Korean J Hematol.  2006 Dec;41(4):321-325. 10.5045/kjh.2006.41.4.321.

A Case of Autoimmune Hemolytic Anemia Treated with Rituximab in a Child

• Lee JH
• Lee KS

• KMID: 2305222
• DOI: http://doi.org/10.5045/kjh.2006.41.4.321

Abstract

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.

Keyword

Autoimmune hemolytic anemia; Rituximab; Children

MeSH Terms

Adolescent
Anemia, Hemolytic, Autoimmune*
Child*
Follow-Up Studies
Hemolysis
Hepatitis B
Humans
Male
Recurrence
Risk Factors
Rituximab

Figure

• 

  Fig. 1 The response of hemoglobin and corrected reticulocyte count before rituximab treatment. SPD, prednisolone; IVGLO, intravenous immunoglobulin O; So-lumedrol pulse, high dose methylprednisolone; C-Reti, corrected reticulocyte count.

• 

  Fig. 2 The hemoglobin and corrected reticulocyte count after rituximab treatment.

Reference

1). George BS. Hemolytic anemias resulting from extracellular factors. Behrman RE, Kliegman RM, Jenson HB, editors. Nelson textbook of pediatrics. 17th ed.Philadelphia: WB Saunders Co;2004. p. 1638–40.

2). Reff ME., Carner K., Chambers KS, et al. Depletion of B cells in vivo by a chimeric mouse human monoclonal antibody to CD20. Blood. 1994. 83:435–45.

3). McLaughlin P., Grillo-Lopez AJ., Link BK, et al. Rituximab chimeric anti-CD20 monoclonal antibody therapy for relapsed indolent lymphoma: half of patients respond to a four-dose treatment program. J Clin Oncol. 1998. 16:2825–33.

4). Yang DH., Lee JJ., Kim YK, et al. The clinical efficacy of R-CHOP chemotherapy in patients with previously untreated diffuse large B-cell lymphoma. Korean J Hematol. 2004. 39:59–65.

5). Lee EJ., Kueck B. Rituxan in the treatment of cold agglutinin disease. Blood. 1998. 92:3490–1.

6). Quartier P., Brethon B., Philippet P., Landman-Parker J., Le Deist F., Fischer A. Treatment of childhood autoimmune haemolytic anaemia with rituximab. Lancet. 2001. 358:1511–3.

7). Zecca M., Nobili B., Ramenghi U, et al. Rituximab for the treatment of refractory autoimmune hemolytic anemia in children. Blood. 2003. 101:3857–61.

8). Ramanathan S., Koutts J., Hertzberg MS. Two cases of refractory warm autoimmune hemolytic anemia treated with rituximab. Am J Hematol. 2005. 78:123–6.

9). Wang J., Wiley JM., Luddy R., Greenberg J., Feuer-stein MA., Bussel JB. Chronic immune thrombocytopenic purpura in children: assessment of rituixmab treatment. J Pediatr. 2005. 146:217–21.

10). Park SJ., Han CW. The long term remission effect of rituximab in two patients with autoimmune-associated cytopenias that were refractory to standard treatments. Korean J Hematol. 2005. 40:101–5.

11). Park US., Choi CB., Kim JS, et al. A case of post-transplantation lymphoproliferative disease developed in renal transplant recipient and treated with rituximab. Korean J Med. 2004. 67:94–9.

12). Heisel MA., Ortega JA. Factors influencing prognosis in childhood autoimmune hemolytic anemia. Am J Pediatr Hematol Oncol. 1983. 5:147–52.

13). Hamaki T., Kami M., Kusumi E, et al. Prophylaxis of hepatitis B reactivation using lamivudine in a patient receiving rituximab. Am J Hematol. 2001. 68:292–4.

14). Tsutsumi Y., Tanaka J., Kawamura T, et al. Possible efficacy of lamivudine treatment to prevent hepatitis B virus reactivation due to rituximab therapy in a patient with non-hodgkin's lymphoma. Ann Hematol. 2004. 83:58–60.