Published online December 15, 2008
PEDIATRICS Vol. 123 No. 1 January 2009, pp. e159-e163 (doi:10.1542/peds.2008-2361)
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ARTICLE

B-Cell Depletion for Autoimmune Thrombocytopenia and Autoimmune Hemolytic Anemia in Pediatric Systemic Lupus Erythematosus

Sathish Kumar, MDa, Susanne M. Benseler, MDa,b, Melanie Kirby-Allen, MD, FRCPCc and Earl D. Silverman, MD, FRCPCa,d

Divisions of a Rheumatology
c Hematology, Department of Pediatrics, Hospital for Sick Children, Toronto, Ontario, Canada
b Department of Health Policy Management and Evaluation and Public Health Sciences
d Department of Immunology, University of Toronto, Toronto, Ontario, Canada

OBJECTIVES. Our goal was to determine long-term efficacy and safety of B-cell–depletion therapy for children with autoimmune thrombocytopenia and autoimmune hemolytic anemia in pediatric systemic lupus erythematosus.

PATIENTS AND METHODS. A retrospective, single-center cohort study was conducted including all patients with pediatric systemic lupus erythematosus who were diagnosed with autoimmune thrombocytopenia and/or autoimmune hemolytic anemia and treated with rituximab. Treatment efficacy and safety parameters were monitored and recorded.

RESULTS. Nine patients with pediatric systemic lupus erythematosus were included in the study: 5 had autoimmune thrombocytopenia, 3 had autoimmune hemolytic anemia, and 1 had both. There were 5 female and 4 male patients; median age at diagnosis of pediatric systemic lupus erythematosus was 14 years (range: 8–16 years); and median pediatric systemic lupus erythematosus disease duration to time of rituximab treatment was 6 months (range: 2–30 months). Complete response was achieved in all 6 children with autoimmune thrombocytopenia (median time to complete response: 2 weeks [range: 1–12 weeks]). Two patients' conditions flared at 48 and 68 weeks, respectively, and were re-treated. The remaining 4 patients continued to be in remission at 24, 32, 36, and 88 weeks, respectively. All 4 children with autoimmune hemolytic anemia achieved complete response at a median time of 4 weeks (range: 4–32 weeks). All patients remained in complete response at 24, 44, 84, and 100 weeks of follow-up. Complete B-cell depletion was seen in all children after rituximab treatment. No serious infections occurred, but 1 patient had an infusion reaction.

CONCLUSIONS. Preliminary evidence suggests that B-cell–depletion therapy with rituximab is an efficacious and safe treatment for autoimmune thrombocytopenia and autoimmune hemolytic anemia in pediatric systemic lupus erythematosus. Despite the prolonged effect on B-cell numbers and function, no serious infections were observed.

Key Words: B-cell depletion • rituximab • systemic lupus erythematosus • children

Abbreviations: SLE—systemic lupus erythematosus • pSLE—pediatric systemic lupus erythematosus • AITP—autoimmune thrombocytopenia • AIHA—autoimmune hemolytic anemia • Ig—immunoglobulin • IVIg—intravenous immunoglobulin • CR—complete response • PR—partial response

Accepted Oct 8, 2008.


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eLetters:

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Serious Infections and "Iatrogenic Immunodeficiency in a Patient treated with Rituximab
Susan J. Schuval, et al.
Pediatrics Online, 8 May 2009 [Full text]