Literature DB >> 7477379

Polyglutamine expansion as a pathological epitope in Huntington's disease and four dominant cerebellar ataxias.

Y Trottier1, Y Lutz, G Stevanin, G Imbert, D Devys, G Cancel, F Saudou, C Weber, G David, L Tora.   

Abstract

A polyglutamine expansion (encoded by a CAG repeat) in specific proteins causes neurodegeneration in Huntington's disease (HD) and four other disorders, by an unknown mechanism thought to involve gain of function or toxicity of the mutated protein. The pathological threshold is 37-40 glutamines in three of these diseases, whereas the corresponding normal proteins contain polymorphic repeats of up to about 35 glutamines. The age of onset of clinical manifestations is inversely correlated to the length of the polyglutamine expansion. Here we report the characterization of a monoclonal antibody that selectively recognizes polyglutamine expansion in the proteins implicated in HD and in spinocerebellar ataxia (SCA) 1 and 3. The intensity of signal depends on the length of the polyglutamine expansion, and the antibody also detects specific pathological proteins expected to contain such expansion, in SCA2 and in autosomal dominant cerebellar ataxia with retinal degeneration, whose genes have not yet been identified.

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Year:  1995        PMID: 7477379     DOI: 10.1038/378403a0

Source DB:  PubMed          Journal:  Nature        ISSN: 0028-0836            Impact factor:   49.962


  152 in total

Review 1.  Properties of polyglutamine expansion in vitro and in a cellular model for Huntington's disease.

Authors:  A Lunkes; Y Trottier; J Fagart; P Schultz; G Zeder-Lutz; D Moras; J L Mandel
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

Review 2.  Evidence for both the nucleus and cytoplasm as subcellular sites of pathogenesis in Huntington's disease in cell culture and in transgenic mice expressing mutant huntingtin.

Authors:  A S Hackam; J G Hodgson; R Singaraja; T Zhang; L Gan; C A Gutekunst; S M Hersch; M R Hayden
Journal:  Philos Trans R Soc Lond B Biol Sci       Date:  1999-06-29       Impact factor: 6.237

3.  Cellular defects and altered gene expression in PC12 cells stably expressing mutant huntingtin.

Authors:  S H Li; A L Cheng; H Li; X J Li
Journal:  J Neurosci       Date:  1999-07-01       Impact factor: 6.167

4.  Inhibition of huntingtin fibrillogenesis by specific antibodies and small molecules: implications for Huntington's disease therapy.

Authors:  V Heiser; E Scherzinger; A Boeddrich; E Nordhoff; R Lurz; N Schugardt; H Lehrach; E E Wanker
Journal:  Proc Natl Acad Sci U S A       Date:  2000-06-06       Impact factor: 11.205

Review 5.  Hereditary spastic paraparesis: a review of new developments.

Authors:  C McDermott; K White; K Bushby; P Shaw
Journal:  J Neurol Neurosurg Psychiatry       Date:  2000-08       Impact factor: 10.154

6.  Glutamine/proline-rich PQE-1 proteins protect Caenorhabditis elegans neurons from huntingtin polyglutamine neurotoxicity.

Authors:  Peter W Faber; Cindy Voisine; Daphne C King; Emily A Bates; Anne C Hart
Journal:  Proc Natl Acad Sci U S A       Date:  2002-12-16       Impact factor: 11.205

7.  Allele-specific silencing of dominant disease genes.

Authors:  Victor M Miller; Haibin Xia; Ginger L Marrs; Cynthia M Gouvion; Gloria Lee; Beverly L Davidson; Henry L Paulson
Journal:  Proc Natl Acad Sci U S A       Date:  2003-06-02       Impact factor: 11.205

8.  A structural model of polyglutamine determined from a host-guest method combining experiments and landscape theory.

Authors:  John M Finke; Margaret S Cheung; José N Onuchic
Journal:  Biophys J       Date:  2004-09       Impact factor: 4.033

Review 9.  Aggregation of expanded huntingtin in the brains of patients with Huntington disease.

Authors:  Guylaine Hoffner; Sylvie Souès; Philippe Djian
Journal:  Prion       Date:  2007 Jan-Mar       Impact factor: 3.931

10.  Huntington's disease (HD): degeneration of select nuclei, widespread occurrence of neuronal nuclear and axonal inclusions in the brainstem.

Authors:  Udo Rüb; Matthias Hentschel; Katharina Stratmann; Ewout Brunt; Helmut Heinsen; Kay Seidel; Mohamed Bouzrou; Georg Auburger; Henry Paulson; Jean-Paul Vonsattel; Herwig Lange; Horst-Werner Korf; Wilfred den Dunnen
Journal:  Brain Pathol       Date:  2014-03-03       Impact factor: 6.508

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