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Journal of Child Neurology
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Evidence of Cellular Immune Activation in Children With Opsoclonus-Myoclonus: Cerebrospinal Fluid Neopterin

Michael R. Pranzatelli, MD

Department of Neurology, Southern Illinois University School of Medicine, Springfield, IL, www.omsusa.org, Department of Pediatrics, Southern Illinois University School of Medicine, Springfield, IL

Keith Hyland, PhD

Institute of Metabolic Research, Baylor University Medical Center, Dallas, TX

Elizabeth D. Tate, CFNP, MN

Department of Neurology, Southern Illinois University School of Medicine, Springfield, IL

Lauren A. Arnold, MS

Institute of Metabolic Research, Baylor University Medical Center, Dallas, TX

Tyler J. Allison, BS

Department of Neurology, Southern Illinois University School of Medicine, Springfield, IL

Gamini S. Soori, MD

The Division of Hematology/Oncology, Department of Medicine, Creighton University, Omaha, NB

To evaluate cellular immune activation in opsoclonus-myoclonus syndrome, we measured the inflammatory marker neopterin in the cerebrospinal fluid of 16 children with opsoclonus-myoclonus and neuroblastoma, 24 children with opsoclonus-myoclonus but no tumor, and 19 age-matched controls. The mean concentration in opsoclonus-myoclonus was 2.3-fold higher than in controls (P = .008). Neopterin was greatly elevated in four of the most neurologically severe cases, up to 8.3-fold above the highest control level. Thirteen of the 40 children with opsoclonus-myoclonus but no controls had a neopterin concentration > 2 SD above the control mean (P = .005). In this high neopterin subgroup, neurologic severity was significantly greater and the duration of neurologic symptoms was less. In 16 children re-examined on immunotherapy, including adrenocorticotropic hormone (ACTH) combination therapy, treatment was associated with a significant reduction in both neopterin and neurologic severity. Neopterin did not differ significantly between the tumor and non-tumor opsoclonus-myoclonus etiologies. No abnormalities of tetrahydrobiopterin were found. Although cerebrospinal fluid neopterin lacked the sensitivity to be a biomarker of disease activity in opsoclonus-myoclonus, elevated concentrations do support a role for T-cell activation and cell-mediated immunity in its pathophysiology. (J Child Neurol 2004;19:919—924).

Journal of Child Neurology, Vol. 19, No. 12, 919-924 (2004)
DOI: 10.1177/08830738040190120201
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