opportunistic aspergillosis , 2. AspeHere, we report a rare case of retroperitoneal aspergilloma successfully treated by surgical resection after pharmacological therapy.A retroperitoneal mass on the left side was found in a 65-year-old-man who was then referred to the Urology Department. He had been receiving methylprednisolone and cyclosporine as treatment for rheumatoid arthritis with vasculitis in the Rheumatology Department of the same hospital. He complained of exertional dyspnea and was hospitalized on suspicion of atypical pneumonia. Blood tested positive for both β-D-glucan and aspergillus antigen. A diffuse nodular shadow across both lungs was seen on chest computed tomography (CT) and a diagnosis of pulmonary aspergilloma was made. Treatment with voriconazole (200 mg twice a daily) was initiated.On subsequent CT 8 months thereafter, the nodular shadow in the lungs appeared smaller, but a mass in the left retroperitoneum was now seen. Consequently, he was referred to the Urology Department. His past medical history included rheumatoid arthritis with vasculitis, steroid-induced diabetes, hyperlipidemia, and compression fracture of the lumbar vertebrae. He had been receiving methylprednisolone and cyclosporine treatment for 8 years. In addition, he received bezafibrate and his diabetes was controlled with hypodermic insulin and oral sitagliptin. He had no notable family history. The patient’s physical findings were normal, except for chest and joint symptoms related to rheumatoid arthritis with vasculitis.Blood test results on admission were as follows: white blood cells, 11830/μL; C-reactive protein, 2.16 mg/dL; glucose, 250 mg/dL; total-cholesterol, 230 mg/dL; triglyceride, 642 mg/dL. There was no apparent liver or kidney dysfunction. The aspergillus antigen level was 0.7  and the β-D glucan level was 240 pg/ml (> 11.0). The frequent occurrence of a diffuse nodular shadow across both lungs on a simple CT image of the chest was noted. Thus, intravenous voriconazole was commenced. On subsequent CT 3 weeks later, a reduction in the pulmonary nodular shadow and improvement of the previous pneumonia was identified .In this case, even though the patient had already received voriconazole treatment previously which had reduced the pulmonary lesion, the retroperitoneal lesion appeared to be increasing in size. The disease course did not allow us to rule out a retroperitoneal malignant tumor for which the treatment of choice is surgical resection, as same as previous reports. This led to the appropriate treatment for this patient despite an erroneous indication. We have presented a very rare case of retroperitoneal aspergilloma, thought to be only the second in the literature. Thus, physicians should bear in mind the possibility of failure of anti-mycotic treatment when diagnosing such immuno-deficient patients, and should select appropriate treatment.


Acute Spinal Subdural Hematoma (ASSH) is a rarely recognized condition that may result in severe irreversible neurologic complication. A 7-yr old girl presented to Neurology Department, Mofid Hospital, ShahidBeheshti University of Medical Sciences, Tehran, Iran with limping and pain in lower extremities and acute paraplegia without history of direct trauma. The patient had muscle weakness in lower limbs and was unable to bear weight. Deep Tendon Reflexes (DTR) in lower extremities had increased. Her MRI showed spinal subdural hematoma we reextended from T2 to T6. We performed laminectomy from T2 to T5 and about 70 cc of subdural hematoma was evacuated. One month after the surgery, the patient's neurological deficit resolved completely. The results showed the pivotal role of attention to clinical manifestation in acute spinal subdural hematoma and early diagnosis to prevent irreversible neurologic complication. Acute spinal subdural hematoma (ASSH) is an uncommonly recognized condition that may result in severe irreversible neurologic complication. It usually presents with severe and localized back pain, rapidly progressive myelopathy and a poor outcome for functional recovery unless rapid surgical decompression is performed . Plain CT and MR can be used as complementary studies if needed to confirm the diagnosis of ASSH . Only a Here, we presente a 7-yr old girl with ASSH without obvious history of trauma.A 7-yr old girl was transferred to Mofid Hospital, ShahidBeheshti University of Medical Sciences, Tehran, Iran with limping and pain in lower extremities and acute paraplegia. She had no history of direct trauma to back, no previous disease, no medication and no family history of neurologic disorders. . In physical examination, she was good in general appearance with BP: 90/60, HR: 80, RR: 20, T: 36.5 axillary. She was unable to bear weight. Deep Tendon Reflexes (DTR) in lower extremities had increased with extensor plantar response. Cranial nerve and cerebellar examination were normal. Laboratory tests include complete blood count (CBC), prothrombin time, partial thromboplastin time, biochemistry and inflammatory markers were normal. Her MRI showed spinal subdural hematoma extended from T2 to T6 vertebra (In her history, she had a ring dancing just for one time, and no history of traumavertebra ,2. The pWe presented a 7-yr old girl with ASSH and with lower extremities pain as well as weakness without history of trauma or medication. For diagnosis, in addition to history and physical examination, CT or MRI is needed. MRI and CT provide characteristic findings including: hyperdensity within the dural sac, clumping, location, and streaking of blood within the dural sac; an inhomogeneous and variable signal intensity on all MRI pulse sequences . In our patient, MRI showed subdural hematoma from T2 to T6 within the dural sac. Both conservatives and surgical management could be an option in management of ASSH considering the neurological course . In mostIn conclusion, results showed the importance of attention to clinical manifestation in ASSH and early diagnosis to prevent irreversible neurologic complication. MRI and plain CT are necessary and provide characteristic findings.


We here report an extremely rare case of metastatic hepatocellular carcinoma to the nasal cavity only with MRI scan including diffusion-weighted imaging and a brief review of previous literature case reports. A brain MRI scan that was performed to identify brain metastasis showed a solid mass occupying the left nasal cavity (3.7 × 1.8 × 2.8 cm). The mass showed iso-signal intensity on ed image . Gradiened image . There wed image . The tum3 Knowledge of the location and radiographical appearance of metastatic HCC is therefore important for accurate tumour staging, to assure the patient the most appropriate treatment and best chance for survival. Because of rarity and lack of distinguishing features, metastasis of malignant neoplasms to the sinonasal region is often mistaken for primary neoplasm of the sinonasal tract.7 The kidney is the most common primary site, but other sources include the lung, breast, urogenital tract, gastrointestinal tract and thyroid gland.2 HCC easily metastasizes, and extrahepatic metastasis occurs in more than 50% of HCC patients, with the most common metastatic sites being the lungs, abdominal lymph nodes, bone and adrenal glands.5 Metastasis of HCC to the sinonasal region, however, is uncommon and metastasis to the nasal cavity is exceedingly rare, with only 13 cases reported in the literature 13 and allow metatastic emboli to bypass to the pulmonary venous system, giving rise to metastasis to the head and neck region without involvement of the lungs.13 The lymphatic system provides another route of spread. Tumour emboli from the regional lymph nodes can flow into the thoracic duct. In such cases, invasion of the hepatic, peripancreatic, celiac and para-aortic lymph nodes would be expected before the disease would spread into the head and neck.14 Metastases can reach the head and neck via retrograde flow through the intercostal, mediastinal or supraclavicular lymph vessels.15 In our case, because there was no metastatic lymphadenopathy but lung metastases were present, lymphatic spread seems more likely than haematogenous spread.HCC metastasizes by either lymphogenous or haematogenous spread.16 Recurrent profuse epistaxis appears to be specific to haemangiomas and certain metastatic tumours, including renal cell carcinoma and melanoma.17 Frequent nasal bleeding from these metastatic tumours are known to be associated with their hypervascularity. In addition to these tumours, metastatic HCC might be a candidate for recurrent profuse epistaxis owing to the high vascularity of the tumour as well as coagulopathy owing to underlying liver cirrhosis.18 The present patient, however, had no sinonasal symptoms when he was referred for headache and the nasal cavity mass was incidentally found on brain MRI scan. Nasal obstruction, facial pain and rhinorrhea had developed during follow-up as the size of the mass rapidly increased. Thus, as in the present case, the only clue of metastasis might be a history of a primary tumour elsewhere.Metastatic tumours to the sinonasal cavity have no distinctive clinical features that may facilitate their early