Mutation in glycerol-3-phosphate dehydrogenase 1 like gene (GPD1-L) decreases cardiac Na+ current and causes inherited arrhythmias

Circulation. 2007 Nov 13;116(20):2260-8. doi: 10.1161/CIRCULATIONAHA.107.703330. Epub 2007 Oct 29.

Abstract

Background: Brugada syndrome is a rare, autosomal-dominant, male-predominant form of idiopathic ventricular fibrillation characterized by a right bundle-branch block and ST elevation in the right precordial leads of the surface ECG. Mutations in the cardiac Na+ channel SCN5A on chromosome 3p21 cause approximately 20% of the cases of Brugada syndrome; most mutations decrease inward Na+ current, some by preventing trafficking of the channels to the surface membrane. We previously used positional cloning to identify a new locus on chromosome 3p24 in a large family with Brugada syndrome and excluded SCN5A as a candidate gene.

Methods and results: We used direct sequencing to identify a mutation (A280V) in a conserved amino acid of the glycerol-3-phosphate dehydrogenase 1-like (GPD1-L) gene. The mutation was present in all affected individuals and absent in >500 control subjects. GPD1-L RNA and protein are abundant in the heart. Compared with wild-type GPD1-L, coexpression of A280V GPD1-L with SCN5A in HEK cells reduced inward Na+ currents by approximately 50% (P<0.005). Wild-type GPD1-L localized near the cell surface to a greater extent than A280V GPD1-L. Coexpression of A280V GPD1-L with SCN5A reduced SCN5A cell surface expression by 31+/-5% (P=0.01).

Conclusions: GPD1-L is a novel gene that may affect trafficking of the cardiac Na+ channel to the cell surface. A GPD1-L mutation decreases SCN5A surface membrane expression, reduces inward Na+ current, and causes Brugada syndrome.

Publication types

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

MeSH terms

  • Animals
  • Brugada Syndrome / genetics*
  • Brugada Syndrome / physiopathology*
  • COS Cells
  • Chlorocebus aethiops
  • Chromosomes, Human, Pair 3
  • Family Health
  • Female
  • Glycerolphosphate Dehydrogenase / genetics*
  • Glycerolphosphate Dehydrogenase / metabolism
  • Heart / physiology
  • Humans
  • Italy
  • Kidney / cytology
  • Male
  • Muscle Proteins / genetics*
  • Muscle Proteins / metabolism
  • NAV1.5 Voltage-Gated Sodium Channel
  • Pedigree
  • Point Mutation
  • Sodium / metabolism
  • Sodium Channels / genetics*
  • Sodium Channels / metabolism
  • Sugar Alcohol Dehydrogenases / genetics*
  • Sugar Alcohol Dehydrogenases / metabolism
  • Ventricular Fibrillation / genetics
  • Ventricular Fibrillation / physiopathology

Substances

  • Muscle Proteins
  • NAV1.5 Voltage-Gated Sodium Channel
  • SCN5A protein, human
  • Sodium Channels
  • Sodium
  • GPD1L protein, human
  • Glycerolphosphate Dehydrogenase
  • Sugar Alcohol Dehydrogenases