Knockdown of the mitochondria-localized protein p13 protects against experimental parkinsonism

EMBO Rep. 2018 Mar;19(3):e44860. doi: 10.15252/embr.201744860. Epub 2018 Jan 25.

Abstract

Mitochondrial dysfunction in the nigrostriatal dopaminergic system is a critical hallmark of Parkinson's disease (PD). Mitochondrial toxins produce cellular and behavioural dysfunctions resembling those in patients with PD Causative gene products for familial PD play important roles in mitochondrial function. Therefore, targeting proteins that regulate mitochondrial integrity could provide convincing strategies for PD therapeutics. We have recently identified a novel 13-kDa protein (p13) that may be involved in mitochondrial oxidative phosphorylation. In the current study, we examine the mitochondrial function of p13 and its involvement in PD pathogenesis using mitochondrial toxin-induced PD models. We show that p13 overexpression induces mitochondrial dysfunction and apoptosis. p13 knockdown attenuates toxin-induced mitochondrial dysfunction and apoptosis in dopaminergic SH-SY5Y cells via the regulation of complex I. Importantly, we generate p13-deficient mice using the CRISPR/Cas9 system and observe that heterozygous p13 knockout prevents toxin-induced motor deficits and the loss of dopaminergic neurons in the substantia nigra. Taken together, our results suggest that manipulating p13 expression may be a promising avenue for therapeutic intervention in PD.

Keywords: Parkinson's disease; cell death; complex I; mitochondria; p13.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Apoptosis / genetics
  • CRISPR-Cas Systems
  • Cell Line
  • Dopaminergic Neurons / metabolism
  • Dopaminergic Neurons / pathology
  • Flow Cytometry
  • Gene Expression Regulation, Developmental / genetics
  • Humans
  • Mice
  • Mice, Knockout
  • Mitochondria / genetics*
  • Mitochondria / pathology
  • Mitochondrial Diseases / genetics*
  • Mitochondrial Diseases / metabolism
  • Mitochondrial Diseases / pathology
  • Mitochondrial Proteins / genetics*
  • Oxidative Phosphorylation
  • Oxidative Stress / genetics
  • Parkinson Disease / genetics*
  • Parkinson Disease / metabolism
  • Parkinson Disease / pathology
  • Parkinsonian Disorders / genetics*
  • Parkinsonian Disorders / metabolism
  • Parkinsonian Disorders / pathology

Substances

  • Mitochondrial Proteins

Supplementary concepts

  • Parkinson Disease, Mitochondrial