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Huntington's disease alters human neurodevelopment.
Barnat, Monia; Capizzi, Mariacristina; Aparicio, Esther; Boluda, Susana; Wennagel, Doris; Kacher, Radhia; Kassem, Rayane; Lenoir, Sophie; Agasse, Fabienne; Braz, Barbara Y; Liu, Jeh-Ping; Ighil, Julien; Tessier, Aude; Zeitlin, Scott O; Duyckaerts, Charles; Dommergues, Marc; Durr, Alexandra; Humbert, Sandrine.
Afiliación
  • Barnat M; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Capizzi M; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Aparicio E; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Boluda S; Department of Neuropathology Raymond Escourolle, AP-HP, Pitié-Salpêtrière University Hospital, Paris, France.
  • Wennagel D; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Kacher R; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Kassem R; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Lenoir S; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Agasse F; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Braz BY; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France.
  • Liu JP; Department of Neuroscience, University of Virginia School of Medicine, Charlottesville, VA 22908, USA.
  • Ighil J; AP-HP, Sorbonne University, Service de Gynécologie Obstétrique, Pitié-Salpêtrière Hospital, Paris, France.
  • Tessier A; AP-HP, Unité d'Embryofoetopathologie, Necker Hospital, Paris, France.
  • Zeitlin SO; Department of Neuroscience, University of Virginia School of Medicine, Charlottesville, VA 22908, USA.
  • Duyckaerts C; Department of Neuropathology Raymond Escourolle, AP-HP, Pitié-Salpêtrière University Hospital, Paris, France.
  • Dommergues M; AP-HP, Sorbonne University, Service de Gynécologie Obstétrique, Pitié-Salpêtrière Hospital, Paris, France.
  • Durr A; Sorbonne University, Paris Brain Institute, APHP, INSERM U1127, CNRS UMR7225, Pitié-Salpêtrière Hospital, Paris, France alexandra.durr@upmc.fr sandrine.humbert@inserm.fr.
  • Humbert S; Univ. Grenoble Alpes, INSERM, U1216, Grenoble Institut Neurosciences, Grenoble, France. alexandra.durr@upmc.fr sandrine.humbert@inserm.fr.
Science ; 369(6505): 787-793, 2020 08 14.
Article en En | MEDLINE | ID: mdl-32675289
Although Huntington's disease is a late-manifesting neurodegenerative disorder, both mouse studies and neuroimaging studies of presymptomatic mutation carriers suggest that Huntington's disease might affect neurodevelopment. To determine whether this is actually the case, we examined tissue from human fetuses (13 weeks gestation) that carried the Huntington's disease mutation. These tissues showed clear abnormalities in the developing cortex, including mislocalization of mutant huntingtin and junctional complex proteins, defects in neuroprogenitor cell polarity and differentiation, abnormal ciliogenesis, and changes in mitosis and cell cycle progression. We observed the same phenomena in Huntington's disease mouse embryos, where we linked these abnormalities to defects in interkinetic nuclear migration of progenitor cells. Huntington's disease thus has a neurodevelopmental component and is not solely a degenerative disease.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Enfermedad de Huntington / Proteína Huntingtina / Sistema Nervioso Límite: Animals / Humans Idioma: En Revista: Science Año: 2020 Tipo del documento: Article País de afiliación: Francia

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Enfermedad de Huntington / Proteína Huntingtina / Sistema Nervioso Límite: Animals / Humans Idioma: En Revista: Science Año: 2020 Tipo del documento: Article País de afiliación: Francia