Abstract
We describe our experience with a juvenile patient who had refractory intestinal Behcet's disease that responded to adalimumab, a fully humanized antibody against soluble TNF-α and its receptor. The patient, a 13-year-old girl, presented with oral aphthous ulcers, vulvar pain, and rashes on the lower extremities. She gradually developed a low-grade fever, abdominal pain, diarrhea, and hematochezia. Lower gastrointestinal endoscopy revealed ulcers in the terminal ileum, consistent with intestinal Behcet's disease. Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred. The symptoms resolved soon after the administration of adalimumab. Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation. This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease. Adalimumab is a good choice for intestinal Behcet's disease refractory to conventional treatment.
Highlights
Behcet’s disease is a chronic relapsing vasculitis that is characterized by recurrent oral aphthous and genital ulcers with uveitis [1]
We report on a juvenile patient with corticosteroid-dependent intestinal Behcet’s disease who responded to subcutaneous adalimumab injections
Because the clinical findings were corticosteroid-dependent, the patient was begun on adalimumab, a fully humanized anti-TNF-α monoclonal antibody
Summary
Behcet’s disease is a chronic relapsing vasculitis that is characterized by recurrent oral aphthous and genital ulcers with uveitis [1]. In addition to the main symptoms, there are some much less common manifestations, such as gastrointestinal or central nervous system disease. Pediatric Behcet’s disease occurs in approximately 0.3 cases per 100,000 amongst Japanese children [2]. The etiology of Behcet’s disease is still unknown, studies have revealed an association between tumor necrosis factor-alpha (TNF-α) and the clinical features, and the efficacy of some anti-TNF agents is reported in Behcet’s disease patients [3]. We report on a juvenile patient with corticosteroid-dependent intestinal Behcet’s disease who responded to subcutaneous adalimumab injections
Sign-in/Register to view highlights & summary Sign-in/Register to access full text options 
Free) 
Talk to us
Join us for a 30 min session where you can share your feedback and ask us any queries you have
Schedule a call
