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Recent studies have revealed severe cases of microcephaly resulting from human mutations in the <jats:italic>NDE1<\/jats:italic> gene, which is involved in the regulation of cytoplasmic dynein. Here using <jats:italic>in utero<\/jats:italic> electroporation of NDE1 short hairpin RNA (shRNA) in embryonic rat brains, we observe cell cycle arrest of proliferating neural progenitors at three distinct stages: during apical interkinetic nuclear migration, at the G2-to-M transition and in regulation of primary cilia at the G1-to-S transition. RNAi against the NDE1 paralogue NDEL1 has no such effects. However, NDEL1 overexpression can functionally compensate for NDE1, except at the G2-to-M transition, revealing a unique NDE1 role. In contrast, NDE1 and NDEL1 RNAi have comparable effects on postmitotic neuronal migration. These results reveal that the severity of <jats:italic>NDE1<\/jats:italic>-associated microcephaly results not from defects in mitosis, but rather the inability of neural progenitors to ever reach this stage.<\/jats:p>","DOI":"10.1038\/ncomms12551","type":"journal-article","created":{"date-parts":[[2016,8,24]],"date-time":"2016-08-24T10:59:06Z","timestamp":1472036346000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":79,"title":["Severe NDE1-mediated microcephaly results from neural progenitor cell cycle arrests at multiple specific stages"],"prefix":"10.1038","volume":"7","author":[{"given":"David J.","family":"Doobin","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shahrnaz","family":"Kemal","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tiago J.","family":"Dantas","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard B.","family":"Vallee","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2016,8,24]]},"reference":[{"key":"BFncomms12551_CR1","doi-asserted-by":"publisher","first-page":"1348","DOI":"10.1093\/brain\/aws019","volume":"135","author":"AJ Barkovich","year":"2012","unstructured":"Barkovich, A. 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