Rheumatology International

, Volume 33, Issue 12, pp 3105–3108

A young girl with refractory intestinal Behçet’s disease: a case report and review of literatures on pediatric cases who received an anti-tumor necrosis factor agent

  • Shojiro Watanabe
  • Tomomi Aizawa-Yashiro
  • Kazushi Tsuruga
  • Manabu Kinjo
  • Etsuro Ito
  • Hiroshi Tanaka
Short Communication

DOI: 10.1007/s00296-012-2628-5

Cite this article as:
Watanabe, S., Aizawa-Yashiro, T., Tsuruga, K. et al. Rheumatol Int (2013) 33: 3105. doi:10.1007/s00296-012-2628-5

Abstract

Behçet’s disease (BD) is a chronic, relapsing, multisystem inflammatory disorder classified as vasculitis and characterized by recurrent oral and genital ulcerations, uveitis, and protean clinical signs of skin, central nervous system, musculoskeletal, and gastrointestinal involvements. Among the protean manifestations due to BD, intestinal BD is often intractable, but effective treatment for intestinal BD has not been fully established. Tumor necrosis factor (TNF)-α plays a central role in inflammation in BD patients; however, there are very few reports on the successful treatment of pediatric-onset cases of intestinal BD using anti-TNF-α agents. We report the case of a 6-year-old Japanese girl with refractory intestinal BD who was successfully treated with multidrug therapy including etanercept (ETN). Also, we performed a review of literatures on pediatric cases who received an anti-TNF-α agent. To our knowledge, this is the youngest patient with intestinal BD who was successfully treated using ETN. Although further studies are needed to determine the efficacy and safety of anti-TNF-α agents in the treatment for pediatric-onset BD, we believe that even in very young patients with refractory BD, an anti-TNF-α agent may be beneficial.

Keywords

Behçet’s disease Etanercept Intestinal Behçet Multidrug therapy Pediatric-onset cases 

Copyright information

© Springer-Verlag Berlin Heidelberg 2012

Authors and Affiliations

  • Shojiro Watanabe
    • 1
  • Tomomi Aizawa-Yashiro
    • 1
  • Kazushi Tsuruga
    • 1
  • Manabu Kinjo
    • 2
  • Etsuro Ito
    • 1
  • Hiroshi Tanaka
    • 1
    • 3
  1. 1.Department of PediatricsHirosaki University HospitalHirosakiJapan
  2. 2.Section of PediatricsAomori Rosai HospitalHirosakiJapan
  3. 3.Department of School Health Science, Faculty of EducationHirosaki UniversityHirosakiJapan

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