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Pranzatelli, Michael R.; Tate, Elizabeth D.; Swan, Jennifer A.; Travelstead, Anna L.; Colliver, Jerry A.; Verhulst, Steven J.; Crosley, Carl J.; Graf, William D.; Joseph, Suja A.; Kelfer, Howard M.; Raju, G. Praveen
Movement disorders, 30 January 2010, Letnik: 25, Številka: 2Journal Article
Twelve immunotherapy‐naïve children with opsoclonus‐myoclonus syndrome and CSF B cell expansion received rituximab, adrenocorticotropic hormone (ACTH), and IVIg. Motor severity lessened 73% by 6 mo and 81% at 1 yr (P < 0.0001). Opsoclonus and action myoclonus disappeared rapidly, whereas gait ataxia and some other motor components improved more slowly. ACTH dose was tapered by 87%. Reduction in total CSF B cells was profound at 6 mo (‐93%). By study end, peripheral B cells returned to 53% of baseline and serum IgM levels to 63%. Overall clinical response trailed peripheral B cell and IgM depletion, but improvement continued after their levels recovered. All but one non‐ambulatory subject became ambulatory without additional chemotherapy; two relapsed and remitted; four had rituximab‐related or possibly related adverse events; and two had low‐titer human anti‐chimeric antibody. Combination of rituximab with conventional agents as initial therapy was effective and safe. A controlled trial with long‐term safety monitoring is indicated. © 2009 Movement Disorder Society
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JCR | SNIP | JCR | SNIP | JCR | SNIP | JCR | SNIP |
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in: SICRIS
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