Hsp110 mitigates α-synuclein pathology in vivo

Proc Natl Acad Sci U S A. 2019 Nov 26;116(48):24310-24316. doi: 10.1073/pnas.1903268116. Epub 2019 Nov 4.

Abstract

Parkinson's disease is characterized by the aggregation of the presynaptic protein α-synuclein and its deposition into pathologic Lewy bodies. While extensive research has been carried out on mediators of α-synuclein aggregation, molecular facilitators of α-synuclein disaggregation are still generally unknown. We investigated the role of molecular chaperones in both preventing and disaggregating α-synuclein oligomers and fibrils, with a focus on the mammalian disaggregase complex. Here, we show that overexpression of the chaperone Hsp110 is sufficient to reduce α-synuclein aggregation in a mammalian cell culture model. Additionally, we demonstrate that Hsp110 effectively mitigates α-synuclein pathology in vivo through the characterization of transgenic Hsp110 and double-transgenic α-synuclein/Hsp110 mouse models. Unbiased analysis of the synaptic proteome of these mice revealed that overexpression of Hsp110 can override the protein changes driven by the α-synuclein transgene. Furthermore, overexpression of Hsp110 is sufficient to prevent endogenous α-synuclein templating and spread following injection of aggregated α-synuclein seeds into brain, supporting a role for Hsp110 in the prevention and/or disaggregation of α-synuclein pathology.

Keywords: Lewy body; chaperone; disaggregase; proteomics; synapse.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, Non-P.H.S.

MeSH terms

  • Animals
  • Brain / metabolism
  • Brain / pathology*
  • Disease Models, Animal
  • Green Fluorescent Proteins / genetics
  • Green Fluorescent Proteins / metabolism
  • HEK293 Cells
  • HSP110 Heat-Shock Proteins / genetics
  • HSP110 Heat-Shock Proteins / metabolism*
  • Humans
  • Mice, Transgenic
  • Parkinson Disease / etiology*
  • Parkinson Disease / pathology
  • Spinal Cord / metabolism
  • Spinal Cord / pathology
  • Synucleinopathies / genetics
  • Synucleinopathies / mortality
  • Synucleinopathies / pathology
  • alpha-Synuclein / genetics
  • alpha-Synuclein / metabolism*

Substances

  • HSP110 Heat-Shock Proteins
  • HSPH1 protein, human
  • SNCA protein, human
  • alpha-Synuclein
  • Green Fluorescent Proteins