Reduced antiviral seropositivity among patients with inflammatory bowel disease treated with immunosuppressive agents.

BACKGROUND: Although live-attenuated vaccines are contraindicated under immunosuppression, the immune status of patients with inflammatory bowel disease (IBD) has not been fully assessed prior to immunosuppressive therapy. AIMS: To investigate antiviral serostatus against viruses requiring live vaccines for prevention in IBD patients undergoing immunosuppressive therapy. METHODS: This multicenter study included IBD patients who were aged <40 years and were treated with thiopurine monotherapy, molecular-targeted monotherapy, or combination therapy. Gender- and age-matched healthy subjects (HS) living in the same areas were included as control group. Antibody titers against measles, rubella, mumps, and varicella were measured by enzyme-linked immunosorbent assays. RESULTS: A total of 437 IBD patients (163 ulcerative colitis [UC] and 274 Crohn's disease [CD]) and 225 HS were included in the final analysis. Compared with HS, IBD patients had lower seropositivity rates for measles (IBD vs. HS = 83.91% vs. 85.33%), rubella (77.55% vs. 84.89%), mumps (37.50% vs. 37.78%), and varicella (91.26% vs. 96.44%). Gender- and age-adjusted seropositivity rates were lower in UC patients than in both CD patients and HS for measles (UC, CD, and HS = 81.60%, 85.29%, and 85.33%), rubella (76.40%, 78.23%, and 84.89%), mumps (27.16%, 43.70%, and 37.78%), and varicella (90.80%, 91.54%, and 96.44%); the difference was significant for all viruses except measles. Divided by the degree of immunosuppression, there were no significant differences in seropositivity rates among IBD patients. CONCLUSIONS: IBD patients, especially those with UC, exhibit reduced seropositivity rates and may benefit from screening prior to the initiation of immunosuppressive therapy.

A Case of Intussusception Associated with Pneumatosis Cystoides Intestinalis.

Pneumatosis cystoides intestinalis (PCI) is an uncommon disease that generally lacks symptoms and is rarely associated with intussusception. A 29-year-old man visited our hospital for right upper abdominal pain. Computed tomography (CT) scan revealed multiple air-filled cysts along the intestinal wall and a pseudokidney sign in the transverse colon. A gastrographin enema examination showed a so-called crab finger appearance and multiple elevated translucency in the transverse colon. From these findings, the diagnosis of intussusception associated with PCI was made. The enema and manipulative reduction improved the intussusception. Comparing the enema findings before and after the reduction, we thought that mobile cecum could play an important role in the intussusception. Colonoscopy was performed after the reduction and showed multiple elevated lesions in the ascending colon, which were similar to cluster of grapes. The CT scan of the next day revealed no recurrence of the intussusception, and the patient has not had symptoms of recurrence ever since.

Refractory sclerosing mesenteritis involving the small intestinal mesentery: a case report and literature review.

A 28-year-old Japanese man presented with upper abdominal pain. Computed tomography (CT) revealed a soft tissue mass in the small bowel mesentery. We diagnosed the patient with sclerosing mesenteritis according to the histological findings of small bowel mesentery. Although he was treated with prednisolone, colchicine and azathioprine, neither his symptoms nor CT findings improved. This case is rare in that the disease was refractory. The characteristics of Japanese patients with sclerosing mesenteritis involving small bowel mesentery are not well understood. We herein describe the details of such patients based on a literature review including 32 recently reported Japanese cases.

A case report of segmental arterial mediolysis in which computed tomography angiography was useful for diagnosis.

A 48-year-old male presented to our hospital with abdominal pain. Laboratory studies showed no abnormality, the severity of his abdominal pain decreased, and the patient was discharged. Five days later, the patient visited a neighborhood clinic because of fever with a 3-day history of temperatures of approximately 38 °C. The patient was admitted to our hospital 6 days after his initial visit. Laboratory investigation revealed a C-reactive protein level of 18.2 mg/dL. Abdominal computed tomography (CT) showed an 80 × 60 mm hematoma behind the descending colon, but no extravasation was detected. Thin-slice maximum-intensity-projection images from CT angiography (CTA) showed irregular narrowing and intermittent fusiform dilatations of the left colonic artery, suggesting a vascular disease, such as segmental arterial mediolysis (SAM). Digital subtraction angiography showed local irregularity, and 'beading and narrowing' of the left colonic artery, similar to the findings on CTA. Left hemicolectomy was electively performed on the twenty-fifth hospital day. Histological findings were consistent with SAM. Thus, CTA was a useful modality for the early diagnosis of SAM.

