PARIS (ZNF746) repression of PGC-1α contributes to neurodegeneration in Parkinson's disease

Cell. 2011 Mar 4;144(5):689-702. doi: 10.1016/j.cell.2011.02.010.

Abstract

A hallmark of Parkinson's disease (PD) is the preferential loss of substantia nigra dopamine neurons. Here, we identify a new parkin interacting substrate, PARIS (ZNF746), whose levels are regulated by the ubiquitin proteasome system via binding to and ubiquitination by the E3 ubiquitin ligase, parkin. PARIS is a KRAB and zinc finger protein that accumulates in models of parkin inactivation and in human PD brain. PARIS represses the expression of the transcriptional coactivator, PGC-1α and the PGC-1α target gene, NRF-1 by binding to insulin response sequences in the PGC-1α promoter. Conditional knockout of parkin in adult animals leads to progressive loss of dopamine (DA) neurons in a PARIS-dependent manner. Moreover, overexpression of PARIS leads to the selective loss of DA neurons in the substantia nigra, and this is reversed by either parkin or PGC-1α coexpression. The identification of PARIS provides a molecular mechanism for neurodegeneration due to parkin inactivation.

Publication types

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

MeSH terms

  • Animals
  • Brain / metabolism
  • Brain / pathology
  • Dopamine / metabolism
  • Humans
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • NF-E2-Related Factor 1 / metabolism
  • Neurodegenerative Diseases / metabolism
  • Neurons / metabolism
  • Nuclear Respiratory Factor 1 / metabolism
  • Parkinson Disease / metabolism*
  • Peroxisome Proliferator-Activated Receptor Gamma Coactivator 1-alpha
  • Rats
  • Repressor Proteins / metabolism*
  • Trans-Activators / metabolism*
  • Transcription Factors
  • Ubiquitin-Protein Ligases / metabolism

Substances

  • NF-E2-Related Factor 1
  • Nrf1 protein, mouse
  • Nuclear Respiratory Factor 1
  • Peroxisome Proliferator-Activated Receptor Gamma Coactivator 1-alpha
  • Ppargc1a protein, mouse
  • Repressor Proteins
  • Trans-Activators
  • Transcription Factors
  • ZNF746 protein, mouse
  • Ubiquitin-Protein Ligases
  • parkin protein
  • Dopamine