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The V471A Polymorphism in Autophagy-Related Gene ATG7 Modifies Age at Onset Specifically in Italian Huntington Disease Patients

  • The REGISTRY investigators of the European Huntington's Disease Network
  • University of Tubingen
  • Leiden University
  • Panum Institute
  • University of Manchester
  • University of Vienna
  • Foundation IRCCS Neurological Institute C. Besta Milan
  • Rehazentrum
  • University of Innsbruck
  • University of Pécs
  • Ruhr-University Bochum
  • Institute of Psychiatry and Neurology, Warszawa
  • Boʇaziçi University
  • University and Teaching Hospital
  • University of Brussels
  • SAS Automotive Ltd.
  • Banska Bystrica
  • University of Regensburg
  • Klinikum für Psychiatrie
  • BKHTaufkirchen
  • Hannover University of Music
  • Université Paris Est
  • Columbus-Gemelli University Hospital
  • Catholic University of the Sacred Heart
  • Institute of Genetics and Biomedical Research-National Research Council
  • Sigmund Freud University
  • Medizinische Universität Graz
  • Swiss Huntington's Disease Centre
  • University of Bern
  • Swiss HD Zentrum
  • Brain Repair Group
  • Cardiff University
  • Department of Neurology
  • Rikshospitalet-Radiumhospitalet HF
  • University of Rostock
  • University of Ulm
  • University of Massachusetts Medical School
  • Hospital Virgen del Camino
  • University of Copenhagen, Denmark
  • University of Oslo
  • University of Turku
  • Barcelona-Hospital Mútua de Terrassa
  • University of London
  • KU Leuven
  • Institut de Pathologie et de Génétique
  • Charles University
  • Sahlgrenska University Hospital/Östra
  • Copenhagen University Hospital Rigshospitalet

Abstract

The cause of Huntington disease (HD) is a polyglutamine repeat expansion of more than 36 units in the huntingtin protein, which is inversely correlated with the age at onset of the disease. However, additional genetic factors are believed to modify the course and the age at onset of HD. Recently, we identified the V471A polymorphism in the autophagy-related gene ATG7, a key component of the autophagy pathway that plays an important role in HD pathogenesis, to be associated with the age at onset in a large group of European Huntington disease patients. To confirm this association in a second independent patient cohort, we analysed the ATG7 V471A polymorphism in additional 1,464 European HD patients of the "REGISTRY" cohort from the European Huntington Disease Network (EHDN). In the entire REGISTRY cohort we could not confirm a modifying effect of the ATG7 V471A polymorphism. However, analysing a modifying effect of ATG7 in these REGISTRY patients and in patients of our previous HD cohort according to their ethnic origin, we identified a significant effect of the ATG7 V471A polymorphism on the HD age at onset only in the Italian population (327 patients). In these Italian patients, the polymorphism is associated with a 6-years earlier disease onset and thus seems to have an aggravating effect. We could specify the role of ATG7 as a genetic modifier for HD particularly in the Italian population. This result affirms the modifying influence of the autophagic pathway on the course of HD, but also suggests population-specific modifying mechanisms in HD pathogenesis.

Original languageEnglish
Article numbere68951
JournalPLoS ONE
Volume8
Issue number7
ISSN1553-7390
DOIs
Publication statusPublished - 22.07.2013

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

  1. SDG 3 - Good Health and Well-being
    SDG 3 Good Health and Well-being
  2. SDG 10 - Reduced Inequalities
    SDG 10 Reduced Inequalities

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