Weber-Christian disease developing into mediastinitis and pleuritis with massive pleural effusion.

A 53-year-old man visited our hospital complaining of high fever. Chest computed tomography showed left pleural effusion and mediastinitis. He developed painful red subcutaneous nodules in his bilateral lower extremities. Thoracoscopy-assisted exploratory excision showed visceral pleura thickening; panniculitis in the periaortic area was histologically proven. The patient was treated with corticosteroid therapy which immediately reduced the fever. Subsequent imaging examinations after corticosteroid therapy showed improvement of mediastinitis and pleural effusion. This case reminds us that Weber-Christian disease (WCD) should be included in the differential diagnosis of mediastinitis although WCD is rarely associated with thoracic involvement.

Successful treatment of sepsis caused by Staphylococcus lugdunensis in an adult with 22q11.2 deletion syndrome.

A 27-year-old woman visited our hospital because of high fever. She had been diagnosed as 22q11.2 deletion syndrome (22q11.2DS) due to her cardiac history (tetralogy of Fallot), thymic hypoplasia and 22q11.2 deletion. She had a normal CD4/CD8 ratio, a slightly decreased lymphocyte count and normal serum immunoglobulin levels. Blood cultures were positive for Staphylococcus lugdunensis (S. lugdunensis). Infection route of S. lugdunensis in this case was unclear. The patient was successfully treated with several intravenous antibiotics. Infection should be considered when managing patients with 22q.11.2DS. regardless of whether their immune system is impaired.

Long-term Taenia saginata infection successfully treated with meglumine/diatrizoate sodium.

A 46-year-old Japanese man visited our hospital for chronic abdominal pain, persistent diarrhea and discharge of proglottids for 7 years. He had been living in Lao People's Democratic Republic. Ileography using meglumine/diatrizoate sodium (Gastrografin) revealed a long tapeworm. A Taenia saginata including the scolex was excreted through the intestinal tract by the administration of total 780 ml of Gastrografin. Taeniasis is an important disease in the differential diagnosis of imported diseases in Japan. Parasite infection should be suspected in patients with chronic abdominal pain or persistent diarrhea regardless of the findings for small bowel obstruction when there is a history of overseas travel.

[A case of pedunculated colonic adenoma with pseudocarcinomatous invasion and squamous metaplasia occurring in the sigmoid colon].

A case of pedunculated colonic adenoma with pseudocarcinomatous invasion and squamous metaplasia occurring in the sigmoid colon is reported. The patient was a 65-year-old man who visited our hospital for further evaluation of a positive fecal occult blood test. Colonoscopy revealed a pedunculated polyp with a thick swollen stalk in the sigmoid colon. EUS revealed a hyperechoic lesion in the stalk. However, endosonographically, the third and fourth layers of the colonic wall were shown to be normal. It was suspected to be a colonic adenoma with pseudocarcinomatous invasion from these endoscopic findings. Endoscopic polypectomy was performed. Histological examination of the specimen revealed tubular adenoma with moderate epithelial atypia and submucosal displacement of adenomatous glands with variable cystic changes. Squamous metaplasia and hemosiderin deposition were also seen in the mucosal and submucosal layer, respectively. This polyp was diagnosed as a colonic adenoma with pseudocarcinomatous invasion with squamous metaplasia.

Esophageal large fish bone (sea bream jawbone) impaction successfully managed with endoscopy and safely excreted through the intestinal tract.

A 68-year-old man consulted our hospital because of back pain. A chest computed tomography (CT) demonstrated a high-density foreign body in the esophageal wall. There was no evidence of pneumomediastinum. Endoscopic examination demonstrated a large fish bone that was stuck in the esophageal wall. It was dislodged and moved into the stomach. The bone was excreted through the intestinal tract on the seventh hospital day. Unintentional ingestion of large fish bones must be considered potentially dangerous. Complications such as esophageal perforation or mediastinitis should be confirmed by CT; and then, the esophageal foreign body should be removed as soon as possible.

Inflammatory myoglandular polyps: a case series of four patients and review of the literature.

