Genetic and clinical heterogeneity in eIF2B-related disorder

J Child Neurol. 2008 Feb;23(2):205-15. doi: 10.1177/0883073807308705.

Abstract

Eukaryotic initiation factor 2B (eIF2B)-related disorders are heritable white matter disorders with a variable clinical phenotype (including vanishing white matter disease and ovarioleukodystrophy) and an equally heterogeneous genotype. We report 9 novel mutations in the EIF2B genes in our subject population, increasing the number of known mutations to more than 120. Using homology modeling, we have analyzed the impact of novel mutations on the 5 subunits of the eIF2B protein. Although recurrent mutations have been found at CpG dinucleotides in the EIF2B genes, the high incidence of private or low frequency mutations increases the challenge of providing rapid genetic confirmation of this disorder, and limits the application of EIF2B screening in cases of undiagnosed leukodystrophy.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural

MeSH terms

  • Adolescent
  • Adult
  • Brain Diseases / genetics*
  • Child
  • Child, Preschool
  • Eukaryotic Initiation Factor-2B / genetics*
  • Female
  • Genetic Heterogeneity*
  • Genetic Predisposition to Disease
  • Genetic Testing
  • Hereditary Central Nervous System Demyelinating Diseases / genetics*
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Models, Genetic
  • Mutation / genetics
  • Protein Subunits / genetics
  • Structural Homology, Protein

Substances

  • Eukaryotic Initiation Factor-2B
  • Protein Subunits