Mitochondrial disorders of the nuclear genome

Acta Myol. 2009 Jul;28(1):16-23.

Abstract

Background: Mitochondrial myopathies are regulated by two genomes: the nuclear DNA, and the mitochondrial DNA. While, so far, most studies have dealt with mitochondrial myopathies due to deletions or point mutations in the mitochondrial DNA, a new field of investigation is that of syndromes due to mutations in the nuclear DNA. These latter disorders have mendelian inheritance.

Results: Three representative cases have been selected: one with COX deficiency and a Leigh syndrome due to a SURF1 gene mutation, one due to a defect of Coenzyme Q synthesis and one with dominant optic atrophy due to a mutation in the OPA1 gene.

Conclusions: Future developments will show that many neurodegenerative disorders are due to mutations of nuclear genes controlling mitochondrial function, fusion and fission.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cell Nucleus*
  • Child
  • DNA / genetics*
  • Female
  • GTP Phosphohydrolases / genetics
  • Genome / genetics*
  • Humans
  • Infant
  • Leigh Disease / diagnosis
  • Leigh Disease / genetics
  • Male
  • Membrane Proteins / genetics
  • Middle Aged
  • Mitochondrial Diseases / diagnosis
  • Mitochondrial Diseases / genetics*
  • Mitochondrial Proteins / genetics
  • Mutation / genetics
  • Optic Atrophy / diagnosis
  • Optic Atrophy / genetics
  • Prostaglandin-Endoperoxide Synthases / deficiency
  • Prostaglandin-Endoperoxide Synthases / genetics
  • Ubiquinone / genetics

Substances

  • Membrane Proteins
  • Mitochondrial Proteins
  • Surf-1 protein
  • Ubiquinone
  • DNA
  • Prostaglandin-Endoperoxide Synthases
  • GTP Phosphohydrolases
  • OPA1 protein, human