BACKGROUND: Inflammatory myoglandular polyp (IMGP) is a nonneoplastic colorectal polyp. Only a small number of cases have been reported, and the pathogenesis remains unclear. METHODS: We analyzed colonoscopy and histologic findings in 4 patients with IMGP. Histologic confirmation of the inflammatory granulation tissue in the lamina propria, proliferation of smooth muscle, and hyperplastic glands with variable cystic changes formed the criteria for the selection of patients. RESULTS: We treated four cases of IMGP and reviewed the literature on this disease. Three cases were located in the sigmoid colon or descending colon. All 4 polyps were identified as red, pedunculated lesions. All 4 cases had no symptoms. In two cases, endoscopic findings of polyps were necessary to be differentiated from juvenile polyps. CONCLUSIONS: Pedunculated lesions are the main pattern of IMGP. An analysis of endoscopic and histologic features in IMGP of the colorectum revealed that colonic IMGPs resembled juvenile polyps. On colonoscopy, IMGP should generally be taken into consideration as a differential diagnosis of peduncular polyp.

Pedunculated inverted hyperplastic polyp of the sigmoid colon treated with endoscopic polypectomy.

A case of inverted hyperplastic polyp of the sigmoid colon is reported. The patient was a 67-year-old woman who visited our hospital for further evaluation of constipation. Colonoscopy revealed a pedunculated polyp with linear central depression, about 12 mm in diameter, in the sigmoid colon. Excluding the polyp, there was no lesion in the colorectum. Endoscopic polypectomy was performed. Histological examination of the specimen revealed a stalked polyp that had scattered goblet cells and elongated tubules with serrated profiles in the superficial portion. The nodules of deep tubules impinged on the muscularis mucosae. Proliferation of fibromuscular tissue was not seen in the polyp. There was no evidence of malignancy. This polyp was diagnosed as an inverted hyperplastic polyp. Pedunculated-type inverted hyperplastic polyp of the colon is rare.

Gastric hyperplastic polyp associated with proliferation of xanthoma cells observed by magnification narrow-band imaging endoscopy.

A case of gastric hyperplastic polyp with proliferation of xanthoma cells is reported. The patient was a 69-year-old man who visited our hospital for further evaluation of gastric polyps. Endoscopic examination of the upper digestive tract revealed multiple hyperplastic polyps in the gastric antrum. There was a pedunculated polyp with whitish yellow granules, 7 mm in diameter, arising from the greater curvature of the antrum. Magnification narrow-band imaging endoscopy (GIF-H260Z, Olympus) revealed long microcapillaries in the polyp but did not reveal disappearance of the mucosal microstructure or irregular branched capillaries. Endoscopic mucosal resection (EMR) was performed. Histological examination of the specimen revealed the lengthened gastric foveolae in the superficial portion and tight sheet of foamy histiocytes in the lamina propria. Diagnosis of gastric hyperplastic polyp with proliferation of xanthoma cells was made. There was no evidence of malignancy. It is necessary to know that a gastric hyperplastic polyp may associate with gastric xanthoma, although such association is very rare.

Composite paraganglioma-ganglioneuroma in the retroperitoneum.

BACKGROUND: Paragangliomas occur most commonly in head and neck region and much less frequently, they are found in the retroperitoneum. Composite paraganglioma-ganglioneuroma of the retroperitoneum is very rare. CASE PRESENTATION: We present an unusual case of retroperitoneal composite paraganglioma-ganglioneuroma discovered on computed tomography in a 63-year-old female patient. Routine hematological examination and biochemical tests were within normal limits. Plasma adrenaline was 0.042 ng/ml, plasma noradrenaline 0.341 ng/ml, and plasma dopamine <0.01 ng/ml. An abdominal contrast-enhanced CT scan and magnetic resonance imaging revealed a 6.5 cm heterogeneous retroperitoneal mass with a cystic component. The retroperitoneal tumor accumulated (131)I-Metaiodobenzylguanidine ((131)I-MIBG) 48 hours after radioisotope injection. Under the diagnosis of paraganglioma in the retroperitoneum, the patient underwent surgery. The resected tumor (6.5 x 5 x 3 cm) was solid and easily removed en bloc. The cut surface of the tumor and histology revealed two different components in the tumor: paraganglioma centrally and ganglioneuroma on the periphery. She remains disease-free 18 months after surgery. CONCLUSION: This case reminds us that neuroendocrine tumor should be included in the differential diagnosis of a retroperitoneal mass although composite paraganglioma-ganglioneuroma in the retroperitoneum is very rare.

[A case of liposarcoma of the ascending colon mesenterium].

