Autoimmune hemolytic anemia (AIHA) in children usually responds well to short-term steroid therapy. However, in some cases, AIHA requires prolonged immunosuppressive therapy, with the subsequent development of severe side effects. Compared with previous conventional immunosuppressive therapy, rituximab, an anti-CD20 chimeric monoclonal antibody, shows good therapeutic efficacy and safety in the treatment of autoimmune disorders. Herein, the case of a 13-year-old male patient, who showed a remarkable and durable response to rituximab, at a dose of 375mg/m2, is reported. Before this trial, he had been a hepatitis B carrier and steroid dependent AIHA for 4 years, with a cushingoid facial appearance and growth retardation. After 10 months, he experienced a recurrence of hemolysis, which was successfully retreated, and was then taken off the steroid therapy. The therapy was well tolerated, without serious complications. It is suggested that rituximab could be a new option in the treatment of steroid-dependent AIHA. Therefore, long-term follow-up and studies of the risk factors of a relapse are warranted.

Keywords: Autoimmune hemolytic anemia, Rituximab, Children