Steroid-responsive neurologic relapses in a child with a proteolipid protein-1 mutation

Neurology. 2007 Apr 17;68(16):1305-7. doi: 10.1212/01.wnl.0000259522.49388.53.

Abstract

A 10-year-old boy developed corticosteroid-responsive relapsing neurologic signs, including nystagmus and ataxia. MRI revealed multifocal T2 white matter hyperintensities; several were gadolinium-enhancing. CSF contained oligoclonal bands. Although the patient met criteria for multiple sclerosis (MS), the proteolipid protein-1 gene (PLP1) contained a mutation in exon 3B (c.409C>T), predicting a tryptophan-for-arginine substitution. This case raises questions about the role of inflammation in PLP1-related disorders and, conversely, PLP1 mutations in MS.

Publication types

  • Case Reports
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amino Acid Substitution / genetics
  • Central Nervous System / metabolism
  • Central Nervous System / pathology
  • Central Nervous System / physiopathology
  • Cerebellar Diseases / genetics
  • Cerebellar Diseases / immunology
  • Cerebellar Diseases / physiopathology
  • Child
  • DNA Mutational Analysis
  • Disease Progression
  • Exons / genetics
  • Genetic Predisposition to Disease / genetics*
  • Humans
  • Inflammation / genetics
  • Inflammation / immunology
  • Inflammation / physiopathology
  • Interferon beta-1a
  • Interferon-beta / therapeutic use
  • Magnetic Resonance Imaging
  • Male
  • Membrane Proteins / genetics*
  • Methylprednisolone / therapeutic use
  • Multiple Sclerosis, Relapsing-Remitting / drug therapy*
  • Multiple Sclerosis, Relapsing-Remitting / genetics*
  • Multiple Sclerosis, Relapsing-Remitting / immunology
  • Mutation / genetics*
  • Myelin Proteolipid Protein / genetics*
  • Neuroprotective Agents / therapeutic use
  • Oligoclonal Bands / cerebrospinal fluid
  • Remission Induction
  • Steroids / therapeutic use*
  • Treatment Outcome

Substances

  • Membrane Proteins
  • Myelin Proteolipid Protein
  • Neuroprotective Agents
  • Oligoclonal Bands
  • PLP1 protein, human
  • Steroids
  • Interferon-beta
  • Methylprednisolone
  • Interferon beta-1a