Published online December 15, 2004
PEDIATRICS Vol. 115 No. 1 January 2005, pp. e115-e119 (doi:10.1542/10.1542/peds.2004-0845)
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ELECTRONIC ARTICLE

Immunologic and Clinical Responses to Rituximab in a Child With Opsoclonus-Myoclonus Syndrome

Michael R. Pranzatelli, MD*,{ddagger}, Elizabeth D. Tate, FNP-C, MN*, Anna L. Travelstead, BS, MT(ASCP)§ and Darryl Longee, MD||

* Departments of Neurology
{ddagger} Pediatrics
§ Medical Microbiology and Immunology, Southern Illinois University School of Medicine, Springfield, Illinois
|| Department of Pediatric Hematology/Oncology, Carilion Roanoke Community Hospital, Roanoke, Virginia

Opsoclonus-myoclonus syndrome (OMS) is an autoimmune disorder with serious neurodevelopmental morbidity and limited treatment options. We treated a toddler with moderately severe OMS with rituximab, a monoclonal anti-B cell antibody. The patient's clinical response was documented on videotape and scored with the OMS Evaluation Scale. Cerebrospinal fluid lymphocyte subsets were evaluated by flow-cytometric immunophenotyping, with a comprehensive panel of monoclonal antibodies. Eradication of cerebrospinal fluid B cells, which previously were expanded, was associated with dramatic clinical improvement. There also were secondary changes in other lymphocyte subsets that might be relevant to the clinical response and lack of serious infections. In addition to clarifying the immune response to B-cell depletion, these data reveal a promising new therapy for OMS that warrants a phase I clinical trial.

Key Words: neuroblastoma • Kinsbourne syndrome • dancing eyes syndrome • paraneoplastic disorder • cerebrospinal fluid lymphocyte immunophenotyping

Abbreviations: OMS, opsoclonus-myoclonus syndrome • CSF, cerebrospinal fluid • NK, natural killer • IVIg, intravenously administered immunoglobulin • Ig, immunoglobulin

Accepted Aug 16, 2004.


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 J Child NeurolHome page
M. R. Pranzatelli, E. D. Tate, A. L. Travelstead, C. A. Baumgardner, N. V. Gowda, S. N. Halthore, P. Kerstan, B. D. Kossak, W. G. Mitchell, and J. W. Taub
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[Abstract] [PDF]