The D355V mutation decreases EGR2 binding to an element within the Cx32 promoter

Neurobiol Dis. 2001 Aug;8(4):700-6. doi: 10.1006/nbdi.2001.0397.

Abstract

Mutations in the early growth response 2 (EGR2) gene are associated with some forms of Charcot--Marie--Tooth disease (CMT) and other demyelinating neuropathies. These mutations modify the EGR2 binding to specific DNA sequences suggesting a role in the transcriptional control of myelination-specific genes. Here we show that the D355V mutation, associated with a CMT case combining axonal and demyelinating abnormalities, reduces three times the affinity of EGR2 to its consensus sequence and ten times its affinity to a sequence in the human Cx32 promoter. These findings could indicate that this EGR2 mutation leads to the development of CMT1 through the transcriptional deregulation of Cx32 gene.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Binding, Competitive / physiology
  • Charcot-Marie-Tooth Disease / genetics*
  • Charcot-Marie-Tooth Disease / metabolism
  • Child
  • Connexins / genetics*
  • DNA-Binding Proteins / metabolism*
  • Early Growth Response Protein 2
  • Female
  • Gap Junction beta-1 Protein
  • Gene Expression / physiology
  • Humans
  • Myelin Sheath / physiology
  • Point Mutation*
  • Promoter Regions, Genetic / physiology*
  • Transcription Factors / metabolism*
  • Transcription, Genetic / physiology

Substances

  • Connexins
  • DNA-Binding Proteins
  • EGR2 protein, human
  • Early Growth Response Protein 2
  • Transcription Factors

Grants and funding