diagnosis. Epistaxis, facial deformity, pain and nasal obstructions are the common presenting symptoms, which are identical to those produced by primary tumours in the same area.T2 signal intensity with no restricted diffusion on DWI. Virtually all sinonasal tumours are highly cellular, with relatively little intracellular and intercellular water.19 As a result, the majority of these tumours have intermediate signal intensity on T2 weighted images.19 It is rare for sinonasal malignant tumours to have inherently high T2 weighted signal intensity. Such high T2 weighted signal intensity may occur with benign or low-grade minor salivary gland tumours, schwannomas, haemangiomas and polypoid tumours such as inverted papillomas.20 Technically feasible in the head and neck regions, the addition of DWI increases detection of malignant lesion and is useful for differentiating both solid from cystic lesions and benign from malignant lesions.21 Mostly, malignant lesions have lower ADC values compared with benign lesions. In a retrospective study of 33 patients with 17 benign and 16 malignant head and neck lesions, an optimal ADC threshold of 1.3 × 10−3 mm2 s−1 was established for diagnosis of malignant tumours from benign lesions.22 In the present patient, the mass showed high signal intensity with intermediate signal foci on T2 weighted images and restricted diffusion of the tumour was not apparent  on DWI. This finding probably related to the differentiation of the metastatic HCC, as it is known that histopathological differentiation of HCC is inversely correlated with the ADC value.23 The metastatic HCC presented here was moderately differentiated. This suggested that metastatic HCC may have varying T2 signal intensity with a wide range of ADC values, and may mimic benign tumours in the nasal cavity. Further investigation should be performed with a larger case series.In this case, the metastatic HCC showed high 3 metastasis to the sinonasal region is also associated with advanced disease and early mortality.25 The mean survival time of patients is reported to range from 4 weeks to 26 months after the identification of sinonasal metastasis.27 In our review of the literature,32 most patients were dead less than 2 months after diagnosis of the nasal cavity metastasis. Three of 13 reported patients died of terminal hepatic failure, one of sudden respiratory failure and one of multiple metastases; the remaining four cases were in the following 8 to 15 months. Various treatments for metastatic HCC to the sinonasal cavity have been reported, including surgical resection, palliative radiotherapy and transcatheter arterial embolisation to control nasal bleeding. The treatment should be selected on an individual basis and the purpose of treatment.32Metastasis to the nasal cavity is usually associated with advanced disease and early mortality. As most extrahepatic HCC occurs in patients with an advanced intrahepatic stage of tumour,In conclusion, although metastatic HCC to the nasal cavity is rare and the imaging findings are rather nonspecific, clinicians and radiologists should be aware of this unusual presentation because of its poor prognosis and the possibility of rapid deterioration in the setting of underlying HCC. We present a rare case of metastatic HCC to the nasal cavity with MRI scan including DWI.Metastasis to the nasal cavity of HCC is extremely rare. But when it occurs, it usually associated with advanced disease and early mortality.T2 weighted image and have lower ADC values compared with benign tumours.Most of sinonasal malignant tumours are highly cellular, with relatively little intracellular and intercellular water, so they have intermediate signal intensity on T2 weighted images, and restricted diffusion of the tumour was not apparent on DWI depending on histopathological differentiation.But, even sinonasal metastatic cancer of HCC has high signal intensity on Written informed consent for the case to be published  was obtained from the patient(s) for publication of this case report, including accompanying images.


Sclerosing stromal tumors (SST) are rare, benign tumors classified as sex cord stromal tumors. To our knowledge, positron emission tomography with computed tomography (PET-CT) findings of SST have only been described in one report and imaging findings on diffusion-weighted imaging (DWI) have only been described in three reports. Characteristic imaging features of SST on PET-CT and DWI have not yet been identified. Here we report a case of multilocular SST with solid components showing mild FDG uptake and slight hyperintensity on DWI, and reviewed the literature.Seventeen-year-old woman presented with a complaint of abdominal pain and was admitted due to infectious colitis. Ultrasonography incidentally revealed a multiseptated cystic mass in the pelvis. Magnetic resonance imaging (MRI) showed a large multilobulated cystic mass with irregularly thickened septa and solid components originating in the left adnexa. On T2WI, the cystic components had the same signal intensity (SI) as water, and the irregularly thickened septa and solid components showed intermediate SI higher than the SI of the uterine myometrium. The septa and solid components also showed early strong enhancement on contrast-enhanced T1WI and slight hyperintensity on DWI. The PET-CT showed mild FDG uptake in the solid components of the tumor (SUV: 2.11). According to previous articles, the morphology of SSTs are various; solid mass, well-circumscribed multilobular mass, well-demarcated mass, and multilocular cysticmass. According to the reports describing DWI findings of SST, the SI varies from significant hyperintensity to slightly hyperintensity like in this case. Only one report describing PET-CT findings of SST showed intense FDG uptake (SUV max: 7.0).The findings on DWI and PET-CT of our case and the past reports describing PET and DWI findings of SSTs are not consistent. The wide variety of the signal intensity on MRI and FDG uptake on PET could be due to the pathological diversity caused by the cellular areas undergoing collagenous sclerosis, which transforms the tumor into admixture of the collagen and the densely fibrous components with edema. Sclerosing stromal tumors (SST) are benign ovarian neoplasms first described in 1973 by Chalvardjian and Scully .To our knowledge, however, there has been only one report describing positron emission tomography with 2-deoxy-2-[fluorine-18]fluoro- D-glucose (FDG) integrated with computed tomography (PET-CT) findings of SST in the English-language literature  and threTherefore, here we report a case of SST with solid components showing mild FDG uptake : 2.11) and slight hyperintensity on DWI.Seventeen-year-old woman, nulligravida, presented with a complaint of abdominal pain and was admitted due to infectious colitis. Ultrasonography incidentally revealed a multiseptated cystic mass in the pelvis. Family history and past medical history were unremarkable, and her menstrual cycle was regular. Blood cell counts and blood biochemistry were normal. Serum levels of alpha-fetoprotein (AFP), carcinoembryonic antigen (CEA) and carbohydrate antigen 19–9 (CA19–9) were all within normal limits, while cancer antigen 125 (CA-125) was elevated at 76.3 U/mL . Levels of serum hormones including estradiol , luteinizing hormone  /ml and follicle-stimulating hormone  were normal.Pelvic magnetic resonance imaging (MRI) showed a 141 × 96 × 127-mm well-demarcated multilocular cystic mass with irregularly thickened septa and solid components originating in the left adnexa. On T2WI, the signal intensities of the cystic components had the same SI as water, and those of the irregularly thickened septa and solid components had intermediate SI, higher than the SI of uterine myometrium . Although SST tended to show high SI on DWI, the exact degree of high SI varied between cases.SST of the ovary are rare, benign tumors classified as sex cord stromal tumors. The tumor tends to affect younger women predominantly in their 20s and 30s, in contrast with other sex cord stromal tumors such as thecomas and fibromas, which are most common in women in their 50s and 60s . PreoperThere have been three reports describing DWI of SST in which nodular solid areas in SST showed significant hyperintensity , hyperinIn this case, a malignant ovarian tumor was ruled out as a preoperative diagnosis due to the low FDG uptake. In contrast, only one report describing PET-CT findings of SST described that a solid component of the tumor that corresponded to well enhanced area on contrast-enhanced T1WI showed intense FDG uptake (SUV max: 7.0) . AlthougThe wide variety of morphology, signal intensity on MRI and FDG uptake on PET among SST might be affected by one of their characteristic features, that is, the tendency of the cellular areas to undergo collagenous sclerosis, producing a variegated appearance due to admixture of cellular, densely fibrous and edematous tissue on low power microscopic examination Fig. a. FurtheHowever, further studies on correlation between imaging findings on T2WI, DWI and FDG-PET and pathological examination are warranted because there have been only a few reports describing imaging features of SST on DWI and PET-CT.


