Rituximab therapy produced rapid and sustained clinical improvement in a patient with systemic onset juvenile idiopathic arthritis refractory to TNF alpha antagonists.

We describe a patient with systemic onset juvenile idiopathic arthritis refractory to disease-modifying antirheumatic drugs, intravenous gamma globulin, and TNF inhibitors (etanercept and infliximab), in whom treatment with rituximab resulted in remission of systemic symptoms (rash and fever), a fall in erythrocyte sedimentation rate, C-reactive protein, and ferritin serum levels, with recovery in disability index and improvement of arthritis. A total of 4 cycles of rituximab were given over 18 months because of relapses. Since her last course, she remains stable and asymptomatic. To our knowledge, this is the first case reported on a long-lasting beneficial effect of rituximab in a patient with soJIA.