[Sarcoidosis diagnosed by a tiny esophageal lesion:a case report].

A woman in her 60s was referred to the Department of Gastroenterology with anemia. She had a recurrent transient loss of consciousness 11 years ago, and she was examinated at the cardiology and neurology departments, but the cause was not identified. Epileptic seizures were suspected. Sodium valproate medication was started, and the patient's condition progressed with no recurrence. Esophagogastroduodenoscopy showed a tiny submucosal tumor-like lesion with mild depression in a 21cm thoracic esophagus. Biopsy revealed epithelioid granulomas with multinucleated giant cells in the subepithelial stroma. Computed tomography (CT), positron emission tomography-computed tomography (PET-CT), and magnetic resonance imaging (MRI) showed multiple lesions in the hilar lymph nodes, spleen, and heart that are typical of sarcoidosis. These findings led to the diagnosis of esophageal lesion associated with sarcoidosis. The patient had no subjective symptoms;however, treatment with prednisolone 30mg was started because cardiac sarcoidosis is a risk of death. Gastrointestinal tract involvement in sarcoidosis is rare;esophageal sarcoidosis is particularly rare, and there are few reports on superficial lesions. Here, we report a case of sarcoidosis that was diagnosed from a tiny esophageal lesion.

Mesenteric inflammatory veno-occlusive disease occurring during the course of ulcerative colitis: a case report.

BACKGROUND: Mesenteric inflammatory veno-occlusive disease (MIVOD) is difficult to diagnose because of its rarity, nonspecific clinical findings, and frequent confusion with other diseases including inflammatory bowel disease. This report presents a very rare case of MIVOD that occurred during the course of ulcerative colitis (UC). CASE PRESENTATION: A 32-year-old man, who had been diagnosed with UC at the age of 29 and was in remission maintained by oral administration of 5-aminosalicylic acid (5-ASA), showed exacerbation of diarrhea and was admitted to the hospital. Since it was deemed an exacerbation of UC, intravenous steroid therapy and oral administration of tacrolimus were initiated, but his condition continued to worsen. Abdominal computed tomography (CT) was performed and showed intraperitoneal free air, leading to a diagnosis of gastrointestinal perforation and the performance of emergency surgery (subtotal colectomy and ileostomy). Histopathological examination of the resected colon of the patient showed mucosal inflammatory findings that were not typical of UC, including multiple organized thrombi with recanalization in the veins existing in the submucosal layer to the subserosal layer and an increased infiltration of inflammatory cells. These findings led to the pathological diagnosis of MIVOD. CONCLUSION: We report a very rare case in which MIVOD occurred during the course of UC.

Factors associated with healing of artificial ulcer after endoscopic submucosal dissection with reference to Helicobacter pylori infection, CYP2C19 genotype, and tumor location: Multicenter randomized trial.

BACKGROUND AND AIM: Healing speed of peptic ulcer is affected by a number of factors, including Helicobacter pylori (H. pylori) infection and intragastric pH. Acid inhibition exerted by proton pump inhibitors differs by CYP2C19 genotype. Herein, we investigated whether healing speed of artificial ulcers formed after endoscopic submucosal dissection (ESD) was influenced by H. pylori infection, CYP2C19 genotype, or other factors. METHODS: A total of 96 H. pylori-positive patients with gastric tumors scheduled for ESD were randomly assigned to receive eradication therapy for H. pylori before ESD (pre-ESD eradication) (n = 44) or after (post-ESD eradication) (n = 52). Patients received eradication therapy consisting of lansoprazole 30 mg, amoxicillin 750 mg, and clarithromycin 200 mg twice daily for 1 week. After ESD, lansoprazole 30 mg was given once daily for 8 weeks. Ulcer size was endoscopically measured on the next day and at 4 and 8 weeks after ESD. RESULTS: Mean reduction rate of artificial ulcer area in the pre-ESD eradication group was 94.7% ± 5.5% at 4 weeks, which was similar to that in the post-ESD eradication group (94.7% ± 6.7%, P = 0.987), irrespective of CYP2C19 genotype. In multivariate analyses, location of gastric tumor (middle and upper, odds ratio: 4.05, 95% CI: 1.620-10.230, P = 0.003) was a factor for 97% reduction of artificial ulcer area at 4 weeks post-ESD, but CYP2C19 genotype and H. pylori infection were not. CONCLUSION: Healing speed of ESD-induced artificial ulcer was affected by tumor location, but not by time of H. pylori eradication, resected size, or CYP2C19 genotype.

