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Shu Kojima,1 Satohiro Matsumoto,1 Yudai Koito,1 Takaya Miura,1 Masanari Sekine,1 Takeshi Uehara,1 Takeharu Asano,1 Yasuhiro Yamaguchi,2 Hirosato Mashima1 1Department of Gastroenterology, Jichi Medical University Saitama Medical Center, Saitama, Japan; 2Department of Respiratory Medicine, Jichi Medical University Saitama Medical Center, Saitama, JapanCorrespondence: Shu Kojima, Department of Gastroenterology, Jichi Medical University Saitama Medical Center, 1-847 Amanuma, Omiya, Saitama, 330-8503, Japan, Tel +81 48 647 2111, Fax +81 48 648 5188, Email s.kojima@jichi.ac.jpAbstract: A 51-year-old Japanese man was diagnosed with left-sided ulcerative colitis (UC) at age 41. He was treated with mesalazine and azathioprine and maintained remission. At age 51, the patient developed bloody stools, abdominal pain, scleritis, arthritis, cough, bloody sputum, and pericardial effusion. Considering that pericardial effusion is an atypical extraintestinal complication of UC, and the patient met the diagnostic criteria for relapsing polychondritis (RP), a diagnosis of RP complicating a relapse of UC was made. Steroid therapy was administered, and both diseases improved. Golimumab, an anti-tumor necrosis factor-α inhibitor, was introduced as maintenance therapy for UC. All symptoms, including pericardial effusion, improved. Subsequently, no relapse of UC or RP was observed. As only a few cases of RP overlapping with UC have been reported and no treatment protocol has been established, we considered this case valuable and worthy of publication.Keywords: ulcerative colitis, relapsing polychondritis, anti-tumor necrosis factor-α inhibitor, golimumab

Sota Katsube,1 Satohiro Matsumoto,1 Masahiro Misawa,2 Nao Kakizawa,3 Ryo Hashimoto,1 Taku Mizutani,1 Keita Matsumoto,1 Shuhei Yoshikawa,1 Hirosato Mashima1 1Department of Gastroenterology, Jichi Medical University Saitama Medical Center, Saitama, Japan; 2Department of Obstetrics and Gynecology, Jichi Medical University Saitama Medical Center, Saitama, Japan; 3Department of General and Gastrointestinal Surgery, Jichi Medical University Saitama Medical Center, Saitama, JapanCorrespondence: Sota Katsube, Department of Gastroenterology, Jichi Medical University Saitama Medical Center, 1-847 Amanuma, Saitama, Omiya, 330-8503, Japan, Tel +81 48 647 2111, Fax +81 48 648 5188, Email m08029sk@jichi.ac.jpAbstract: The patient was a 50-year-old Japanese woman who was diagnosed with total-colitis-type ulcerative colitis (UC) at the age of 26 years. She was treated with mesalazine and azathioprine, and her disease activity was well controlled. At the age of 50 years, the patient was experiencing fever, abdominal pain, diarrhea, bloody stool, and anal pain, which led to a diagnosis of a relapse of UC. Although steroid therapy was administered and tended to improve her symptoms, fecaloid vaginal discharge occurred, and rectovaginal fistula (RVF) was confirmed. Colostomy was performed, and infliximab was initiated as maintenance therapy for UC. All symptoms improved, and RVF closure was confirmed 6 months after the initiation of infliximab. To date, she has been free from relapse of UC. There have been only a few reports of UC complicated by RVF, and this condition is often difficult to treat. To the best of our knowledge, no other case of UC complicated by RVF in which the fistula was closed after treatment with colostomy and infliximab has been previously reported; thus, our report of the present case is valuable to the literature.Keywords: ulcerative colitis, rectovaginal fistula, infliximab, colostomy

Hitomi Kashima, Satohiro Matsumoto, Shu Kojima, Yudai Koito, Takaya Miura, Takehiro Ishii, Hirosato Mashima Department of Gastroenterology, Jichi Medical University Saitama Medical Center, Saitama, JapanCorrespondence: Satohiro MatsumotoDepartment of Gastroenterology, Jichi Medical University Saitama Medical Center, 1-847 Amanuma, Omiya, Saitama, Saitama, 330-8503, Japan, Tel +81 48 647 2111, Fax +81 48 648 5188, Email s.w.himananon@ac.auone-net.jpAbstract: A 55-year-old man presented with recurrent ulcers and an enterocutaneous fistula at the anastomotic site after surgery for an ileovesical fistula and was diagnosed with intestinal Behçet’s disease after undergoing surgery for enterocutaneous fistulae twice. The patient was transferred to our hospital because of recurrent enterocutaneous fistulae. He had a history of recurrent oral aphthous ulcers, folliculitis, and epididymitis and met the diagnostic/classification criteria for incomplete Behçet’s disease and thus was diagnosed as having intestinal Behçet’s disease. Remission induction therapy with steroids was administered for an ileal ulcer and an enterocutaneous fistula, and adalimumab was initiated for maintenance therapy. The fistula was closed, and the clinical course was favorable. Two months after initiating adalimumab, a subcutaneous abscess was detected at the site of the enterocutaneous fistula scar, and relapse of intestinal Behçet’s disease was suspected. Steroids were re-administered for remission induction, followed by maintenance therapy, for which adalimumab was switched to infliximab. No relapse was detected after steroid withdrawal. No therapeutic strategies have been established for intestinal Behçet’s disease. Moreover, there have been very few reports on therapeutic strategies and postoperative maintenance therapy for enterocutaneous fistulae. We thus consider this case valuable.Keywords: intestinal Behçet’s disease, adalimumab, infliximab, enterocutaneous fistulae

Satohiro Matsumoto, Keita Matsumoto, Hiromu Takaya, Haruka Otake, Hirosato Mashima Saitama Medical Center, Jichi Medical University, Saitama, Saitama, JapanCorrespondence: Satohiro Matsumoto Email s.w.himananon@ac.auone-net.jpAbstract: Spondyloarthritis (SpA) is a known extraintestinal complication in inflammatory bowel disease (IBD). However, since the prevalence of SpA is lower in Japan than in Europe, some patients may be inaccurately diagnosed and treated. Although non-steroidal anti-inflammatory drugs (NSAIDs) remain the mainstay treatment for IBD-related SpA, anti-tumor necrosis factor-α antibody and ustekinumab have demonstrated efficacy in patients with SpA refractory to NSAIDs. We report here a case of Crohn’s disease with SpA, as an extraintestinal manifestation, in which ustekinumab proved extremely effective, not only for alleviating the arthritis but also against skin manifestations and scleritis. Only a few studies have documented the therapeutic effects of ustekinumab against SpA associated with IBD; therefore, its efficacy remains unclear.Keywords: ustekinumab, spondyloarthritis, Crohn’s disease