Androgen-secreting ovarian neoplasms are rare ovarian tumors that present with hirsutism and virilization which may manifest as severe alopecia, deepening of voice, and clitoromegaly. Most often, ovarian tumors are found to be very small or even undetectable. In such cases, bilateral salpingo-oophorectomy should be performed after ruling out other causes of high androgens. We present a 63-year-old postmenopausal woman with clinically and radiologically undetectable testosterone-secreting ovarian tumor, which was later on detected on biopsy. Hyperandrogenism refers to the condition in which excessive androgens are found circulating in the female body. Clinical manifestations of hyperandrogenism include hirsutism, alopecia, acne, virilization, and emotional distress . Common μg/dL), hyperlipidemia, normal TSH (1.09 IU/mL), LH, FSH and estradiol. Abdominal ultrasound scan and uterine echotexture were normal and Pap smear was negative. CT scan brain showed normal pituitary gland. MRI brain and internal auditory canal showed a 2.1 x 1.1 x 1 x 1 cm right acoustic schwannoma in the internal auditory canal with extension into the cerebellopontine angle cistern with involvement of the right cochlea and the vestibule with no evidence of pituitary tumor or brain compression. Elevated testosterone settled after the trial of Leuprolide. Diagnosis of hyperandrogenism was made and bilateral salpingo-oophorectomy was performed. Bilateral laparoscopic salpingo-oophorectomy revealed left stromal luteoma, bilateral stromal nodular hyperthecosis  with enlarged thyroid gland, clitoromegaly, male pattern baldness  and hirsutism. Laboratory reports showed normal urea (27 mg/dL) and creatinine (1.45 mg/dL), elevated testosterone , raised DHEA-S (235 osis see , and rigAndrogen-secreting ovarian tumors are a rare cause of elevated testosterone in postmenopausal women, accounting for 5% of all ovarian neoplasms. Most often, these tumors are too small to be detected clinically or imaging radiology as seen in our case. In our case, ultrasound scan, CT scan, and MRI imaging gave no clue to elevated testosterone. It is important to distinguish the source of androgens in order to improve the classification, the understanding of androgen excess disorders, and subsequent management . Then asOvarian hyperthecosis is a rare nonmalignant entity encountered in postmenopausal hyperandrogenism . Other oThe present study reports a rare androgen-secreting ovarian hyperthecosis and stromal luteoma, which was not detected, on USG, CT scan, and MRI imaging. Failure of imaging techniques led to the trial of leuprolide and bilateral laparoscopic salpingo-oophorectomy. This case study demonstrates the importance of investigation of elevated testosterone in a postmenopausal woman when there is no evidence of adrenal tumor.We describe a postmenopausal woman with an androgen secreting ovarian tumors with elevated testosterone levels which may be undetectable on clinical and radiological examination. In such cases, the patient should be thoroughly investigated ruling out all other causes of elevated androgens before undergoing bilateral salpingo-oophorectomy.


Nodular fasciitis is a reactive proliferative lesion. It is often difficult to distinguish from sarcoma, such as malignant peripheral nerve sheath tumor (MPNST). Therefore, both pathological findings and clinical features must be considered.A 75-year-old man presented with a 1 × 1 cm mass on his left forearm that had enlarged rapidly without specific symptoms. Magnetic resonance imaging revealed a mass adjacent to brachioradialis. The lesion showed iso-intensity to muscle on T1-weighted images and hyperintensity on T2-weighted images. We made a clinical diagnosis of nodular fasciitis. Under observation, the mass gradually decreased in size and disappeared after two months. Four years later, another lesion appeared at the same site. We performed total excisional biopsy and histopathological examination indicated low grade MPNST or recurrent nodular fasciitis. Since malignancy could not be excluded, wide resection was performed. At 4 years postoperatively, there has been no recurrence.In this patient, nodular fasciitis of the right forearm was diagnosed clinically and showed spontaneous regression. However, recurrence was noted after four years. While the clinical features suggested recurrent nodular fasciitis, pathological findings indicated the possibility of low grade MPNST.When it is difficult to determine whether a lesion such as nodular fasciitis is benign or malignant, the patient should be managed by considering the possibility of malignancy. Histological examination sometimes reveals cellular heterotypia and polymorphism. Therefore, nodular fasciitis is often misdiagnosed as soft tissue sarcoma. Immunohistochemistry is useful for diagnosis, but there are only a few markers for malignant peripheral nerve sheath tumor (MPNST). If immunohistochemical findings are unclear, differential diagnosis is difficult. Also, the clinical and histological features sometimes do not correspond, and making a definite diagnosis can be challenging.We report a patient who presented to a tertiary hospital in Japan with a mass that was clinically diagnosed as nodular fasciitis and showed spontaneous regression. Four years later, recurrence was noted. Histopathological examination suggested low grade MPNST, although clinical findings were consistent with recurrent nodular fasciitis. This case was reported in line with the SCARE criteria .2A 75-year-old man presented to the outpatient department with a 1 × 1 cm mass on his left forearm that had enlarged rapidly over the previous 2 weeks without specific symptoms . His pasFour years later, another lesion appeared at the same site. Contrast computed tomography (CT) demonstrated a mass measuring 23 × 10 mm . We consHistopathological examination revealed spindle-shaped polymorphonuclear cells with eosinophilic cytoplasm and vacuoles . The tumSince malignancy could not be excluded, wide resection was performed with a lateral margin of 20 mm from the previous surgical scar and partial removal of brachioradialis muscle at the deep margin . The wou3Nodular fasciitis is a benign reactive lesion that often grows rapidly over several weeks before the patient presents with a mass ,3. It waMPNST is a neurogenic sarcoma, and about 50% of patients have neurofibromatosis type 1. In sporadic cases, MPNST often arises from a large nerve such as the sciatic nerve . MPNST uOur patient’s tumor was 2 cm in diameter, which is typical of nodular fasciitis and rather small for MPNST. Spontaneous regression is a feature of nodular fasciitis, but not MPNST. Also, our patient had no history of neurofibromatosis. Moreover, the tumor did not clearly arise from a nerve, suggesting it was not MPNST. Based on these points, our clinical diagnosis of nodular fasciitis seems correct.On MR imaging, nodular fasciitis shows a homogeneous low signal intensity on T1-weighted images and a heterogeneous intermediate signal intensity on T2-weighted images, with surrounding edema and slightly inhomogeneous enhancement . It can In our patient, S-100 staining was negative in most of the tumor and was weakly positive in some areas. While pathological findings tended to suggest MPNST, the clinical course supported a diagnosis of nodular fasciitis. Making a definite diagnosis was impossible because it was difficult to decide whether S-100 staining was positive or negative.If making an accurate diagnosis from clinical findings is difficult, the surgeon often cannot clearly exclude “possible sarcoma”. It is usual to place more weight on the pathological findings in such cases. If the clinical course does not fit the pathological diagnosis, the final diagnosis should be made by integrated assessment of all available data. Chromosomal analysis and genetic testing are performed for research, but are generally unavailable in routine practice. When it is difficult to determine whether a lesion is benign or malignant, management should be based on the possibility of malignancy.4We reported a rare case of clinical nodular fasciitis of the right forearm showing spontaneous regression and recurrence after four years. While the clinical features suggested recurrent nodular fasciitis, pathological findings were consistent with low grade MPNST. When clinical and pathological features differ, the final diagnosis should be based on comprehensive assessment. However, reaching a definite conclusion may sometimes be impossible.There are no sources of funding to declare.A case report is exempt from ethical approval in our institution.We obtained consent to publish a case report from the patient.HN drafted the article. TK, SA and RA had revised the manuscript critically. TK had revised the histopathological findings. All authors contributed to study concept or design at this submission and approved the final version.N/A.Corresponding author; Hisato Nagano.Not commissioned, externally peer-reviewed.There are no conflict of interests to declare.


