Childhood autoimmune hemolytic anemia (AIHA) of the warm type is usually successfully managed with corticosteroids and/or immunoglobulin infusions. In a small proportion of patients AIHA follows a more severe and protracted pathway resulting in the use of immunosuppressive therapy and frequently culminating with the need for splenectomy. Rituximab is an anti-CD20 (B-cell) monoclonal antibody used for the treatment of patients with relapsed or refractory low-grade or follicular, CD20 positive, B-cell non-Hodgkin's lymphoma. Case reports on the use of rituximab for childhood AIHA are scant. The authors describe the first report in which rituximab was effectively employed to induce a long-term remission in a young child with the longest history of chronic relapsing AIHA prior to receiving rituximab. All immunosuppressive therapy was successfully discontinued and splenectomy was avoided.