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Huntington's disease alters human neurodevelopment.

Monia BarnatMariacristina CapizziEsther AparicioSusana BoludaDoris WennagelRadhia KacherRayane KassemSophie LenoirFabienne AgasseBarbara Yael BrazJeh-Ping LiuJulien IghilAude TessierScott O ZeitlinCharles DuyckaertsMarc DommerguesAlexandra DürrFrédéric Saudou
Published in: Science (New York, N.Y.) (2020)
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.
Keyphrases
  • cell cycle
  • endothelial cells
  • stem cells
  • gene expression
  • cell proliferation
  • single cell
  • preterm infants
  • congenital heart disease
  • pluripotent stem cells