A 33-year-old male with a past medical history of hypertension, migraine, depression, and alcohol abuse presented with complaints of nausea, vomiting, and abdominal pain. He was found to have an elevated lipase and ethanol level. CT scan of the abdomen was done which showed peripancreatic fat stranding around the pancreatic head consistent with mild acute uncomplicated pancreatitis. CT scan of the abdomen also revealed multiple clustered nodes in the central mesentery with fatty haziness and ground-glass appearance of the mesenteric fat, representing mesenteric panniculitis. Mesenteric panniculitis (MP) is a fibrosing inflammatory disease affecting the adipose tissue of the mesentery. It can be acute or chronic and is associated with abdominal trauma, abdominal surgery, malignancies, inflammatory, granulomatous, and autoimmune conditions. It was first described in 1924 by Jura and is also known as sclerosing mesenteritis, lipsclerotic mesenteritis, mesenteric lipodystrophy, or mesenteric Weber-Christian disease. It is most commonly diagnosed incidentally during an imaging study, mainly a CT scan, which is the most sensitive imaging test but a definitive diagnosis requires biopsy. It can be asymptomatic but can also present with abdominal pain, nausea, vomiting, diarrhea or constipation, intestinal obstruction, or a palpable abdominal mass. Laboratory results are usually normal in the absence of other conditions. It is a self-limiting disease and may show signs of improvement without treatment on follow-up imaging studies. Sometimes, symptoms can improve but the imaging findings do not change if there is a persistent underlying cause. Prognosis is good due to its benign nature [A 33-year-old male with a past medical history of hypertension, depression, and alcohol abuse presented with complaints of nausea, vomiting, and abdominal pain. He had a history of tobacco and alcohol abuse. He was drinking six cans of beer and a half-pint of vodka daily for the last six months. He did not have any significant medical issues in his family and family history was negative for any cancers. His temperature was 97.8° F, blood pressure was 136/88 mmHg, pulse was 87/minute, and respiratory rate was 14/minute. Physical examination was positive for moderate tenderness in the epigastrium on deep palpation.Initial workup revealed laboratory values as mentioned in Table CT scan of the abdomen was done, which showed peripancreatic fat stranding around the pancreatic head consistent with mild acute uncomplicated pancreatitis. CT scan of the abdomen also revealed multiple clustered nodes in the central mesentery with fatty haziness which is also known as "misty mesentery" and ground-glass appearance of the mesenteric fat representing mesenteric panniculitis Figure .The pancreas was obscured by bowel gas and was not visualized well Figure .The patient was started initially on conservative management for suspected acute pancreatitis. He was kept nothing-by-mouth and was started on intravenous fluids and intravenous pain medications. On the second day after admission, all laboratory abnormalities were improving. AST and ALT levels had normalized. The lipase level was down to 926 U/L. The patient was still having abdominal pain, but it was now around the umbilicus and not in the epigastrium. Nausea and vomiting remained persistent. On the third day, his lipase level came down to 267 U/L and had normalized. Complete blood count, metabolic panel, and hepatic function remained unremarkable. Lactic acid was checked and returned normal.An abdominal X-ray was done and came back negative for small bowel obstruction and showed a small amount of stool in the ascending colon Figure .Due to persistent abdominal pain, nausea, and vomiting, a gastroenterology consultation was obtained. His symptoms were believed to be secondary to mesenteric panniculitis seen on the CT scan and not secondary to acute pancreatitis. The patient was started on 40 mg of oral prednisone daily. After starting prednisone, his symptoms completely resolved within 36 hours. He started tolerating diet and was discharged home with a gastroenterology follow-up. He was prescribed prednisone oral taper over a period of two weeks.The patient was closely followed after discharge and had a repeat CT scan of the abdomen done at six months, which showed the complete resolution of the ground-glass appearance of the fat but the adenopathy persisted Figure .The patient was completely asymptomatic by this time but had unfortunately continued to drink alcohol although he had cut down his frequency of drinking to three to four days in a week but had not cut down on the amount of alcohol. On a repeat CT scan of the abdomen, the patient was again found to have findings of mesenteric panniculitis with persistent adenopathy. He was eventually released from gastroenterology care.MP is a rare fibrosing inflammatory disease characterized by acute or chronic inflammation affecting, mainly, the small and, less commonly, the large intestinal mesenteric adipose tissue. It can be both acute and chronic and is more commonly found in men as compared to women with a male-to-female ratio of 2-3:1; it is more common in Caucasian men [The exact mechanism of pathogenesis is not clear but the non-specific immune response to various stimuli has been suggested to trigger the inflammation of the adipose tissue. It can be associated with inflammatory conditions such as gastritis and pancreatitis, malignancies such as mesenteric tumors or non-Hodgkin's lymphoma, and granulomatous, rheumatic, and autoimmune diseases such as primary sclerosing cholangitis. It has also been reported to be associated with peptic ulcer disease, thermal or chemical injuries, gall stones, abdominal trauma, abdominal surgery, and infections such as typhoid, tuberculosis, and so on. Mesenteric panniculitis findings have also been seen on imaging in patients with mesenteric ischemia or small bowel obstruction . PatientOccasionally, anemia and hypoalbuminemia can be seen but labs can be normal in many patients except inflammatory markers, including the erythrocyte sedimentation rate, and C-reactive protein may be elevated in the majority of patients due to the inflammatory nature of the disease -6. MesenConfirmation with pathology may be required with CT scan findings of the soft tissue mass or suspicious adenopathy to rule out other etiologies such as malignancies or granulomatous diseases but it is not recommended. If the follow-up imaging studies show resolution of the soft tissue mass or adenopathy, the biopsy may not be necessary. Since biopsy is invasive, the risks versus the benefits of biopsy should be considered on a case-by-case basis. When the biopsy is obtained, the most common histologic findings are chronic inflammation and fibrosis .The differential diagnosis of mesenteric panniculitis includes disease that can affect mesentery such as non-Hodgkin's lymphoma, peritoneal carcinomatosis, involving mesentery, tumors arising primarily in the mesentery, carcinoid tumor, mesenteric edema caused by conditions such as congestive heart failure, liver cirrhosis, abdominal trauma, and so on . It is iThe main goal of treatment is the relief of symptoms. If patients are asymptomatic, no treatment is required and most patients remain asymptomatic during follow-up also. Treatment does not always prevent complications, but it improves the symptoms in the majority of patients. If the patient is presenting with symptoms of bowel obstruction or mesenteric ischemia due to thrombosis of the vessel, treatment should be offered. Treatment can be offered with steroids, tamoxifen, azathioprine, cyclophosphamide, colchicine, and, sometimes, radiotherapy. The majority of patients who have a higher degree of inflammation respond very well to steroids and tamoxifen. The patients who don't respond to tamoxifen can be treated with azathioprine. In some cases, surgery is required when patients develop obstructive or compressive symptoms. A follow-up CT scan is not always necessary but can be done; however, imaging findings may or may not show resolution .Mesenteric panniculitis is a rare condition. In asymptomatic patients, it is usually found incidentally on imaging studies during the workup for non-specific abdominal symptoms but even in patients who are symptomatic, diagnosis can be difficult, as many other conditions can also have similar symptoms. The CT scan is the most sensitive test and but cannot be used for confirming the diagnosis. The purpose of this case was to create more awareness about the condition, as improved understanding can help in the timely and better recognition of this condition and can help develop less invasive diagnostic methods and improve its treatment to prevent future complications.


In selected patients, the absorbable fibrin patch TachoSil® is superior to standard surgical treatment in reducing air leakage after pulmonary lobectomy. Pulmonary involvement is not considered a main feature of Marfan syndrome (MFS); however, spontaneous pneumothorax (SP) with a high rate of recurrence is frequently reported. We describe the use of TachoSil® in the supportive treatment of recurrent pneumothorax in a girl with MFS.  A 12-year-old girl with a previous diagnosis of MFS and recurrent history of left spontaneous pneumothorax was submitted to thoracoscopic atypical lung resection. Two patches (9.5 × 4.8 cm) were cut from the adhesive/foam complex (TachoSil®) and were pressed against the sutured area as supportive treatment. The patient recovered with no further SP recurrences.  The use of the TachoSil® surgical patch may be useful in pneumothorax supportive treatment, particularly in pediatric MFS by ameliorating the mechanical strength of the lung. Marfan syndrome (MFS) is a connective tissue disorder comprising multiple organ manifestations caused by a mutation in the gene FBN1 that codes for the protein, fibrillin-1 , 3. SP iTachoSil® is a sterile ready-to-use absorbable patch for intraoperative topical application. In selected patients, TachoSil® is more effective than standard treatment of air leakage after pulmonary lobectomy . LimitedA 12-year-old girl with a previous diagnosis of MFS was admitted in our Pediatric Surgery Unit with sudden onset of chest pain and shortness of breath after sneezing. Her past medical history revealed two previous left spontaneous pneumothorax , managed with conservative measures and pleural drainage.Upon examination, both physical and radiographic findings were compatible with a pneumothorax recurrence on the left side. A thoracic CT scan showed bullous pulmonary dysplasia . ThoracoOne week later, a pneumothorax recurrence necessitated a second thoracoscopic intervention. Further pulmonary resection of the atypical upper left lobe was performed using a stapling device. Two patches (9.5 × 4.8 cm) were cut from the adhesive/foam complex (TachoSil®), rolled and inserted into the 5 mm trocar, and subsequently stretched over the sutures. No drainage tubes were inserted. The postoperative period was uneventful, and the patient was discharged one week later. Gross findings showed severe emphysematous involvement in the upper lobe with marked bulla formation . MicroscThree months later the patient was admitted again, this time with a right-sided pneumothorax. A thoracoscopic intervention on the atypical section of the upper right lobe was performed using a stapling device , and newMFS is a rare genetic connective tissue disorder with skeletal, ocular, and cardiovascular manifestations . PulmonaSpecific histological changes in a Marfan lung have not been completely characterized in microscopic detail; however, it is commonly accepted that in MFS, the gross pathological condition of the lung corresponds with “fragile lung” at microscopic evaluation . EmphyseIt is however essential to remember that distal acinar emphysema is not a distinguishing feature of MFS and may be seen in other disease processes, notably in tall, thin males with SP. It is important to interpret pathological findings in the context of clinical history and imaging studies.The relationship between upper lobe pulmonary fibrosis and MFS has also been reported in MFS . Thus, iTherapeutic options for SP include bed rest, oxygen supplementation, manual aspiration, chest tube drainage, and thoracoscopic and surgical interventions . There iTachoSil® is a sterile ready-to-use absorbable patch for intraoperative topical application. It consists of an equine collagen patch coated with fibrin glue components: human fibrinogen and human thrombin. It thus combines the benefits of a pliable material with the hemostatic and adhesive properties of coagulation factors. The product is currently approved in Europe for supportive hemostatic surgical treatment where standard techniques are insufficient. The efficacy and safety of TachoSil® have been demonstrated in adults following digestive and hepatobiliary surgery (liver resection), in pulmonary lobectomy, in sealing the percutaneous nephrolithotomy tract, kidney resection, open repair of abdominal aortic aneurysm, during splenectomy for oncohemathological conditions, after distal pancreatectomy, for obstetric and gynecologic surgery, inguinofemoral and axillary lymphadenectomy, and in a case of corneal perforation –21.There are few studies which have evaluated the use of TachoSil in pediatric surgery. Vida et al.  and GiorThere are no reports in the literature on TachoSil use in pediatric thoracic surgery in MFS. We hypothesized that additional sealing of the anastomosis with a fibrin patch, containing human fibrinogen and thrombin, would improve pulmonary mechanical strength. TachoSil® may be considered effective support in surgical treatment to prevent SP recurrence in children and adolescents with MFS.In MFS patients, definitive surgical treatment should be performed at the first occurrence of pneumothorax because of the high rate of recurrence. The use of the TachoSil® surgical patch should be useful in pneumothorax supportive treatment in MFS in order to ameliorate the mechanical strength of the lung, limit the use of postsurgical drainage tubes, and prevent PS recurrence in the pediatric age.


