Diagnostic algorithm for relapsing acquired demyelinating syndromes in children

Neurology. 2017 Jul 18;89(3):269-278. doi: 10.1212/WNL.0000000000004117. Epub 2017 Jun 14.

Abstract

Objective: To establish whether children with relapsing acquired demyelinating syndromes (RDS) and myelin oligodendrocyte glycoprotein antibodies (MOG-Ab) show distinctive clinical and radiologic features and to generate a diagnostic algorithm for the main RDS for clinical use.

Methods: A panel reviewed the clinical characteristics, MOG-Ab and aquaporin-4 (AQP4) Ab, intrathecal oligoclonal bands, and Epstein-Barr virus serology results of 110 children with RDS. A neuroradiologist blinded to the diagnosis scored the MRI scans. Clinical, radiologic, and serologic tests results were compared.

Results: The findings showed that 56.4% of children were diagnosed with multiple sclerosis (MS), 25.4% with neuromyelitis optica spectrum disorder (NMOSD), 12.7% with multiphasic disseminated encephalomyelitis (MDEM), and 5.5% with relapsing optic neuritis (RON). Blinded analysis defined baseline MRI as typical of MS in 93.5% of children with MS. Acute disseminated encephalomyelitis presentation was seen only in the non-MS group. Of NMOSD cases, 30.7% were AQP4-Ab positive. MOG-Ab were found in 83.3% of AQP4-Ab-negative NMOSD, 100% of MDEM, and 33.3% of RON. Children with MOG-Ab were younger, were less likely to present with area postrema syndrome, and had lower disability, longer time to relapse, and more cerebellar peduncle lesions than children with AQP4-Ab NMOSD. A diagnostic algorithm applicable to any episode of CNS demyelination leads to 4 main phenotypes: MS, AQP4-Ab NMOSD, MOG-Ab-associated disease, and antibody-negative RDS.

Conclusions: Children with MS and AQP4-Ab NMOSD showed features typical of adult cases. Because MOG-Ab-positive children showed notable and distinctive clinical and MRI features, they were grouped into a unified phenotype (MOG-Ab-associated disease), included in a new diagnostic algorithm.

MeSH terms

  • Adolescent
  • Algorithms*
  • Aquaporin 4 / immunology
  • Autoantibodies / blood
  • Brain / diagnostic imaging
  • Child
  • Child, Preschool
  • Clinical Decision-Making*
  • Encephalomyelitis / blood
  • Encephalomyelitis / diagnosis*
  • Encephalomyelitis / diagnostic imaging
  • Encephalomyelitis / immunology
  • Female
  • Follow-Up Studies
  • Herpesvirus 4, Human
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Multiple Sclerosis / blood
  • Multiple Sclerosis / diagnostic imaging*
  • Multiple Sclerosis / immunology
  • Myelin-Oligodendrocyte Glycoprotein / immunology
  • Neuromyelitis Optica / blood
  • Neuromyelitis Optica / diagnosis*
  • Neuromyelitis Optica / diagnostic imaging
  • Neuromyelitis Optica / immunology
  • Oligoclonal Bands
  • Optic Neuritis / blood
  • Optic Neuritis / diagnosis*
  • Optic Neuritis / diagnostic imaging
  • Optic Neuritis / immunology
  • Recurrence
  • Retrospective Studies
  • Viral Load

Substances

  • Aquaporin 4
  • Autoantibodies
  • MOG protein, human
  • Myelin-Oligodendrocyte Glycoprotein
  • Oligoclonal Bands