Increased copper toxicity in Saccharomyces cerevisiae lacking VPS35, a component of the retromer and monogenic Parkinson disease gene in humans

Biochem Biophys Res Commun. 2016 Aug 5;476(4):528-533. doi: 10.1016/j.bbrc.2016.05.157. Epub 2016 Jun 1.

Abstract

The Saccharomyces cerevisiae gene VPS35 encodes a component of the retromer complex which is involved in vesicle transport from endosomes to the trans-Golgi network. Yeast and human VPS35 orthologs are highly conserved and mutations in human VPS35 cause an autosomal dominant form of late-onset Parkinson disease (PD). We now show that deletion of VPS35 in yeast (vps35Δ) leads to a dose-dependent growth defect towards copper. This increased sensitivity could be rescued by transformation with yeast wild-type VPS35 but not by the expression of a construct harboring the yeast equivalent (i.e. D686N) of the most commonly identified VPS35-associated PD mutation, p.D620N. In addition, we show that expression of one copy of α-synuclein, which is known to directly interact with copper, leads to a pronounced aggravation of copper toxicity in vps35Δ cells, thereby linking the regulation of copper homeostasis by Vps35p in yeast to one of the key molecules in PD pathophysiology.

Keywords: Copper; Heavy metal homeostasis; Parkinson disease; Retromer; VPS35; α-synuclein.

MeSH terms

  • Copper / toxicity*
  • Gene Knockout Techniques
  • Genes, Fungal
  • Genetic Complementation Test
  • Humans
  • Mutation
  • Parkinson Disease / genetics*
  • Parkinson Disease Associated Proteins / genetics
  • Recombinant Proteins / genetics
  • Recombinant Proteins / metabolism
  • Saccharomyces cerevisiae / drug effects*
  • Saccharomyces cerevisiae / genetics
  • Saccharomyces cerevisiae / metabolism*
  • Saccharomyces cerevisiae Proteins / genetics
  • Saccharomyces cerevisiae Proteins / metabolism*
  • Vesicular Transport Proteins / deficiency
  • Vesicular Transport Proteins / genetics*
  • Vesicular Transport Proteins / metabolism*
  • alpha-Synuclein / genetics
  • alpha-Synuclein / metabolism

Substances

  • Parkinson Disease Associated Proteins
  • Recombinant Proteins
  • SNCA protein, human
  • Saccharomyces cerevisiae Proteins
  • VPS35 protein, S cerevisiae
  • VPS35 protein, human
  • Vesicular Transport Proteins
  • alpha-Synuclein
  • Copper

Supplementary concepts

  • Parkinson Disease, Familial, Type 1