Abstract
Pediatric opsoclonus–myoclonus syndrome (OMS) is a neuroblastoma-associated neuroinflammatory disorder. The purpose of this study was to evaluate the capacity of immunotherapies used in treating OMS to normalize several putative biomarkers of disease activity. Soluble BAFF and APRIL and a variety of chemokine ligands for CXCR3, CXCR5, CCR4, CCR7 were measured in cerebrospinal fluid (CSF) and serum by ELISA in 433 children (296 OMS, 109 controls, 28 other inflammatory neurological disorders/OIND). A blinded scorer rated motor severity on the validated OMS evaluation scale. In untreated OMS, significant elevations of CSF BAFF (+57%), CXCL10 (+2.7-fold), CXCL13 (+16.5-fold) and serum CCL22 (+55%) and CCL17 (+121%) concentrations were found ( P P = 0.0003). In conclusion, striking distinctions in immunotherapy effects on BAFF/APRIL, CXCL13/CXCR5, CCL22/CCR4, and CCL21/CCR7 signaling were found. The contrasting pharmacodynamic patterns may offer utility as treatment biomarkers.
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