The Parkinson's disease-related protein DJ-1 protects dopaminergic neurons in vivo and cultured cells from alpha-synuclein and 6-hydroxydopamine toxicity

Neurodegener Dis. 2015;15(1):13-23. doi: 10.1159/000367993. Epub 2014 Dec 11.

Abstract

Background: Dopaminergic degeneration is a major finding in brains of patients with Parkinson's disease (PD), together with Lewy bodies, intraneuronal inclusions mainly composed of the fibrillogenic protein α-synuclein (α-syn). The familial-PD-related protein DJ-1 was reported to reduce dopaminergic degeneration triggered by α-syn or by the dopaminergic-selective neurotoxin 6-hydroxydopamine (6-OHDA).

Objective: The aim was to further investigate the role of DJ-1 in dopaminergic degeneration and to see whether a cell-permeable recombinant form of DJ-1 (TAT-DJ-1) could restore dopamine depletion in vivo, thus representing an innovative therapeutic approach.

Methods: We developed in vitro (PC12/TetOn cells and mouse primary mesencephalic neurons) and in vivo models [including DJ-1 knockout (-/-) mice] to investigate DJ-1 in dopaminergic degeneration.

Results: We found that in PC12/TetOn cells overexpressing α-syn with the familial-PD linked mutation A30P, DJ-1 silencing increased α-syn (A30P) toxicity. Primary mesencephalic neurons from DJ-1 (-/-) mice were more vulnerable to a cell-permeable form of α-syn (TAT-α-syn) and to 6-OHDA. Intrastriatally administered TAT-DJ-1 reduced 6-OHDA toxicity in vivo in C57BL/6 mice. Finally, when we injected TAT-α-syn (A30P) in the striatum of DJ-1 (-/-) animals, dopamine was depleted more than in the control strain.

Conclusion: DJ-1 appears to have a protective role against dopaminergic degeneration triggered by α-syn or 6-OHDA, reinforcing the possible therapeutic importance of this protein in PD.

MeSH terms

  • Animals
  • Cells, Cultured
  • Disease Models, Animal
  • Dopaminergic Neurons / drug effects*
  • Dopaminergic Neurons / metabolism
  • Dopaminergic Neurons / pathology
  • Mesencephalon / metabolism
  • Mesencephalon / pathology
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Mutation / genetics
  • Nerve Degeneration / metabolism
  • Nerve Degeneration / pathology
  • Nerve Degeneration / prevention & control*
  • Oncogene Proteins / genetics
  • Oncogene Proteins / metabolism
  • Oncogene Proteins / pharmacology*
  • Oxidopamine / metabolism
  • Oxidopamine / pharmacology*
  • Parkinson Disease / metabolism
  • Parkinson Disease / pathology
  • Peroxiredoxins / genetics
  • Peroxiredoxins / metabolism
  • Peroxiredoxins / pharmacology*
  • Protein Deglycase DJ-1
  • Up-Regulation
  • alpha-Synuclein / metabolism
  • alpha-Synuclein / pharmacology*

Substances

  • Oncogene Proteins
  • alpha-Synuclein
  • Oxidopamine
  • Peroxiredoxins
  • PARK7 protein, mouse
  • Protein Deglycase DJ-1