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PEDIATRICS Vol. 103 No. 6 June 1999, pp. 1295-1297

EXPERIENCE AND REASON:
Thalidomide Responsiveness in an Infant With Behçet's Syndrome

Received May 7, 1998; accepted Nov 25, 1998.

Lynette Pei-Chi Shek*, Yoke Sun Lee MDDagger , and Bee Wah Lee*

* Departments of Pediatrics and Dagger  Pathology National University of Singapore Singapore 119074

Thomas Joseph Anjorge Lehman

Division of Pediatric Rheumatology Hospital for Special Surgery, New York 10021

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.  Key words:  vasculitis, mouth ulcers, Behçet's syndrome, thalidomide.


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