Thalidomide responsiveness in an infant with Behçet's syndrome

Pediatrics. 1999 Jun;103(6 Pt 1):1295-7. doi: 10.1542/peds.103.6.1295.

Abstract

Background: Behçet's syndrome is a chronic relapsing vasculitis characterized by aphthous stomatitis, genital ulcerations, and uveitis. The disease is a multisystem illness, which may involve the skin, joints, gastrointestinal tract, blood vessels, central nervous system, and other organs.

Case study: We report a case of an infant with features of Behçet's syndrome who did not respond to conventional immunosuppression with high-dose corticosteroids and cytotoxic agents. Thalidomide, a potent immune response-modifying drug, was used with marked improvement and resolution of symptoms.

Conclusion: This finding supports a trial of thalidomide treatment in patients with Behçet's syndrome who are unresponsive to other forms of immunosuppressive therapy or develop undesirable side effects.

Publication types

  • Case Reports

MeSH terms

  • Behcet Syndrome / drug therapy*
  • Female
  • Humans
  • Immunosuppressive Agents / therapeutic use*
  • Infant
  • Thalidomide / therapeutic use*
  • Treatment Outcome

Substances

  • Immunosuppressive Agents
  • Thalidomide