Article abstract


Nature Cell Biology 9, 402 - 414 (2007)
Published online: 25 March 2007 | doi:10.1038/ncb1553

Proteome analysis of soluble nuclear proteins reveals that HMGB1/2 suppress genotoxic stress in polyglutamine diseases

Mei-Ling Qi1,2,8, Kazuhiko Tagawa1,9, Yasushi Enokido1,9, Natsue Yoshimura1, Yo-ichi Wada1, Kei Watase2, Sho-ichi Ishiura3, Ichiro Kanazawa4, Juan Botas5, Minoru Saitoe6, Erich E. Wanker7 & Hitoshi Okazawa1,2,6,8


Nuclear dysfunction is a key feature of the pathology of polyglutamine (polyQ) diseases. It has been suggested that mutant polyQ proteins impair functions of nuclear factors by interacting with them directly in the nucleus. However, a systematic analysis of quantitative changes in soluble nuclear proteins in neurons expressing mutant polyQ proteins has not been performed. Here, we perform a proteome analysis of soluble nuclear proteins prepared from neurons expressing huntingtin (Htt) or ataxin-1 (AT1) protein, and show that mutant AT1 and Htt similarly reduce the concentration of soluble high mobility group B1/2 (HMGB1/2) proteins. Immunoprecipitation and pulldown assays indicate that HMGBs interact with mutant AT1 and Htt. Immunohistochemistry showed that these proteins were reduced in the nuclear region outside of inclusion bodies in affected neurons. Compensatory expression of HMGBs ameliorated polyQ-induced pathology in primary neurons and in Drosophila polyQ models. Furthermore, HMGBs repressed genotoxic stress signals induced by mutant Htt or transcriptional repression. Thus, HMGBs may be critical regulators of polyQ disease pathology and could be targets for therapy development.

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  1. Department of Neuropathology, Medical Research Institute, Tokyo Medical and Dental University, Japan.
  2. 21st Century COE Program for Brain Integration and Its Dysfunction, Tokyo Medical and Dental University, Japan.
  3. Faculty of Culture and Science, The University of Tokyo, Japan.
  4. National Center for Neurology and Psychiatry, Japan.
  5. Department of Molecular and Human Genetics, Baylor College of Medicine, USA.
  6. Department of Molecular Therapeutics, Tokyo Metropolitan Institute for Neuroscience, Japan.
  7. Department of Neuroproteomics, Max-Delbrück Center for Molecular Medicine, Germany.
  8. PRESTO, Japan Science and Technology Agency (JST), Japan.
  9. These authors contributed equally to this work.

Correspondence to: Hitoshi Okazawa1,2,6,8 e-mail: okazawa-tky@umin.ac.jp



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