Introduction: Posterior mediastinal masses present unique diagnostic and therapeutic challenges, particularly when large highly vascularized tumors extend toward or emanate from the spinal cord. The rare nature of these tumors precludes the development of standardized management algorithms, underscoring the importance of case reports.Presentation of case: A 57 year old female presented with exertional dyspnea and right chest pressure. Chest radiography followed by computed tomography (CT) scan demonstrated a 13 cm posterior mediastinal mass involving the T7 vertebral body. CT-guided percutaneous biopsy confirmed benign schwannoma. During open exploration, the tumor bled easily with contact. Angiography with intercostal arterial embolization decreased tumor vascularity while preserving spinal cord perfusion. Subsequent piecemeal resection facilitated exposure of the tumor base and complete resection. Postoperative recovery was uneventful.Discussion: Neurogenic tumors are most commonly located in the posterior mediastinum. When untreated, schwannomas continue to grow, and will inevitably cause compressive symptoms if given sufficient time. Therefore, resection is recommended. This may be performed thoracoscopically in select patients with small tumors, avoiding the morbidity of a thoracotomy incision.Conclusion: Large posterior mediastinal schwannomas require posterolateral thoracotomy and resection. Preoperative angiography helps identify arteries shared by the tumor and the spinal cord, and embolization may reduce tumor vascularity and operative blood loss thereby permitting safer resection. PosteriWe present a case of a symptomatic posterior mediastinal schwannoma in which the late presentation and large tumor size necessitated intercostal artery embolization followed by thoracotomy with piecemeal resection. This work was performed in accordance with SCARE criteria . Written2The patient is a 57 year old female with no underlying medical conditions or daily medications who had smoked one pack of cigarettes daily for ten years and had no family history of benign or malignant thoracic tumors. She initially presented to an emergency department with exertional dyspnea and right chest pressure exacerbated by dry cough. Chest radiography demonstrated a large round opacity in the right chest , promptiCT-guided core needle biopsy of the mass demonstrated a spindle cell neoplasm consistent with schwannoma. She was subsequently referred to us for Thoracic Surgery evaluation. Elective operative resection was recommended, primarily for symptom relief.A right posterolateral thoracotomy was performed at the sixth intercostal space, and a short segment of the seventh rib was resected. A large posterior mediastinal mass occupying most of the lower portion of the chest and causing compressive atelectasis of the entire right lower lobe was encountered . During The following day, the patient was taken to the interventional radiology suite for descending thoracic aortogram. It was noted that the mass was perfused by right T7-T10 intercostal arteries. Particle embolization of these arteries was performed . The artFour days following angio-embolization, the patient returned to the operating room for re-exploration. The seventh rib was resected to facilitate exposure. In contrast to the diffuse contact hemorrhage observed with gentle manipulation of the mass during the first operation, the mass now remained relatively hemostatic during dissection. To allow excision without placing excessive traction on the stalk and risk for avulsion injury to the vascular pedicle, the mass was excised completely in a piecemeal fashion. This operation, as well as the initial operation, was performed by board-certified thoracic surgeons at a university teaching hospital.Pain control was achieved with use of thoracic epidural catheter. Her post-operative course was uneventful. Chest tubes were removed on third day and she was discharged home a day later in good condition. She returned to clinic one month following discharge. Apart from on-going requirement for non-steroidal anti-inflammatory and opiate medications for pain control, she was recovering well. She verbalized her gratitude for the care provided and the relief of her dyspnea. Her chest radiograph at follow up showed clear lung fields with complete resolution of pulmonary edema .Fig. 5Po3Schwannomas, like other neurogenic tumors, typically arise in the posterior mediastinum. However, they may also arise from the middle mediastinum, and have been reported to arise from the phrenic nerve, recurrent laryngeal nerve, vagus nerve, and pulmonary artery . BecauseBecause imaging characteristics of mediastinal masses often do not allow for definitive diagnosis and may not differentiate benign from malignant conditions, histopathologic assessment is prudent. Depending on the location of the tumor, this may be accomplished with fine-needle aspiration, ultrasound or CT-guided percutaneous biopsy, mediastinotomy, or video-assisted thoracoscopic surgery . HoweverOn histologic assessment, schwannomas contain areas of dense spindle cells (Antoni A) and hypocellular stromal areas (Antoni B), and exhibit protein S100 positivity .When untreated, schwannomas continue to grow, and will inevitably cause compressive symptoms if given sufficient time. Despite the low likelihood of malignant transformation, resection of mediastinal schwannomas is advised, unless the patient has a short life expectancy or severe comorbidities placing them at substantial risk for perioperative and postoperative complications. When feasible, thoracoscopic approaches minimize the pain and morbidity associated with a thoracotomy incision . HoweverAngioembolization has the potential to decrease the vascularity of the tumor and decrease intraoperative blood loss. Madariaga et al.  recently4Angiography prior to resection of large schwannomas is useful for identifying tumor blood supply, aberrant vascular anatomy, and arteries shared by the tumor and spinal cord. Intercostal artery embolization serves to reduce tumor vascularity and operative blood loss. Pursuing this approach would most likely have spared our patient the intraoperative blood loss incurred at the first operation, and the need for a second surgery.Future research should seek to establish standardized protocols to select patients with posterior mediastinal masses for preoperative angiography.None.None.This study was exempt from approval by the University of Florida Institutional Review.Written informed consent was obtained from the patient. This study was exempt from approval by the University of Florida Institutional Review.TJL and OO contributed to study conception and design. All authors contributed to the literature review. TJL drafted the manuscript. MP, TM, and OO provided critical revisions. All authors approved the final version of the manuscript.A case report is a medical/educational activity that does not meet the Department of Health and Human Services definition of “research”, which is: "a systematic investigation, including research development, testing and evaluation, designed to develop or contribute to generalizable knowledge."TJL and OO accept full responsibility for this work.Not commissioned, externally peer reviewed.