[A case of calcineurin-inhibitor induced pain syndrome associated with tacrolimus therapy for ulcerative colitis].

A 23-year-old woman was admitted with a relapse of ulcerative colitis. Tacrolimus therapy was initiated following inadequate response to corticosteroid therapy. Although the symptoms partially improved, she suddenly developed severe pain localized to the lower limbs on day 16 of tacrolimus therapy. By day 17, she was unable to move. Magnetic resonance imaging revealed born marrow edema in the lower limbs. We suspected calcineurin-inhibitor induced pain syndrome (CIPS) due to tacrolimus therapy. The pain improved within approximately four weeks of tacrolimus cessation. CIPS that is not associated with organ transplantation is a rare occurrence. Here we report a rare case of CIPS that was caused by tacrolimus therapy in a patient with ulcerative colitis.

[A case of ascending colon neuroendocrine carcinoma drained through a fistula of the descending part of the duodenum].

A 70-year-old woman was admitted for investigation of an abdominal tumor. Abdominal CT revealed an ascending colonic mass measuring 10 × 10 cm, with evidence of liver and lung metastasis. Colonoscopy revealed a cancerous lesion with a central ulcer in the ascending colon. Upper gastrointestinal endoscopy revealed an ulcerative lesion in the descending part of the duodenum. Histologically, the tumor showed features of neuroendocrine carcinoma. The patient died of the primary cancer two and a half months after admission. Autopsy revealed a fistula connecting the ascending colonic mass with the ulcerative lesion in the duodenum.

Flare up of ulcerative colitis during pregnancy treated by adsorptive granulocyte and monocyte apheresis: therapeutic outcomes in three pregnant patients.

PURPOSE: Treatment of ulcerative colitis with drugs during pregnancy potentially may harm the mother and the unborn child. Granulocytapheresis depletes elevated/activated myeloid lineage leucocytes as sources of inflammatory cytokines. We were interested in the safety and efficacy of granulocytapheresis in patients who had ulcerative colitis flare up during pregnancy. METHODS: Three pregnant cases with active ulcerative colitis received Adacolumn granulocytapheresis, up to 10 sessions within 3-6 weeks. Case 1: a 33-year-old woman with left-sided colitis and bloody diarrhoea 7-9 times/day showed loss of mucosal vascular patterns, and contact bleeding from the rectum to the sigmoid colon. Case 2: a 36-year-old woman with pancolitis and bloody diarrhoea 6-8 times/day had loss of mucosal vascular patterns and pus from the rectum to the sigmoid colon. Case 3: a 36-year-old woman with pancolitis and diarrhoea 4-5 times/day (first episode) had erosions and pus in the mucosa from the rectum to the transverse colon. RESULTS: Colitis flare was in weeks 5, 13 and 22 of pregnancy in cases 1, 2, 3, respectively. The corresponding granulocytapheresis sessions were 5, 7, and 10, reflecting an increasing trend with the pregnancy week. Patients 1 and 2 achieved complete remission, patient 3 achieved clinical remission. CONCLUSION: In these three cases with active ulcerative colitis during pregnancy, granulocytapheresis as a non-pharmacologic treatment was effective and safe. In case 3 that did not respond well to the initial granulocytapheresis sessions, a moderate dose of prednisolone enhanced the efficacy of granulocytapheresis and tapering of prednisolone shortly after administration was not associated with relapse.

[Successful treatment of a case of advanced sigmoid colon cancer with occlusion of the common celiacomesenteric trunk].

