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J Med Microbiol 55 (2006), 1587-1590; DOI: 10.1099/jmm.0.46722-0
© 2006 Society for General Microbiology
ISSN 1473-5644

Case Report

Intractable colitis associated with chronic granulomatous disease

Yoshiaki Arimura1, Akira Goto1, Kentaro Yamashita1, Takao Endo1, Hideyuki Ikeda2, Kaori Tanaka3, Hiroyuki Tsutsumi3, Yasuhisa Shinomura1 and Kohzoh Imai1

First Department of Internal Medicine1 , Department of Surgical Pathology2 and Department of Pediatrics3 , Sapporo Medical University, S-1, W-16, Chuo-ku, Sapporo 060-8543, Japan

Correspondence
Yoshiaki Arimura
arimura{at}sapmed.ac.jp

Received 15 May 2006
Accepted 13 July 2006

The case of a 20-year-old Japanese man, diagnosed as having autosomal recessive chronic granulomatous disease (CGD), who was being treated with corticosteroids for intractable unclassified colitis, is described. He died from multiple organ failure following disseminated intravascular coagulation secondary to disseminated varicella-zoster virus (VZV) infection. He was diagnosed as an index case of CGD when 2 years old, was inoculated against VZV at the age of 5 years and had had an unremarkable course for 19 years. He was admitted to hospital because of a third episode of recurrent bloody diarrhoea. Clinical remission for each episode was achieved by intravenous corticosteroid therapy. Unclassified colitis associated with CGD was diagnosed based on a colonic biopsy demonstrating characteristic macrophages with lipofuscin deposits. From a treatment viewpoint, idiopathic inflammatory bowel disease (IBD) should be differentiated from secondary IBD occurring in CGD, in which immunosuppressive drugs including corticosteroids, still the mainstay of IBD treatment, should be avoided.

Abbreviations: CD, Crohn's disease; CGD, chronic granulomatous disease; GI, gastrointestinal; IBD, inflammatory bowel disease; UC, ulcerative colitis.




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Important role of corticosteroids in chronic granulomatous disease
J. Med. Microbiol., September 1, 2007; 56(9): 1253 - 1253.
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Y. Arimura, A. Goto, K. Yamashita, T. Endo, H. Ikeda, K. Tanaka, H. Tsutsumi, Y. Shinomura, and K. Imai
Authors' reply to 'Important role of corticosteroids in chronic granulomatous disease'
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