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Here we show that <jats:italic>Cenpj<\/jats:italic>, also known as <jats:italic>CPAP<\/jats:italic>, a microcephaly gene, is a transcriptional target of Ascl1 in the embryonic cerebral cortex. We have characterized the role of <jats:italic>Cenpj<\/jats:italic> during cortical development by <jats:italic>in utero<\/jats:italic> electroporation knockdown and found that silencing <jats:italic>Cenpj<\/jats:italic> in the ventricular zone disrupts centrosome biogenesis and randomizes the cleavage plane orientation of radial glia progenitors. Moreover, we show that downregulation of <jats:italic>Cenpj<\/jats:italic> in post-mitotic neurons increases stable microtubules and leads to slower neuronal migration, abnormal centrosome position and aberrant neuronal morphology. Moreover, rescue experiments shows that <jats:italic>Cenpj<\/jats:italic> mediates the role of Ascl1 in centrosome biogenesis in progenitor cells and in microtubule dynamics in migrating neurons. These data provide insights into genetic pathways controlling cortical development and primary microcephaly observed in humans with mutations in <jats:italic>Cenpj.<\/jats:italic><\/jats:p>","DOI":"10.1038\/ncomms7474","type":"journal-article","created":{"date-parts":[[2015,3,10]],"date-time":"2015-03-10T11:16:42Z","timestamp":1425986202000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":65,"title":["Cenpj\/CPAP regulates progenitor divisions and neuronal migration in the cerebral cortex downstream of Ascl1"],"prefix":"10.1038","volume":"6","author":[{"given":"Patricia P.","family":"Garcez","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Javier","family":"Diaz-Alonso","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ivan","family":"Crespo-Enriquez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Diogo","family":"Castro","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donald","family":"Bell","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fran\u00e7ois","family":"Guillemot","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2015,3,10]]},"reference":[{"key":"BFncomms7474_CR1","doi-asserted-by":"publisher","first-page":"509","DOI":"10.1002\/ajmg.1320180320","volume":"18","author":"WB Dobyns","year":"1984","unstructured":"Dobyns, W. 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