SDHAF4 promotes mitochondrial succinate dehydrogenase activity and prevents neurodegeneration

Cell Metab. 2014 Aug 5;20(2):241-52. doi: 10.1016/j.cmet.2014.05.012. Epub 2014 Jun 19.

Abstract

Succinate dehydrogenase (SDH) occupies a central place in cellular energy production, linking the tricarboxylic cycle with the electron transport chain. As a result, a subset of cancers and neuromuscular disorders result from mutations affecting any of the four SDH structural subunits or either of two known SDH assembly factors. Herein we characterize an evolutionarily conserved SDH assembly factor designated Sdh8/SDHAF4, using yeast, Drosophila, and mammalian cells. Sdh8 interacts specifically with the catalytic Sdh1 subunit in the mitochondrial matrix, facilitating its association with Sdh2 and the subsequent assembly of the SDH holocomplex. These roles for Sdh8 are critical for preventing motility defects and neurodegeneration in Drosophila as well as the excess ROS generated by free Sdh1. These studies provide insights into the mechanisms by which SDH is assembled and raise the possibility that some forms of neuromuscular disease may be associated with mutations that affect this SDH assembly factor.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Animals
  • Drosophila / metabolism
  • Drosophila Proteins / antagonists & inhibitors
  • Drosophila Proteins / genetics
  • Drosophila Proteins / metabolism
  • Electron Transport Complex II / metabolism
  • Male
  • Mice
  • Mitochondria / enzymology*
  • Mitochondrial Proteins / antagonists & inhibitors
  • Mitochondrial Proteins / genetics
  • Mitochondrial Proteins / metabolism*
  • Neurodegenerative Diseases / metabolism
  • Neurodegenerative Diseases / pathology
  • Oxidative Stress
  • RNA Interference
  • RNA, Small Interfering / metabolism
  • Saccharomyces cerevisiae / metabolism
  • Saccharomyces cerevisiae Proteins / antagonists & inhibitors
  • Saccharomyces cerevisiae Proteins / genetics
  • Saccharomyces cerevisiae Proteins / metabolism
  • Succinate Dehydrogenase / metabolism*

Substances

  • Drosophila Proteins
  • Mitochondrial Proteins
  • RNA, Small Interfering
  • SDH1 protein, S cerevisiae
  • Saccharomyces cerevisiae Proteins
  • Electron Transport Complex II
  • Succinate Dehydrogenase