Clinical features in very early-onset demyelinating disease with anti-MOG antibody

Masahiro Nishiyama, Hiroaki Nagase, Masaaki Matsumoto, Kazumi Tomioka, Hiroyuki Awano, Tsukasa Tanaka, Daisaku Toyoshima, Kyoko Fujita, Azusa Maruyama, Yoshinobu Oyazato, Keisuke Saeki, Kazuhiro Shiraishi, Satoshi Takada, Kimihiko Kaneko, Toshiyuki Takahashi, Ichiro Nakashima, Kazumoto Iijima

Research output: Contribution to journalArticlepeer-review

3 Citations (Scopus)

Abstract

Background The clinical features of patients with very early-onset acquired demyelinating syndrome (ADS) with the anti-myelin oligodendrocyte glycoprotein (MOG) antibody are unknown. We investigated the clinical characteristics and described detailed treatment of weekly intramuscular interferon β-1a (IFNβ-1a) in children aged <4 years with ADS and the anti-MOG antibody. Methods We conducted a retrospective chart review of patients with anti-MOG positivity who were diagnosed as having multiple sclerosis (MS) at <4 years of age. Results Subjects comprised 2 boys and 2 girls. Initial symptoms included ataxia, facial paresis, status epilepticus, and encephalopathy. Abnormal lesions on magnetic resonance imaging scans were often detected in the brainstem and cerebellum as well as the cerebrum. All patients started receiving IFNβ-1a at age 3.1–3.5 years. The initial doses ranged from 3 to 6 μg, which were 1/10–1/5 doses, respectively, for adults. During 0.6–4.3 years of IFNβ-1a administration, all patients had flu-like symptoms, and 1 patient had an increased liver enzyme level. Although 1 patient discontinued IFNβ-1a therapy because of frequent relapses, no patient discontinued therapy due to severe adverse events. Conclusions This case series adds novel information regarding the clinical features of children <4 years old with ADS and the anti-MOG antibody.

Original languageEnglish
Pages (from-to)756-762
Number of pages7
JournalBrain and Development
Volume39
Issue number9
DOIs
Publication statusPublished - 2017 Oct

Keywords

  • Anti-MOG antibody
  • Beta-interferon
  • Children
  • Disease modifying therapies
  • Early childhood
  • Multiple sclerosis
  • Treatment

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Developmental Neuroscience
  • Clinical Neurology

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