Objective: To report the beneficial effects of thalidomide on recurrent perforating intestinal ulcers in a patient with Behçet's disease (BD).
Case summary: A 24-year-old Turkish woman with BD was admitted to our hospital because of severe abdominal pain and vomiting. She had been receiving colchicine 1.5 mg/day and azathioprine 150 mg/day for treatment of BD for 2 years. During emergency laparatomy, 2 perforating ulcers were detected in the anterior cecum, which were treated with debridement and primary repair. She experienced 2 more episodes of intestinal perforations during the second and fifth weeks despite intense immunosuppressive treatment with methylprednisolone and cyclophosphamide. New intestinal perforations were found in the posterolateral cecum and transverse colon during the second operation and in the terminal ileum during the third one. Thalidomide 100 mg/day was then started, and the symptoms disappeared within 2 weeks. The woman experienced no other intestinal perforation during the follow-up period of 4 months.
Discussion: The mode of action of thalidomide in BD is still unclear. In BD, various cytokines have been shown to be abnormally expressed and neutrophils are overactive. This is a possible mechanism of action with thalidomide reducing both tumor necrosis factor and the neutrophil migration.
Conclusions: Thalidomide may be an effective alternative treatment for BD patients with recurrent and perforating intestinal ulcers despite intense immunosuppressive therapy.