Primary synovial sarcoma of the stomach--a case report and review of the literature.

Synovial sarcoma (SS) is a mesenchymal spindle cell tumor which displays variable epithelial differentiation. It commonly arises around the major joints or tendon sheaths in young adults, but is not commonly seen in the stomach. We experienced a case of primary gastric SS. The patient is a 22-year-old male, who presented with epigastric pain. Upper endoscopy showed an ulcer of 25 mm in diameter with marginal elevation on the posterior mid-gastric body. Biopsy of the ulcer base showed monotonous proliferation of small spindle-shaped cells on HE-stain. On immunohistochemical staining, these cells were positively stained with vimentin, cytokeratin, epithelial membrane antigen, and CD99, but were negative for KIT, CD34, desmin, and S-100 protein. These findings were compatible with SS of monophasic type. Diagnosis of primary gastric SS was made because there were no other primary lesions, nor metastatic lesions. The wedge resection was performed. Reverse transcriptase polymerase chain reaction (RT-PCR), using the RNA from frozen neoplastic tissue of the resected specimen, detected a fusion gene called SYT-SSX1, specific for SS. Though SS arising in the stomach is rare, it should be considered in the differential diagnosis of KIT-negative gastric spindle cell tumor.

Thiopurine maintenance therapy for ulcerative colitis: the clinical significance of monitoring 6-thioguanine nucleotide.

BACKGROUND: 6-Mercaptopurine (6-MP) is an effective maintenance medication in patients with ulcerative colitis (UC), but toxic effects like myelosuppression limit its clinical benefit. In the blood, 6-thioguanine (6-TGN) is formed from 6-MP and mediates the therapeutic efficacy and most of the toxicities of 6-MP. The level of 6-TGN depends on the activity of thiopurine methyltransferase (TPMT), inherited as 1 of its 3 polymorphic forms with low, moderate, or normal/high activity. Accordingly, the 6-MP dose needs to be pharmacogenetically guided. METHODS: Patients with quiescent UC received 6-MP as maintenance therapy and 6-TGN was assayed as its concentrations in red blood cells (RBCs) done by high-performance liquid chromatography. In a preliminary investigation, 30 mg/day 6-MP (n = 50) was given orally over 12 weeks to determine the time course of blood 6-TGN level. Then 257 patients were given 6-MP at 15-80 mg/day in a stepwise manner based on RBC 6-TGN, white blood cell count, and body weight to monitor 6-MP efficacy and safety profiles. RESULTS: At 30 mg/day 6-MP, RBC 6-TGN peaked over 4-8 weeks. In the main dosing study, the mean RBC 6-TGN level in patients who remained in remission during the 1-year observation time (n = 151) was 322.3 +/- 119.5 pmole/8 x 10(8) RBC versus 204.8 +/- 78.7 pmole/8 x 10(8) RBC in patients (n = 19) who relapsed (P < 0.001). Bone marrow suppression was seen almost exclusively at high 6-TGN concentration ranges. Further, a regression plot showed an inverse relationship between 6-TGN levels in RBC and TPMT enzyme activity. CONCLUSIONS: By regularly measuring RBC 6-TGN in patients with quiescent UC receiving 6-MP as maintenance therapy, we could monitor bone marrow suppression as well as other toxic side effects. Potentially, this strategy should enable physicians to avoid thiopurine-related adverse effects and identify individuals who may benefit most from 6-MP maintenance therapy.

Hepatocellular carcinoma with mesothelioma-like dissemination.

Reported herein is a case of hepatocellular carcinoma (HCC) with unusual peritoneal dissemination masquerading as peritoneal mesothelioma. A 61-year-old man was clinically found to have multiple tumors in his abdominal cavity; peritonitis carcinomatosa was suspected. An autopsy revealed numerous tumors of various sizes in the abdominal serosa, omentum, and diaphragm. No signs of tumor, fibrosis, or cirrhosis were found in the liver, except for a small nodule in the hepatic triangular ligament. Histologically, the tumor cells proliferated in thick trabeculae or in sheets and formed a few canaliculi and tubules with homogenously brown contents in their lumina, which stained positively with Hall stain. Immunohistochemically, these tumors were positive for hepatocyte, alpha-fetoprotein (AFP) and low-molecular-weight cytokeratin; were focally positive for pan-cytokeratin and epithelial membrane antigen (EMA); and were negative for high-molecular-weight cytokeratin, vimentin, and calretinin. Carcinoembryonic antigen (CEA) produced a bile canalicular immunohistochemical staining pattern. Thus, the tumor was diagnosed as an HCC (Edmondson II type) of the triangular ligament with massive peritoneal dissemination. The origin of this tumor and its differential diagnosis (malignant mesothelioma, hepatoid adenocarcinoma, and hepatoid yolk sac tumor) are discussed.