Periampullary neoplasms are a heterogeneous group of tumors arising within 2 cm of the ampulla of Vater. Neuroendocrine tumors can originate throughout the entire body, from neuroendocrine cells. These neoplasms exhibit deep differences, according to their origin and biological behavior.We describe a case of a 79-year-old man who underwent pancreaticoduodenectomy for adenocarcinoma of the ampulla of Vater after proper staging. At gross histology, an incidental pancreatic neuroendocrine tumor was also documented. Despite two synchronous neoplasms, the patient survived 34 months with no evidence of recurrence at follow-up.The synchronous presence of a second primitive tumor in patients affected by a neuroendocrine tumor is reported in the literature; incidence is variable and the most common site is the gastrointestinal tract. Diagnostic workup for ampullary neoplasms includes abdominal computed tomography (CT) scan, magnetic resonance imaging (MRI) and endoscopic ultrasound (EUS). These investigations infrequently may detect subcentimetric lesions.We believe this case is currently extremely rare. Preoperative diagnosis of synchronous PanNET would not have changed our approach since surgical therapy represents the gold standard in resectable ampullary neoplasms, and it has a primary role in the prognosis of the present patient. Periampullary neoplasms are a heterogeneous group of tumors arising within 2 cm of the ampulla of Vater (AoV), including ampullary adenocarcinoma, distal cholangiocarcinoma, duodenal adenocarcinoma, and pancreatic ductal adenocarcinoma , having To the best of our knowledge, only another one case has been reported in the literature about the synchronous unknown presence of a pancreatic neuroendocrine tumor (PanNET) and AoV adenocarcinoma in the same resected specimen . We hereA 79-year-old man was admitted to the emergency department of our hospital for abdominal pain and jaundice. He was referred to our attention by the family physician. His medical history consisted of benign prostatic hyperplasia (BPH), mild mitral regurgitation, and chronic obstructive pulmonary disease (COPD), without previous surgical procedures; he had no family history of neoplasms. He was a former smoker. On physical examination, no mass or tenderness was detected. Vital signs were normal. Blood tests showed hyperbilirubinemia (4.5 mg/dl). Abdominal ultrasonography (US) was performed, showing dilatation of both biliary systems.The patient was admitted to our department of general surgery in order to perform further diagnostic tests. The abdominal computed tomography (CT) scan Figure  revealedMagnetic resonance imaging (MRI) documented a 15-mm low-intensity mass, seemingly protruding in the duodenal lumen with the involvement of the major pancreatic duct (MPD) Figures -3. Due tEndoscopic ultrasound (EUS) and endoscopic retrograde cholangiopancreatography (ERCP) confirmed an 18x17 mm ipoechogenic nodular lesion close to AoV, with infiltration of the duodenal muscularis propria and pancreatic parenchyma. An enlarged lymph node was detected at the porta hepatis. During the examination, fine needle aspiration (FNA) of the ampullary lesion was performed, revealing the presence of malignant cells. After a multidisciplinary team meeting, surgery was recommended. A preoperative evaluation was performed, including a chest X-ray, and the patient underwent the Whipple procedure (pancreaticoduodenectomy) with portal and peri-choledocical lymphadenectomy. The postoperative course was uneventful, and the patient was discharged on his eleventh postoperative day in good clinical condition.The macroscopic surgical pathology report described two distinctive neoplasms Figure . The firThe patient survived 34 months after surgical treatment. He died during a hospitalization due to a urinary tract infection. Septic state was the cause of death. No evidence of disease recurrence was found at the last oncological follow-up performed two months prior that hospitalization.Periampullary neoplasms and neuroendocrine neoplasms (NENs) exhibit significant differences, according to their origin and biological behavior.The first ones are both benign and malignant lesions that arise at the inlet of the CBD and MPD, in proximity to AoV . AoV adeNENs develop from neuroendocrine cells, which are located in several organs ,5. PanNEPreoperative diagnosis can be formulated with high accuracy and sensitivity through EUS, which can detect subcentimetric lesions with mean detection rates of over 90% . FurtherIn the present case, the patient came to our attention with a diagnosis of adenocarcinoma of AoV, and he underwent proper staging by abdominal CT scan, MRI, and EUS. Those investigations did not raise suspicion of any other lesions.In conclusion, we believe that the present case is extremely rare since we found only another one case in the literature regarding the synchronous presence of PanNET and AoV adenocarcinoma. Despite proper preoperative workup, PanNET was reported only on histological examination of the surgical specimen. In the present case, according to our experience, a preoperative diagnosis of synchronous PanNET would not have changed our approach. Surgical therapy represented the gold standard treatment and played a primary role in the patient’s prognosis.


Histiocytic sarcoma (HS) is an aggressive malignant neoplasm. HS in the central nervous system is exceptionally rare and associated with a poor prognosis. This report documents a case of primary HS of the central nervous system with treatment including surgery, radiotherapy, and chemotherapy.Our patient was a 47 year old female presenting with progressive ataxia, headaches, imbalance, nausea, vomiting, and diplopia. MRI showed a heterogeneously enhancing lesion approximately 2.9 × 3.0 × 2.3 cm centered upon the cerebellar vermis with mild surrounding vasogenic edema and abnormal enhancement of multiple cranial nerves. The patient underwent surgical debulking, which revealed histiocytic sarcoma with grossly purulent drainage. Staging revealed diffuse leptomeningeal involvement, primarily involving the brain and lower thoracic and lumbar spine. She underwent adjuvant radiotherapy to the brain and lower spine and was started on high dose methotrexate. However, she experienced progressive disease in the cervical and thoracic spine as well as pulmonary involvement. Genomic sequencing of her tumor showed a mutation in the platelet-derived growth factor receptor A (p.V0681) which could be targeted with Dasatinib. However, she did not tolerate Dasatinib and she succumbed to progressive disseminated disease eight months from original diagnosis. Our pathologic evaluation also revealed expression of PD-L1 and PD-L2 by tumor cells raising the potential therapeutic role for immune checkpoint inhibition.This case provides an example of effective CNS control with resection and moderate doses of radiation therapy. A review of the literature confirms aggressive multidisciplinary treatment is the most effective treatment against this disease. In addition, genomic sequencing may play an important role in determining new therapeutic options. However, CNS histiocytic sarcoma remains an aggressive disease with a propensity for early widespread dissemination and few long term survivors. Histiocytic sarcoma (HS) is an uncommon malignant neoplasm with morphological and immunophenotypic features of histiocytic tissue. HS presents most commonly with a mass involving soft tissues, gastrointestinal tract, and/or lymph nodes . PrimaryOur patient was a 47 year old female with a history of ulcerative colitis, Sjogren’s syndrome, migraines, and fibromyalgia who presented with a 6 month history of left lower extremity paresthesia followed by a 2 month history of progressive headaches, imbalance, ataxia, nausea, vomiting, and diplopia. Neurologic examination revealed severe gait ataxia requiring assistance to stand or walk and nystagmus with lateral gaze. MRI revealed a 3.0 cm heterogeneously enhancing mixed cystic and solid mass centered upon the cerebellar vermis with mild surrounding vasogenic edema and abnormal thickened enhancement of the bilateral vestibular nerves, left facial nerve, and right trigeminal nerve Fig. .Fig. 1a)The patient was promptly started on steroids, admitted to the hospital, and underwent a midline suboccipital craniotomy for debulking of the large intracerebellar tumor. Upon entering the cystic cavity, grossly purulent material without hemorrhage was noted. Gram stain and cultures were negative for infection. A subtotal resection was achieved and final pathology rendered the diagnosis of primary CNS histiocytic sarcoma see Fig. . The H&EPost-operative MRI showed a midline posterior occipital craniotomy with subtotal resection of tumor and continued bilateral-enhancement along multiple cranial nerves, concerning for leptomeningeal spread Fig. .Fig. 3a)A staging PET scan was performed two weeks following surgery suggesting diffuse leptomeningeal spread. A complete spinal MRI confirmed diffuse leptomeningeal spread in the lower thoracic and lumbar spine as well as cauda equina involvement. Due to the convincing evidence on MRI, a CSF analysis was deferred. Standard lab work was within limits, including LDH Fig. .Fig. 4a)Three weeks following surgery, due to progressive lower back pain, she initiated radiation therapy including whole brain radiotherapy (30.6 Gy in 17 fractions) followed by a posterior fossa boost (5.4 Gy in 3 fractions) and a gross tumor boost (9 Gy in 5 fractions), for a total dose of 45 Gy in 25 fractions to gross disease with simultaneous radiation therapy to the lower thoracic and lumbar spine, receiving 40 Gy in 20 fractions, encompassing the areas of leptomeningeal disease in the spine and cauda equina. Full craniospinal radiotherapy was not performed to limit profound cytopenias that may have prevented further cytotoxic chemotherapy.Interval follow up MRI showed a partial response with persistent posterior fossa disease but near complete resolution of previous leptomeningeal