| Literature DB >> 32675289 |
Mariacristina Capizzi1, Esther Aparicio1, Monia Barnat1, Susana Boluda2, Doris Wennagel1, Radhia Kacher1, Rayane Kassem1, Sophie Lenoir1, Fabienne Agasse1, Barbara Y Braz1, Jeh-Ping Liu3, Julien Ighil4, Aude Tessier5, Scott O Zeitlin3, Charles Duyckaerts2, Marc Dommergues4, Alexandra Durr6, Sandrine Humbert7.
Abstract
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.Entities:
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Year: 2020 PMID: 32675289 PMCID: PMC7859879 DOI: 10.1126/science.aax3338
Source DB: PubMed Journal: Science ISSN: 0036-8075 Impact factor: 47.728