Refractory ulcerative colitis complicated by a cytomegaloviral infection requiring surgery: report of a case.

Cytomegalovirus (CMV) infection has been reported to be a cause of refractory ulcerative colitis (UC). We herein report a case of refractory ulcerative colitis complicated by CMV infection requiring surgery. A 22-year-old man was admitted to our hospital with lower abdominal pain and bloody diarrhea. Under a diagnosis of acute UC, he was treated with prednisone 60 mg/day and sulfasalazine. Since his symptoms appeared to improve, the prednisone dosage was gradually reduced to 20 mg/day. After 5 months, he had an unexpected flare-up with fever and fresh anal bleeding. Colonoscopy demonstrated a punched out ulcer in the sigmoid colon. Biopsies by colonoscopy revealed cytomegalic inclusion bodies. Serologic and immunologic studies also suggested a recent CMV infection. Under a diagnosis of intractable UC complicated by a CMV infection, ganciclovir therapy was carried out, and the steroid therapy was tapered. Although the serum antigenemia became negative after the antiviral therapy, follow-up colonoscopy confirmed the severe stenosis after the punched-out ulcer healed completely. Since his symptoms did not improve, it was necessary to perform an elective proctocolectomy despite antiviral therapy. He was discharged with an uneventful postoperative course. It is important to recognize CMV colitis as a complication of inflammatory bowel disease, particularly in severe steroid-resistant colitis. Furthermore, in cases which fail to respond to antiviral treatment, the patient may ultimately require surgery.

[Clinical significance of faecal calprotectin levels in patients with ulcerative colitis].

AIMS: To assess the clinical significance of faecal calprotectin levels (a neutrophil protein) in patients with ulcerative colitis (UC). METHODS: 25 patients with UC provided stool samples for calprotectin assay and the amount of calprotectin was related to UC disease activity index in each patient. Of 25 patients 4 with prednisolone refractory UC received 10 granulocyte and monocyte adsorption apheresis (GMCAP) sessions of 60 minutes duration, flow rate 30 mL per minute for 10 consecutive weeks. RESULTS: Calprotectin level in consecutive faecal samples from three patients was stable. However, increased calprotectin levels were significantly (p < 0.005) associated with Matts's endoscopic index, reflecting the level of colorectal inflammation. The 4 patients who received GMCAP therapy had a clinical activity index < 2 at week 7, the calprotectin level declined with improving Matts' index. CONCLUSIONS: Assay of faecal calprotectin holds promise as a sensitive biomarker to identify colorectal inflammation.

Immunohistochemical localization of angiotensin II receptor and local renin-angiotensin system in human colonic mucosa.

Angiotensin II (Ang II) regulates water and sodium transport in renal tubules and gastrointestinal tract. Two types of Ang II receptors have been cloned, but their distributions have not been determined in human colon. In addition, tissue renin-angiotensin systems (RAS) are believed to exist and to regulate local actions in human colon. We studied by reverse transcription-polymerase chain reaction (RT-PCR) and immunohistochemistry (IHC) the presence and localization of Ang II receptors Type 1 (AT(1)), Type 2 (AT(2)), and RAS components [angiotensinogen, renin, and angiotensin-converting enzyme (ACE)] in normal human colon. AT(1) receptors were localized in vessel walls, myofibroblasts, macrophages, and surface epithelium. AT(2) receptors were found in mesenchymal cells and weakly in parts of surface epithelium. Renin and ACE were distributed in vessel walls, mesenchymal cells, and in parts of surface epithelium. Angiotensinogen was also detected by RT-PCR. These findings demonstrated that Ang II receptors and RAS components were present in human colon, suggesting the possibility of its local regulation.