enhancement. Our plan was to initiate chemotherapy with a primary CNS lymphoma regimen with high CNS penetrating therapeutic agents including high-dose methotrexate, high-dose Ara-C, and thiotepa followed by high-dose chemotherapy with BCNU and thiotepa rescued by autologous stem cell transplantation. The patient was started on high dose intravenous methotrexate (HD-MTX) therapy, but received only one cycle secondary to patient intolerance.A CT of the chest approximately two weeks after initiation of HD-MTX therapy for left sided rib pain showed new pulmonary nodules and a left sided pleural effusion, suspicious for metastatic disease. Thoracentesis revealed findings consistent with a malignant pleural effusion. The patient was initiated on cladribine (Leustatin).Within two weeks after the completion of this infusion, the patient developed headaches, somnolence, fever, and nuchal rigidity. Malignant meningitis was suspected and the patient was started on IV antibiotics. Lumbar puncture was recommended but the patient declined. Her mental status significantly improved following antibiotic treatment. Brain MRI showed continued response of the cerebellar residual disease and no new intra-cranial lesions, but an MRI of the cervical spine showed new lesions consistent with metastatic disease in the untreated cervical spinal cord and thoracic vertebrae, including intramedullary involvement. She underwent further radiation therapy targeting C4-C6 and T1-T7 (20 Gy in 5 fractions). The patient rapidly showed improvement in symptoms.Genomic sequencing of her tumor showed a novel mutation in the platelet-derived growth factor receptor A (p.V608I) and therefore, she was started on Dasatinib, a tyrosine kinase inhibitor (TKI) with known CNS penetration . UnfortuThe patient was readmitted to the hospital for continued decline in functional status, weakness, and failure to thrive. CT images of the chest, abdomen, and pelvis showed progressive bilateral pulmonary nodules. An MRI of the spine showed persistent but significantly improved leptomeningeal enhancement. Due to progression of symptoms, worsening systemic disease despite control of CNS disease, and limited systemic options available, the patient chose to enroll in hospice. The patient expired four weeks later, eight months after initial diagnosis.Histiocytic sarcoma of the CNS is an uncommon diagnosis with only about 30 total cases reported in the literature. The median age for diagnosis of HS is 46 years, which is equally distributed in males and females . The proThis case was unique and merits discussion for several reasons. First, at the time of surgery, entry into the cerebellar lesion was associated with drainage of purulent material, which has been documented previously in only three other cases , 3, 26. A review of the literature for CNS HS was performed and the identified cases are shown in Table The majority of cases in the literature received some form of surgery, radiation, and/or chemotherapy. The current standard treatment for CNS HS is surgery to achieve a maximal safe resection and for tissue diagnosis and relieving cerebral edema. An aggressive, multimodality approach is preferred with postoperative radiation therapy +/− chemotherapy, when the disease is unifocal. When the disease has become multifocal, further aggressive radiotherapy and combination chemotherapy is essential. Chemotherapeutics used previously include temozolomide (TMZ), cyclophosphamide, doxorubicin, vincristine and prednisone (CHOP), rituximab, ifosfamide, carboplatin, etoposide (ICE), methotrexate, and thalidomide. There are no documented cases in the literature of successful treatment of multifocal disease. Thus, HS is a very aggressive disease when found in the central nervous system which warrants aggressive treatment with multiple modalities. Despite limited long term survivals of this disease, there have been approximately 10% of cases with long term survival with aggressive treatment including surgery, dose escalated radiation therapy, and chemotherapy.In this case we present a patient with a primary histiocytic sarcoma of the CNS, an extremely rare diagnosis with a poor prognosis. This patient was treated aggressively with surgery, radiation therapy, chemotherapy, and targeted therapy informed by genomic sequencing but ultimately expired of her progressive disease approximately 8 months after diagnosis. The genomic sequencing of the tumor tissue showed a novel PDGFR mutation, which could be targeted with TKI’s. We showed expression of PD-L1 and PD-L2 by the tumor cells suggesting potential therapeutic role for immune checkpoint inhibitors. Our case demonstrates a durable response of CNS disease with radiation doses 40 Gy and above. Therefore, we recommend aggressive treatment with curative radiation doses to the CNS whenever appropriate and possible. Novel therapeutic agents such as TKI’s and checkpoint inhibitors need to be explored.


Schwannomas or neurilemmomas are rare, benign, and usually solitary lesions that arise from the nerve sheath. In the majority of cases, these tumors involve the extremities, head, neck, and trunk.  In the present study, a 52-year-old man presented to our surgical department for the evaluation of a symptomatic lump in his left lateral abdominal wall. CT and MRI confirmed the presence of a cystic mass located between the external and internal oblique muscles. Histopathology and immunohistochemistry confirmed it to be benign schwannoma.  Schwannomas have rarely been reported in the abdominal wall. To the best of our knowledge, this is the first case of schwannoma located in the left upper abdominal wall and the fifth case of abdominal wall schwannoma reported according to the reviewed medical literature.  Benign schwannoma should be included in the differential diagnosis of cystic and symptomatic lesions of the abdominal wall. The treatment of choice is surgical excision, and recurrence is extremely rare. Schwannomas, also known as neurilemmomas or neurinomas, are benign and slow-growing neoplasms originating from Schwann cells of the peripheral nerve sheath. They constitute approximately 5% of all benign soft-tissue tumors. The majority of these tumors are commonly localized in the trunk, head, neck, and extremities –3.Their presentation in the abdominal wall is extremely rare, and to the best of our knowledge, this is the sixth reported case of an abdominal wall benign schwannoma –7. In adThe present study reports the case of a 52-year-old man with a schwannoma of the left upper lateral abdominal wall. The patient provided his written informed consent.We present a case of a 52-year-old man who presented to our outpatient clinic complaining of a painful lump in his left lateral abdominal wall. The patient noticed the development of a small nodule about one year ago, and he also stated that the lump was gradually increasing in size. The patient did not refer any previous abdominal trauma or any further pathologies. Palpation revealed a 4 × 5 cm nodular mass protruding through the left upper lateral abdominal wall. The mass was firm, tender, and not fix to the skin of the abdominal wall.Computed tomography (CT) scan of the abdomen with contrast revealed a 32 × 45 mm cystic mass located between the external and internal oblique muscles . AbdominThe patient underwent total resection of the mass under local anaesthesia . The choMacroscopic examination of the resected specimen revealed a well-circumscribed nodular mass measured 6.5 × 4.5 × 3.5 cm . HistopaThe neoplastic cells were immunoreactive with S-100 protein  and vimeSchwannomas are benign neurogenic tumors, originating from Schwann cells, which normally wrap around the axons of the peripheral nerves and can develop anywhere along the peripheral course of the nerve , 2, 8. SNatural history studies consistently show that the overall average patient age at schwannoma presentation is the fourth decade of life, without a significant sex predilection. Schwannomas are usually sporadic, but they can also be associated with genetic disorders such as neurofibromatosis type 2, schwannomatosis, or Carney's complex. Rare descriptions exist of malignant change in long-standing neurilemmomas, usually in patients with an underlying diagnosis of neurofibromatosis. Malignant transformation is extremely rare in isolated lesions , 9.To the best of our knowledge, only five cases of benign schwannoma located in the abdominal wall have been reported in the medical literature at present –7. In thSchwannomas may grow slowly and may be present for months or years without causing symptoms. Symptoms usually appear when they have grown to the point where they are putting pressure to the nerves near them. Symptoms include a visible lump, numbness, muscle weakness, and aching, burning, or sharp pains. In our case, due to the location of the mass below the external oblique muscle, pain may result from the compression and irritation of the thoracoabdominal and subcostal nerves.On CT and MRI, schwannomas appear as well-circumscribed cystic masses of low to intermediate signal intensity on T1-weighted sequences and high signal intensity on T2-weighted sequences, caused by a predominance of hypercellular Antoni A type tissue , 11.Macroscopically, schwannomas are presented as well-circumscribed masses. Histologically, they are characterized by hypercellularity (Antoni type A) and hypocellular-myxoid (Antoni type B) areas. Nuclear palisading around fibrillary process (Verocay bodies) is often seen in cellular areas .By immunohistochemistry, schwannomas show diffuse and strong expression of S-100 protein. Recent markers frequently positive in schwannomas include podoplanin, calretinin, and SOX10 .Although schwannoma of the abdomen wall is extremely rare, it should be considered in the differential diagnosis of cystic and painful lesions of the abdominal wall. Complete surgical excision is the treatment of choice for these lesions, and malignant transformation is extremely rare. Recurrence is unlikely after complete resection.


