Infliximab therapy for Crohn’s-like disease in common variable immunodeficiency complicated by massive intestinal hemorrhage: a case report

BMC Research Notes

Kazuhiko Nakao¹  Masahiro Nakashima³  Tomayoshi Hayashi⁴  Hajime Isomoto¹  Ken Ohnita¹  Naoyuki Yamaguchi¹  Hitomi Minami¹  Kayoko Matsushima¹  Tsutomu Kanda¹  Daisuke Imanishi²  Hiroyuki Yajima¹  Fuminao Takeshima¹  Yuko Akazawa³ 

[1] Department of Gastroenterology and Hepatology, Nagasaki University Hospital, 1-7-1 Sakamoto, 852-8501 Nagasaki, Japan;Department of Haematology, Nagasaki University Hospital, Nagasaki, Japan;Department of Tumor and Diagnostic Pathology, Atomic Bomb Disease Institute, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, Japan;Department of Pathology, Nagasaki University Hospital, Nagasaki, Japan

关键词: Colitis;    T lymphocyte;    Inflammatory bowel disease;    Common variable immunodeficiency;   

Others  :  1132410
DOI  :  10.1186/1756-0500-7-382

 received in 2014-02-04, accepted in 2014-06-19,  发布年份 2014

【 摘 要 】

Background

Common variable immune deficiency is the most frequently encountered immunodeficiency in adults, which is characterized by low levels of serum immunoglobulins. Common variable immune deficiency can present with inflammatory bowel disease-like colitis because of the dysregulated immune system; paradoxically activated T cell receptor pathways are thought to be pivotal in pathogenesis of common variable immune deficiency-related colitis. Treatment for severe complications, such as gastrointestinal bleeding, is not established. We report a case of common variable immune deficiency-related Crohn’s-like disease presenting massive melena, which was successfully treated by short course infliximab therapy.

Case presentation

A 26-year-old Japanese man with history of common variable immune deficiency presented with diarrhea, abdominal pain, and fever. Venous administration of antibiotics did not improve his symptoms. Colonoscopy revealed multiple longitudinal ulcers as well as cobblestone-like change in the ileum end and the ascending colon. Histopathological examination of biopsy specimen showed erosion and infiltration of T lymphocytes with lack of B cells. Intravenous hyperalimentation, mesalazine, and steroid did not improve the symptoms and the patient subsequently presented with massive melena. Colonoscopy revealed a protuberant vessel on one of the ulcers in the ascending colon. Endoscopic clipping was repeatedly performed for hemostasis, which was only temporarily successful. In an attempt to manage the bleeding and colitis, a trial of infliximab was given on week 0, week 2 and week 6. Gastrointestinal hemorrhage from the ulcer halted immediately after the first infliximab injection. Colonoscopy performed after the third infliximab showed remarkable improvement in the ileocolitis. No evidence of increased susceptibility to infections was observed and the patient has been in clinical remission for 3 years.

Conclusions

We present this case together with review of literature to share our experience of encountering common variable immune deficiency complicating severe Crohn’s-like disease and to support that infliximab is a safe and effective treatment that can promptly manage life-threatening intestinal hemorrhage in common variable immune deficiency-related colitis.

【 授权许可】

   
2014 Akazawa et al.; licensee BioMed Central Ltd.

【 图 表 】

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