Mitoguardin Regulates Mitochondrial Fusion through MitoPLD and Is Required for Neuronal Homeostasis

Mol Cell. 2016 Jan 7;61(1):111-24. doi: 10.1016/j.molcel.2015.11.017. Epub 2015 Dec 17.

Abstract

Mitochondria undergo frequent morphological changes through fission and fusion. Mutations in core members of the mitochondrial fission/fusion machinery are responsible for severe neurodegenerative diseases. However, the mitochondrial fission/fusion mechanisms are poorly understood. We found that the loss of a mitochondrial protein encoding gene, mitoguardin (miga), leads to mitochondrial defects and neurodegeneration in fly eyes. Mammals express two orthologs of miga: Miga1 and Miga2. Both MIGA1 and MIGA2 form homotypic and heterotypic complexes on the outer membrane of the mitochondria. Loss of MIGA results in fragmented mitochondria, whereas overexpression of MIGA leads to clustering and fusion of mitochondria in both fly and mammalian cells. MIGA proteins function downstream of mitofusin and interact with MitoPLD to stabilize MitoPLD and facilitate MitoPLD dimer formation. Therefore, we propose that MIGA proteins promote mitochondrial fusion by regulating mitochondrial phospholipid metabolism via MitoPLD.

Keywords: Drosophila; MitoPLD; mitochondrial fusion; neurodegeneration.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Drosophila Proteins / genetics
  • Drosophila Proteins / metabolism*
  • Drosophila melanogaster / genetics
  • Drosophila melanogaster / metabolism*
  • Endoribonucleases
  • Female
  • Genotype
  • HEK293 Cells
  • HeLa Cells
  • Homeostasis
  • Humans
  • Male
  • Membrane Proteins / genetics
  • Membrane Proteins / metabolism*
  • Mice
  • Mice, Knockout
  • Mitochondria / enzymology*
  • Mitochondria / pathology
  • Mitochondrial Dynamics*
  • Mitochondrial Membranes / enzymology
  • Mitochondrial Proteins / genetics
  • Mitochondrial Proteins / metabolism*
  • Mutation
  • NIH 3T3 Cells
  • Neurons / enzymology*
  • Neurons / pathology
  • Phenotype
  • Phospholipase D / genetics
  • Phospholipase D / metabolism*
  • Photoreceptor Cells, Invertebrate / enzymology
  • Protein Multimerization
  • RNA Interference
  • Transfection

Substances

  • Drosophila Proteins
  • Membrane Proteins
  • Mitochondrial Proteins
  • mitoguardin-1 protein, mouse
  • Endoribonucleases
  • Zuc protein, Drosophila
  • Phospholipase D
  • mitoPLD protein, human
  • mitoPLD protein, mouse