'Leukodystrophy-like' phenotype in children with myelin oligodendrocyte glycoprotein antibody-associated disease

Dev Med Child Neurol. 2018 Apr;60(4):417-423. doi: 10.1111/dmcn.13649. Epub 2017 Dec 30.

Abstract

Aim: To review the demographics and clinical and paraclinical parameters of children with myelin oligodendrocyte glycoprotein (MOG) antibody-associated relapsing disease.

Method: In this UK-based, multicentre study, 31 children with MOG antibody-associated relapsing disease were studied retrospectively.

Results: Of the 31 children studied, 14 presented with acute disseminated encephalomyelitis (ADEM); they were younger (mean 4.1y) than the remainder (mean 8.5y) who presented with optic neuritis and/or transverse myelitis (p<0.001). Similarly, children who had an abnormal brain magnetic resonance imaging (MRI) at onset (n=20) were younger than patients with normal MRI at onset (p=0.001) or at follow-up (p<0.001). 'Leukodystrophy-like' MRI patterns of confluent largely symmetrical lesions was seen during the course of the disease in 7 out of 14 children with a diagnosis of ADEM, and was only seen in children younger than 7 years of age. Their disability after a 3-year follow-up was mild to moderate, and most patients continued to relapse, despite disease-modifying treatments.

Interpretation: MOG antibody should be tested in children presenting with relapsing neurological disorders associated with confluent, bilateral white matter changes, and distinct enhancement pattern. Children with MOG antibody-associated disease present with age-related differences in phenotypes, with a severe leukoencephalopathy phenotype in the very young and normal intracranial MRI in the older children. This finding suggests a susceptibility of the very young and myelinating brain to MOG antibody-mediated mechanisms of damage.

What this paper adds: Myelin oligodendrocyte glycoprotein (MOG) antibody-associated demyelination manifest with an age-related phenotype. Children with MOG antibody and 'leukodystrophy-like' imaging patterns tend to have poor response to second-line immunotherapy.

Publication types

  • Multicenter Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age Factors
  • Autoantibodies / blood*
  • Brain / diagnostic imaging
  • Child
  • Disability Evaluation
  • Encephalomyelitis, Acute Disseminated / blood*
  • Encephalomyelitis, Acute Disseminated / immunology*
  • Encephalomyelitis, Acute Disseminated / physiopathology
  • Female
  • Humans
  • Ireland
  • Magnetic Resonance Imaging
  • Male
  • Myelin-Oligodendrocyte Glycoprotein / immunology*
  • Phenotype
  • Retrospective Studies
  • Spinal Cord / diagnostic imaging
  • United Kingdom

Substances

  • Autoantibodies
  • Myelin-Oligodendrocyte Glycoprotein