We encountered a rare case of liposarcoma of the ascending colon mesenterium. A 53-year-old man visited our hospital because of a mass, 12x8cm in diameter, in the right upper abdomen. On computed tomography (CT), the tumor showed irregular density with a smooth surface and was enhanced irregularly. A part of the tumor protruded into the colon in the upper portion of the ascending colon. The tumor was surgically resected by right hemicolectomy. Histological diagnosis of the tumor was well-differentiated liposarcoma, sclerosing variant. Radiological findings seemed to reflect the pathological findings well.

Jejunal small ectopic pancreas developing into jejunojejunal intussusception: a rare cause of ileus.

Intussusception is rare in adults. We describe a 62-year-old man with jejunal ectopic pancreas that led to jejunojejunal intussusception and ileus. The patient was admitted to our hospital because of intermittent abdominal pain. Plain abdominal radiography showed some intestinal gas and fluid levels. Abdominal CT scan demonstrated a target sign suggesting bowel intussusception. Jejunography using a naso-jejunal tube showed an oval-shaped mass about 15 mm in diameter with a smooth surface in the jejunum, which suggested a submucosal tumor (SMT), and edematous mucosa around the mass. Partial jejunal resection was carried out and the resected oval-shaped tumor, 14 mm x 11 mm in size, was found to be covered with normal jejunal mucosa. The tumor was histologically diagnosed as type III ectopic pancreas according to the classification proposed by Heinrich. Abdominal pain resolved postoperatively. This case reminds us that jejunal ectopic pancreas should be included in the differential diagnosis of intussusception caused by an SMT in the intestine.

Pedunculated Brunner's gland hamartoma of the duodenum causing upper gastrointestinal hemorrhage.

A case of pedunculated Brunner's gland hamartoma (BGH) of the duodenum causing upper gastrointestinal (GI) hemorrhage is reported. The patient was a 47-year-old man who visited our hospital for further evaluation of tarry stools and shortness of breath. Endoscopic examination of the upper digestive tract revealed a large peduncular polyp with bleeding, about 30 mm in diameter, arising from the wall of the second portion of the duodenum. GI bleeding occurred from the base of the stalk of the polyp. Endoscopic polypectomy was performed. Histological examination of the specimen revealed that the main body of the polyp contained several lobules of mature Brunner's gland with areas of cystic dilatation. The surface epithelium consisted of normal duodenal mucosa with areas of focal ulceration. This polyp was diagnosed as a BGH. The symptom of tarry stools resolved after endoscopic resection. Our case shows that treatment is necessary for duodenal BGH if GI bleeding occurs.

Usefulness and safety of 0.4% sodium hyaluronate solution as a submucosal fluid "cushion" for endoscopic resection of colorectal mucosal neoplasms: a prospective multi-center open-label trial.

BACKGROUND: Sodium hyaluronate (SH) solution has been used for submucosal injection in endoscopic resection to create a long-lasting submucosal fluid "cushion". Recently, we proved the usefulness and safety of 0.4% SH solution in endoscopic resection for gastric mucosal tumors. To evaluate the usefulness of 0.4% SH as a submucosal injection solution for colorectal endoscopic resection, we conducted an open-label clinical trial on six referral hospitals in Japan. METHODS: A prospective multi-center open-label study was designed. A total of 41 patients with 5-20 mm neoplastic lesions localized in the colorectal mucosa at six referral hospitals in Japan in a single year period from December 2002 to November 2003 were enrolled and underwent endoscopic resection with SH. The usefulness of 0.4% SH was assessed by the en bloc complete resection and the formation and maintenance of mucosal lesion-lifting during endoscopic resection. Safety was evaluated by analyzing adverse events during the study period. RESULTS: The usefulness rate was high (82.5%; 33/40). The following secondary outcome measures were noted: 1) steepness of mucosal lesion-lifting, 75.0% (30/40); 2) intraoperative complications, 10.0% (4/40); 3) time required for mucosal resection, 6.7 min; 4) volume of submucosal injection, 6.8 mL and 5) ease of mucosal resection, 87.5% (35/40). Two adverse events of bleeding potentially related to 0.4% SH were reported. CONCLUSION: Using 0.4% SH solution enabled sufficient lifting of a colorectal intramucosal lesion during endoscopic resection, reducing the need for additional injections and the risk of perforation. Therefore, 0.4% SH may contribute to the reduction of complications and serve as a promising submucosal injection solution due to its potentially superior safety in comparison to normal saline solution.