Genetic analysis of the G4.5 gene in families with suspected Barth syndrome

J Pediatr. 1999 Sep;135(3):311-5. doi: 10.1016/s0022-3476(99)70126-5.

Abstract

Mutations have recently been identified in the G4.5 gene (Xq28), encoding the tafazzin protein, in patients with Barth syndrome. We performed mutational analysis in 5 families with suspected Barth syndrome. In 4 families a male child had all the cardinal features of this syndrome, and mutations of G4.5 were found in each case. A mutation was also found in a fifth family with an extensive history of early infant death from heart disease. The recognition of 5 unrelated families in 1 hospital during a 7-year period suggests that this disease may be underdiagnosed.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cardiomyopathy, Dilated / diagnosis*
  • Cardiomyopathy, Dilated / genetics*
  • Cardiomyopathy, Dilated / metabolism
  • DNA Mutational Analysis*
  • Failure to Thrive / diagnosis*
  • Failure to Thrive / genetics*
  • Failure to Thrive / metabolism
  • Genetic Linkage / genetics*
  • Glutarates / urine*
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Mitochondrial Myopathies / diagnosis
  • Mitochondrial Myopathies / genetics
  • Mitochondrial Myopathies / metabolism
  • Mutagenesis, Insertional / genetics*
  • Mutation, Missense / genetics*
  • Neutropenia / diagnosis*
  • Neutropenia / genetics*
  • Neutropenia / metabolism
  • Pedigree
  • Syndrome
  • X Chromosome / genetics*

Substances

  • Glutarates
  • 3-methylglutaconic acid