Intranuclear degradation of polyglutamine aggregates by the ubiquitin-proteasome system

J Biol Chem. 2009 Apr 10;284(15):9796-803. doi: 10.1074/jbc.M809739200. Epub 2009 Feb 13.

Abstract

Huntington disease and its related autosomal-dominant polyglutamine (pQ) neurodegenerative diseases are characterized by intraneuronal accumulation of protein aggregates. Studies on protein aggregates have revealed the importance of the ubiquitin-proteasome system as the front line of protein quality control (PQC) machinery against aberrant proteins. Recently, we have shown that the autophagy-lysosomal system is also involved in cytoplasmic aggregate degradation, but the nucleus lacked this activity. Consequently, the nucleus relies entirely on the ubiquitin-proteasome system for PQC. According to previous studies, nuclear aggregates possess a higher cellular toxicity than do their cytoplasmic counterparts, however degradation kinetics of nuclear aggregates have been poorly understood. Here we show that nuclear ubiquitin ligases San1p and UHRF-2 each enhance nuclear pQ aggregate degradation and rescued pQ-induced cytotoxicity in cultured cells and primary neurons. Moreover, UHRF-2 is associated with nuclear inclusion bodies in vitro and in vivo. Our data suggest that UHRF-2 is an essential molecule for nuclear pQ degradation as a component of nuclear PQC machinery in mammalian cells.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cell Nucleus / metabolism*
  • Cells, Cultured / metabolism
  • Cytoplasm / metabolism
  • HeLa Cells
  • Humans
  • Kinetics
  • Neurodegenerative Diseases / metabolism*
  • Neurons / metabolism
  • Peptides / chemistry*
  • Proteasome Endopeptidase Complex / chemistry*
  • RNA Interference
  • Reverse Transcriptase Polymerase Chain Reaction
  • Saccharomyces cerevisiae Proteins / metabolism
  • Ubiquitin / chemistry*
  • Ubiquitin / metabolism
  • Ubiquitin-Protein Ligases / metabolism

Substances

  • Peptides
  • Saccharomyces cerevisiae Proteins
  • Ubiquitin
  • polyglutamine
  • SAN1 protein, S cerevisiae
  • UHRF2 protein, human
  • Ubiquitin-Protein Ligases
  • Proteasome Endopeptidase Complex