Blood 2003 May 15;101(10):3857-3861
Rituximab for the treatment of refractory autoimmune hemolytic anemia in children.
Zecca M, Nobili B, Ramenghi U, Perrotta S, Amendola G, Rosito P, Jankovic M, Pierani P, De Stefano P, Bonora MR, Locatelli F.
Oncoematologia Pediatrica, IRCCS Policlinico San Matteo, Pavia, Italy; Clinica Pediatrica I, Seconda Universita degli Studi, Napoli, Italy; Clinica Pediatrica, Universita di Torino, Ospedale Regina Margherita, Torino, Italy; Ematologia-Oncologia Pediatrica, Ospedale di Nocera Inferiore, Italy; Clinica Pediatrica, Universita di Bologna, Policlinico Sant'Orsola, Bologna, Italy; Clinica Pediatrica, Universita di Milano Bicocca, Nuovo Ospedale San Gerardo, Monza, Italy; Clinica Pediatrica, Universita di Ancona, Italy; and Farmacologia Clinica, IRCCS Policlinico San Matteo, Pavia, Italy.
Autoimmune hemolytic anemia (AIHA) in children is sometimes characterized by a severe course, requiring prolonged administration of immunosuppressive therapy. Rituximab is able to cause selective in vivo destruction of B lymphocytes, with abrogation of antibody production. In a prospective study, we have evaluated the use of rituximab for the treatment of AIHA resistant to conventional treatment. Fifteen children with AIHA were given rituximab, 375 mg/m(2)/dose for a median of 3 weekly doses. All patients had previously received 2 or more courses of immunosuppressive therapy; 2 patients had undergone splenectomy. After completing treatment, all children received intravenous immunoglobulin for 6 months. Treatment was well tolerated. With a median follow-up of 13 months, 13 patients (87%) responded, whereas 2 patients did not show any improvement. Median hemoglobin levels increased from 7.7 g/dL to a 2-month posttreatment level of 11.8 g/dL (P <.001). Median absolute reticulocyte counts decreased from 236 to 109 x 10(9)/L (P <.01). An increase in platelet count was observed in patients with concomitant thrombocytopenia (Evans syndrome). Three responder patients had relapse, 7, 8, and 10 months after rituximab infusion, respectively. All 3 children received a second course of rituximab, again achieving disease remission. Our data indicate that rituximab is both safe and effective in reducing or even abolishing hemolysis in children with AIHA and that a sustained response can be achieved in the majority of cases. Disease may recur, but a second treatment course may be successful in controlling the disease.
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