A 74-year-old man was admitted to our hospital with abdominal pain and bloody stool. The patients' history showed that he had had occlusion of the proximal common trunk of the celiac artery (CA) and the superior mesenteric artery (SMA). The inferior mesenteric artery (IMA), and the marginal artery of the colon had developed well. It was assumed that almost the entire visceral blood might be supplied by the IMA to the CA and the SMA. Our investigation revealed that the patient had advanced cancer of the sigmoid colon, which had caused intestinal obstruction. Sigmoidectomy was performed with care to avoid injuring the IMA and the marginal arcade artery. Normal hemodynamics were successfully established followed by sigmoidectomy, and cure was obtained in this patient.

Gastric cancer following total proctocolectomy for colon cancer associated with ulcerative colitis.

Here we report a case of advanced gastric cancer seen after total proctocolectomy for an early colon cancer associated with ulcerative colitis (UC). A 42-year-old man, diagnosed with UC at the age of 21, had undergone total proctocolectomy at the age of 38 for an early ascending colon cancer. Three years later the patient developed tarry stools and epigastric discomfort. Laboratory data showed anemia together with elevated serum p53 antibody. Gastric endoscopy showed thickening folds around a lesion in the stomach body. The pathological diagnosis was poorly differentiated adenocarcinoma with signet-ring cell carcinoma. Total gastrectomy was performed and the resected specimens showed a diffuse infiltrating tumor (scirrhous gastric carcinoma), 11 × 15 cm in size, with multiple lymph node metastases. Histopathological examination revealed diffuse infiltration of cancer cells throughout the gastric wall and invasion of the serosa. Results of cytology on abdominal lavage were positive for cancer cells. Likewise, immunohistochemical staining showed gastric mucin phenotype cancer cells positive for p53. In conclusion, it is important to bear in mind that patients with UC, especially chronically active pancolitis, potentially bear the risk of upper gastrointestinal complications.

[A case report of Ewing's sarcoma with a peripheral primitive neuroectodermal tumor (ES/pPNET) in the abdominal cavity].

In this report, we present a rare case of Ewing's sarcoma with a peripheral primitive neuroectodermal tumor (ES/pPNET) arising from the abdominal cavity in a 20-year-old woman. The patient complained of upper abdominal pain. Radiological imaging showed a 15-cm mass penetrating to the proxymal jejunum in the upper abdominal cavity and peritoneal disseminations. Immunohistochemical studies revealed that the tumor was ES/pPNET. Although the patient underwent radiation therapy, she died of the disease two months after diagnosis. ES/pPNET in the abdominal cavity is extremely rare and our case showed aggressive behavior and an unfortunate outcome.

Lack of association between IBD5 and Crohn's disease in Japanese patients demonstrates population-specific differences in inflammatory bowel disease.

OBJECTIVE: Population-specific differences in the genetic susceptibility to inflammatory bowel disease (IBD) are indicated by the fact that Crohn's disease (CD) in Japanese patients does not have any of the common CARD15 variants that are associated with CD in Caucasians. Recently, the disease-causing mutation in the IBD5 haplotype was identified. The TC haplotype, composed of L503F in SLC22A4 and -207G/C in SLC22A5 promoters, was reported to alter the function of the organic cation transporter and to be associated with CD in Caucasians. To determine whether the TC haplotype is also associated with IBD in a Japanese population, we genotyped L503F and -207G/C variants in Japanese subjects. Furthermore, we also performed a case-control association study with all representative single nucleotide polymorphisms (SNPs) in IBD5 using previous information of linkage disequilibrium extension reported in Japanese patients to determine whether there were variants in IBD5 specifically associated with IBD in Japanese patients. MATERIAL AND METHODS: A total of 758 Japanese individuals, 241 patients with CD, 247 patients with ulcerative colitis (UC) and 270 healthy controls, were analyzed in this study. Genotyping for SNPs was determined by polymerase chain reaction-restriction fragment length polymorphism analysis. RESULTS: We found L503F and -207G/C to be very rare (<1% frequency) in CD, UC and HC in the Japanese population. Furthermore, we also found that none of the representative SNPs in IBD5 was associated with CD or UC in the Japanese subjects. CONCLUSIONS: In contrast to Caucasians, IBD5 is not a major component of the susceptibility to IBD in the Japanese population.