Ectopic pancreas is an infrequent submucosal tumor in the gastrointestinal tract defined as a pancreatic tissue lacking vascular or anatomic continuity with the main body of the pancreas. Ectopic pancreas in the ileum is a rare and often an incidental finding. We report a case of ectopic pancreas in the ileum causing obscure gastrointestinal bleeding and episodes of abdominal pain.59-year-old man with 3 months history of intermittent melena, accompanied by the episodes of abdominal pain in the left upper quadrant and generalized fatigue was admitted to our department. The investigations showed that the patient had a low hemoglobin level, i.e. 10.9 g/dL with hypochromic microcytic anemia pattern seen in complete blood count . Esophagogastroduodenoscopy and colonoscopy did not reveal any abnormalities. Magnetic resonance enterography revealed a large (2.5 × 2.3 cm) pedunculated polyp in the ileum. Examination by single-balloon enteroscopy revealed a polyp with long pedicle located approximately 1.5 m distal to terminal ileum. Polypectomy was performed. Histopathologic examination stated, that the specimen contained ectopic pancreatic tissue which was involving muscular layer of the ileum. Ectopic pancreatic tissue included acinar cells and cystically dilated secretory ducts without islets of Langerhans.Our case report reveals a very rare cause of obscure gastrointestinal bleeding accompanied by the episodes of abdominal pain – an ectopic pancreas located in the ileum. Ectopic pancreas  is an infrequent submucosal tumor in the gastrointestinal (GI) tract, defined as a pancreatic tissue lacking vascular or anatomic continuity with the main body of the pancreas –3. The iA 59-year-old man with 3 months history of intermittent melena accompanied by the episodes of abdominal pain in the left upper quadrant and generalized fatigue was admitted to the department. He denied any other change in bowel habits or a history of hemorrhoids and was referred to hospital for evaluation of the GI bleeding. Patient’s medical history did not include any previous diagnoses. He was not taking any medications. At the time he was a non-smoker and did not consume any alcoholic drinks or recreational drugs. The patient also had two repeated episodes of left upper quadrant abdominal pain and dark black tarry feces within the last 3 months prior to admission to our hospital. However, symptoms resolved spontaneously and the patient did not make an appointment to see the doctor.On examination the patient was pale, the abdomen was tender in the left upper abdominal area with no signs of rebound tenderness, no lump was palpable. Digital rectal examination revealed melena; the rest of the examination was unremarkable. The investigations showed that the patient had a low hemoglobin level, i.e. 10.9 g/dL with hypochromic microcytic anemia pattern seen in complete blood count . In addition, the patient had low serum iron, i.e. 6.4 μmol/L  and low ferritin levels, i.e. 28.8 μg/L . The carcinoembryonic antigen level was 1.2 μg/L . Other routine blood tests including lipase, plain chest and abdominal X rays along with abdominal ultrasound, esophagogastroduodenoscopy and colonoscopy were unremarkable. Thorough conventional evaluation of GI bleeding has failed to reveal a source, therefore, it was rational to proceed with further investigation of the small intestine.Usually most cases of bleeding in the small intestine are caused by abnormal blood vessels in the wall of bowel - angioectasias, angiodysplasias, or arteriovenous malformations. However, there are many other possible causes of bleeding in the small intestine, including Crohn’s disease, benign and malignant tumors, polyps and ulcers.. Furthermore, ulceration marks at the top of the polyp were described.Unfortunately, the capsule endoscopy is not reimbursed by Patient Sickness Fund in Lithuania, therefore we performed magnetic resonance (MR) enterography to help visualise possible bleeding site in the small bowel. MR enterography revealed a large pedunculated  polyp, measuring approximately 2.5 × 2.3 cm and involving middle third of the ileum Fig. .. Furthe. Endoloop-Assisted polypectomy was performed. However, the procedure was complicated with postpolypectomy bleeding from the pedicle. Dilution of adrenaline 20 ml (1/10.000) was injected into the bleeding area and the bleeding was controlled.For further investigation, the patient underwent retrograde single-balloon enteroscopy (SBE) to directly visualize pedunculated polyp, described previously on MR enterography. Examination by SBE revealed a polyp with a long pedicle located approximately 1.5 m distal to the terminal ileum Fig. .. Endolo). Ectopic pancreatic tissue included acinar cells and cystically dilated secretory ducts without islets of Langerhans. Also, there was evidence of mucosal ulceration of the ileum.Brownish polyp with rugged surface was noted in the gross specimen. Cross-section of the polyp revealed a yellow node sized 1.5x1x1 cm. In addition, histopathological examination was performed. The report stated that the specimen contained ectopic pancreatic tissue involving longitudinal muscle layer of the ileum Fig. .. EctopiThe patient recovered after the enteroscopy well and had no further GI symptoms (since discharge until the time of writing).Ectopic pancreas is a congenital anomaly, in which pancreatic tissues lacking anatomical or vascular connections with the normal pancreas may be found anywhere within the abdominal cavity other than its usual location . EctopicEctopic pancreas can be found throughout the entire gastrointestinal tract, however, studies indicate that 70–90% of cases are located in the upper gut, including the stomach 25–47%), duodenum 11.7–36.3%) and jejunum (15–35%) [1.7–36.3%5–47%, duAccording to literature, ectopic pancreas is mainly located in submucosa , following by muscular layer (muscularis propria in 8% of cases) and serous layer (11–13% of cases). Lesions which are located in the stomach and duodenal bulb may involve full thickness of the wall (4% of cases) , 10. In Although the pathogenesis of ectopic pancreas remains unknown, based on literature there are two main theories that have been proposed to explain occurrence of ectopic pancreas - misplacement theory and metaplasia theory. The most widely held misplacement theory claims that during the period of embryonic rotation of the dorsal and ventral buds deposits of pancreatic tissue migrate from the main body of pancreas and are implanted at various ectopic sites , 5, 7, 8Ectopic tissue is capable of reproducing same pathologic conditions that can affect the orthotopic pancreas, therefore pancreatitis, formation of cysts and pseudocysts, neuroendocrine syndromes, benign and malignant neoplasms can occur , 4, 13. It has been noticed, that severity of symptoms depends on ectopic lesion size, hence lesions larger than 1.5 cm were associated with more severe symptoms , 14. AbdEctopic pancreas has several characteristic radiographic and endoscopic features that may lead to its identification. During esophagogastroduodenoscopy the ectopic pancreas classically presents as soft rubbery well-circumscribed broad based yellow submucosal lesion (ranging from 1 mm to 5 cm) , 14, 22.Diagnosis of ectopic pancreas is easier in the upper GI tract, where it may present characteristic features. On the other hand, the distal small bowel is notoriously difficult to visualize, therefore diagnosis of ectopic pancreas in the ileum is a rare and usually incidental.Imaging studies and endoscopic visualization are neither specific nor sensitive enough to make diagnosis of ectopic pancreas and histopathological examination remains the gold standard to confirm this anomaly. In our case report fusion of clinical and radiological evaluation finally led us to endoscopic resection of the polyp and a definitive histopathological diagnosis of ectopic pancreas.The first case of ectopic pancreas in ileum presented as gastrointestinal bleeding was reported by Tanigawa et al. in 1993 . In thisIn another case reported by Huan-Lin Chen et al., authors also describe ectopic pancreas in ileum presented as GI bleeding, which was visualised by capsule endoscopy. Among patients with obscure GI bleeding capsule endoscopy has a high sensitivity and specificity for bleeding source detection and is well-suited as the first choice for evaluating obscure GI bleeding . As mentMi-Jeong Lee at al., Qun-Ying Wang et al. and Woo Hyung Choi et al. all described cases in which ectopic pancreas was found in jejunum and presented as gastrointestinal bleeding . In all three reports lesions were found by capsule endoscopy and in two case reports patient’s examination was continued by barium small bowel series to find the exact location. Finally, patients underwent surgery (laparoscopy or laparotomy) to remove lesion, recovered well and symptoms never reoccurred. In the end, histological examination revealed submucosal ectopic pancreatic tissues , 19, 23.For symptomatic ectopic pancreas patients local surgical or endoscopical resection of the lesion seems to be the most appropriate therapy , 13. WhiEctopic pancreas was classified by Heinrich in 1909. Type I is composed of acini, ducts and endocrine islet cells, Type II includes acini and ducts without islet cells, and Type III includes only pancreatic ducts , 13. MosOur case report reveals a very rare cause of obscure gastrointestinal bleeding accompanied with episodes of abdominal pain – an ectopic pancreas located in the ileum. Although ileal polyp was located by MR enterography and later enteroscopy, diagnosis of ectopic pancreas could not be made until polypectomy and histopathological examination.