title: Infliximab therapy for Crohn's-like disease in common variable immunodeficiency complicated by massive intestinal hemorrhage: a case report.
abstract: 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.
  
  
  METHODS
  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.
authors: Department of Gastroenterology and Hepatology, Nagasaki University Hospital, 1-7-1 Sakamoto, 852-8501 Nagasaki, Japan. akazaway@nagasaki-u.ac.jp.
authors: Yuko
authors: Y
authors: Akazawa
authors: Fuminao
authors: F
authors: Takeshima
authors: Hiroyuki
authors: H
authors: Yajima
authors: Daisuke
authors: D
authors: Imanishi
authors: Tsutomu
authors: T
authors: Kanda
authors: Kayoko
authors: K
authors: Matsushima
authors: Hitomi
authors: H
authors: Minami
authors: Naoyuki
authors: N
authors: Yamaguchi
authors: Ken
authors: K
authors: Ohnita
authors: Hajime
authors: H
authors: Isomoto
authors: Tomayoshi
authors: T
authors: Hayashi
authors: Masahiro
authors: M
authors: Nakashima
authors: Kazuhiko
authors: K
authors: Nakao
mesh_terms: D000328:Adult; D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D017074:Common Variable Immunodeficiency / Q000150:complications*; D003424:Crohn Disease / Q000188:drug therapy* / Q000209:etiology; D005765:Gastrointestinal Agents / Q000627:therapeutic use; D006471:Gastrointestinal Hemorrhage / Q000209:etiology / Q000517:prevention & control*; D006801:Humans; D000069285:Infliximab; D016896:Treatment Outcome
chemical_list: D000911:Antibodies, Monoclonal; D005765:Gastrointestinal Agents; D000069285:Infliximab