APC/C-Cdh1 coordinates neurogenesis and cortical size during development

Nat Commun. 2013;4:2879. doi: 10.1038/ncomms3879.

Abstract

The morphology of the adult brain is the result of a delicate balance between neural progenitor proliferation and the initiation of neurogenesis in the embryonic period. Here we assessed whether the anaphase-promoting complex/cyclosome (APC/C) cofactor, Cdh1--which regulates mitosis exit and G1-phase length in dividing cells--regulates neurogenesis in vivo. We use an embryo-restricted Cdh1 knockout mouse model and show that functional APC/C-Cdh1 ubiquitin ligase activity is required for both terminal differentiation of cortical neurons in vitro and neurogenesis in vivo. Further, genetic ablation of Cdh1 impairs the ability of APC/C to promote neurogenesis by delaying the exit of the progenitor cells from the cell cycle. This causes replicative stress and p53-mediated apoptotic death resulting in decreased number of cortical neurons and cortex size. These results demonstrate that APC/C-Cdh1 coordinates cortical neurogenesis and size, thus posing Cdh1 in the molecular pathogenesis of congenital neurodevelopmental disorders, such as microcephaly.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Anaphase-Promoting Complex-Cyclosome / genetics
  • Anaphase-Promoting Complex-Cyclosome / metabolism*
  • Animals
  • Apoptosis
  • Cdh1 Proteins / genetics
  • Cdh1 Proteins / metabolism*
  • Cell Cycle
  • Cell Cycle Proteins / genetics
  • Cell Cycle Proteins / metabolism
  • Cell Division
  • Cerebral Cortex / cytology
  • Cerebral Cortex / embryology*
  • Cerebral Cortex / enzymology
  • Cerebral Cortex / metabolism
  • Female
  • Male
  • Mice
  • Mice, Knockout
  • Neurogenesis*
  • Neurons / cytology*
  • Neurons / enzymology
  • Neurons / metabolism
  • Organ Size

Substances

  • Cdh1 Proteins
  • Cell Cycle Proteins
  • Fzr1 protein, mouse
  • Anaphase-Promoting Complex-Cyclosome