scyllo-Inositol promotes robust mutant Huntingtin protein degradation

J Biol Chem. 2014 Feb 7;289(6):3666-76. doi: 10.1074/jbc.M113.501635. Epub 2013 Dec 18.

Abstract

Huntington disease is characterized by neuronal aggregates and inclusions containing polyglutamine-expanded huntingtin protein and peptide fragments (polyQ-Htt). We have used an established cell-based assay employing a PC12 cell line overexpressing truncated exon 1 of Htt with a 103-residue polyQ expansion that yields polyQ-Htt aggregates to investigate the fate of polyQ-Htt-drug complexes. scyllo-Inositol is an endogenous inositol stereoisomer known to inhibit accumulation and toxicity of the amyloid-β peptide and α-synuclein. In light of these properties, we investigated the effect of scyllo-inositol on polyQ-Htt accumulation. We show that scyllo-inositol lowered the number of visible polyQ-Htt aggregates and robustly decreased polyQ-Htt protein abundance without concomitant cellular toxicity. We found that scyllo-inositol-induced polyQ-Htt reduction was by rescue of degradation pathways mediated by the lysosome and by the proteasome but not autophagosomes. The rescue of degradation pathways was not a direct result of scyllo-inositol on the lysosome or proteasome but due to scyllo-inositol-induced reduction in mutant polyQ-Htt protein levels.

Keywords: Autophagy; Huntington Disease; Lysosomes; Neurodegenerative Diseases; Proteasome; huntingtin; scyllo-Inositol.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Amyloid beta-Peptides / genetics
  • Amyloid beta-Peptides / metabolism
  • Animals
  • Huntingtin Protein
  • Inositol / pharmacology*
  • Lysosomes / genetics
  • Lysosomes / metabolism*
  • Mutation*
  • Nerve Tissue Proteins / genetics
  • Nerve Tissue Proteins / metabolism*
  • Nuclear Proteins / genetics
  • Nuclear Proteins / metabolism*
  • PC12 Cells
  • Peptides / genetics
  • Peptides / metabolism
  • Proteasome Endopeptidase Complex / genetics
  • Proteasome Endopeptidase Complex / metabolism*
  • Proteolysis / drug effects*
  • Rats
  • alpha-Synuclein / genetics
  • alpha-Synuclein / metabolism

Substances

  • Amyloid beta-Peptides
  • Htt protein, rat
  • Huntingtin Protein
  • Nerve Tissue Proteins
  • Nuclear Proteins
  • Peptides
  • Snca protein, rat
  • alpha-Synuclein
  • scyllitol
  • polyglutamine
  • Inositol
  • Proteasome Endopeptidase Complex