Confirmation of GRHL2 as the gene for the DFNA28 locus

Am J Med Genet A. 2013 Aug;161A(8):2060-5. doi: 10.1002/ajmg.a.36017. Epub 2013 Jun 27.

Abstract

More than 10 years ago, a c.1609_1610insC mutation in the grainyhead-like 2 (GRHL2) gene was identified in a large family with nonsyndromic sensorineural hearing loss, so far presenting the only evidence for GRHL2 being an autosomal-dominant deafness gene (DFNA28). Here, we report on a second large family, in which post-lingual hearing loss with a highly variable age of onset and progression segregated with a heterozygous non-classical splice site mutation in GRHL2. The c.1258-1G>A mutation disrupts the acceptor recognition sequence of intron 9, creating a new AG splice site, which is shifted by only one nucleotide in the 3' direction. cDNA analysis confirmed a p.Gly420Glufs*111 frameshift mutation in exon 10.

Keywords: DFNA28; GRHL2; autosomal dominant hearing impairment; haploinsufficiency; postlingual hearing impairment; progressive hearing loss.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Amino Acid Sequence
  • Base Sequence
  • DNA / analysis
  • DNA / genetics
  • DNA-Binding Proteins / genetics*
  • Female
  • Frameshift Mutation / genetics*
  • Genes, Dominant*
  • Hearing Loss, Sensorineural / diagnosis
  • Hearing Loss, Sensorineural / genetics*
  • Humans
  • Male
  • Middle Aged
  • Molecular Sequence Data
  • Pedigree
  • Polymerase Chain Reaction
  • Transcription Factors / genetics*

Substances

  • DNA-Binding Proteins
  • GRHL2 protein, human
  • Transcription Factors
  • DNA

Supplementary concepts

  • Deafness, Autosomal Dominant 28