Recessive Retinopathy Consequent on Mutant G-Protein β Subunit 3 (GNB3)

JAMA Ophthalmol. 2016 Aug 1;134(8):924-7. doi: 10.1001/jamaophthalmol.2016.1543.

Abstract

Importance: Mutations in phototransduction and retinal signaling genes are implicated in many retinopathies. To our knowledge, GNB3 encoding the G-protein β subunit 3 (Gβ3) has not previously been implicated in human disease.

Observations: In this brief report, whole-exome sequencing was conducted on a patient with distinct inherited retinal disease presenting in childhood, with a phenotype characterized by nystagmus, normal retinal examination, and mild disturbance of the central macula on detailed retinal imaging. This sequencing revealed a homozygous GNB3 nonsense mutation (c.124C>T; p.Arg42Ter). Whole-exome sequencing was conducted from April 2015 to July 2015.

Conclusions and relevance: Expressed in cone photoreceptors and ON-bipolar cells, Gβ3 is essential in phototransduction and ON-bipolar cell signaling. Knockout of Gnb3 in mice results in dysfunction of cone photoreceptors and ON-bipolar cells and a naturally occurring chicken mutation leads to retinal degeneration. Identification of further affected patients may allow description of the phenotypic and genotypic spectrum of disease associated with GNB3 retinopathy.

Publication types

  • Case Reports

MeSH terms

  • Child
  • Codon, Nonsense*
  • DNA / genetics*
  • DNA Mutational Analysis
  • Electroretinography
  • Genes, Recessive
  • Genotype
  • Heterotrimeric GTP-Binding Proteins / genetics*
  • Heterotrimeric GTP-Binding Proteins / metabolism
  • Humans
  • Male
  • Pedigree
  • Retina / diagnostic imaging*
  • Retinal Diseases / diagnosis
  • Retinal Diseases / genetics*
  • Retinal Diseases / metabolism
  • Tomography, Optical Coherence

Substances

  • Codon, Nonsense
  • GNB3 protein, human
  • DNA
  • Heterotrimeric GTP-Binding Proteins