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Clinical features and long-term outcome of a group of Japanese chil… – PubMed – NCBI

Clinical features and long-term outcome of a group of Japanese chil… – PubMed – NCBI.

Clinical features and long-term outcome of a group of Japanese children with inflammatory central nervous system disorders and seropositivity to myelin-oligodendrocyte glycoprotein antibodies.

Hino-Fukuyo N1, Haginoya K2, Nakashima I3, Sato DK3, Takahashi T3, Misu T3, Fujihara K3, Hirose M2, Kakisaka Y2, Uematsu M2, Kobayashi T2, Kure S2.

Author information

  • 1Department of Pediatrics, Tohoku University School of Medicine, Sendai, Japan. Electronic address: naomi-h@zc4.so-net.ne.jp.
  • 2Department of Pediatrics, Tohoku University School of Medicine, Sendai, Japan.
  • 3Department of Neurology, Tohoku University of School of Medicine, Sendai, Japan.

Abstract

BACKGROUND:

Myelin-oligodendrocyte glycoprotein and aquaporin-4 have been extensively analyzed as targets for humoral immune reactions in central nervous system (CNS) demyelinating diseases, and the results indicated a possible role of these antibodies in the pathogenesis of various demyelinating diseases.

OBJECTIVE:

To investigate the antibody titer levels against myelin-oligodendrocyte glycoprotein and aquaporin-4 in pediatric patients with inflammatory CNS disorders, and to evaluate clinical significance to study anti-myelin-oligodendrocyte glycoprotein antibodies.

METHODS:

Sera at onset from patients with acute disseminated encephalomyelitis (ADEM) in 7, optic neuritis (ON) in 5, pediatric MS in 4 and neuromyelitis optica in one were tested for myelin-oligodendrocyte glycoprotein and aquaporin-4 antibodies using cell-based assays with live transfected cells. The duration of the observation periods ranged from 1 to 21years (median, 10years). We also described clinical course of patients with positive anti-myelin-oligodendrocyte glycoprotein antibodies.

RESULTS:

Among 17 patients diagnosed with inflammatory CNS demyelinating diseases nine (52%) were positive to anti-myelin-oligodendrocyte glycoprotein antibodies. Of note, all cases with positive anti-myelin-oligodendrocyte glycoprotein antibodies showed seronegativity against anti-aquaporin-4 antibodies and had a favorable prognosis.

CONCLUSIONS:

This preliminary report showed that anti-myelin-oligodendrocyte glycoprotein antibodies testing at onset could be a useful tool predicting clinical outcome of children with ADEM, ON, and MS.

Copyright © 2015 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

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