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Axon growth is required for the proper establishment of cortical circuits. Malformations in axonal growth and pathfinding might lead to severe neuropathologies, such as corpus callosum dysgenesis. <jats:italic>Cenpj<\/jats:italic>, a microcephaly gene, encodes a scaffold protein that regulates centrosome biogenesis and microtubule stabilization. During corticogenesis, <jats:italic>Cenpj<\/jats:italic> regulates progenitor division and neuronal migration. Since microtubule stabilization is crucial for axon extension, we investigated the role of <jats:italic>Cenpj<\/jats:italic> in axon growth during cortical development in a mouse model. Through loss\u2010 and gain\u2010of\u2010function assays ex vivo and in utero, we quantified callosal axonal length, branching, and growth cone size compared to controls. We observed that silencing <jats:italic>Cenpj<\/jats:italic> results in an increased axonal length. Ex vivo, we assessed the number of branches, the area of growth cones and the stability of microtubules. In silenced <jats:italic>Cenpj<\/jats:italic> axons, there were more branches, larger growth cone area, and more stable microtubules. Rescue experiments confirmed that neurons present axonal length comparable to controls. Here we propose that <jats:italic>Cenpj<\/jats:italic> regulates axon growth by destabilizing microtubules during cortical development. Finally, our findings suggest that Cenpj might be a novel target for axonal regeneration.\n<\/jats:p><\/jats:sec><jats:sec><jats:label\/><jats:p>\n<jats:boxed-text content-type=\"graphic\" position=\"anchor\"><jats:graphic xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" mimetype=\"image\/png\" position=\"anchor\" specific-use=\"enlarged-web-image\" xlink:href=\"graphic\/jnc15568-fig-0009-m.png\"><jats:alt-text>image<\/jats:alt-text><\/jats:graphic><\/jats:boxed-text><\/jats:p><\/jats:sec>","DOI":"10.1111\/jnc.15568","type":"journal-article","created":{"date-parts":[[2021,12,23]],"date-time":"2021-12-23T15:42:10Z","timestamp":1640274130000},"page":"320-334","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Microcephaly gene <i>Cenpj<\/i> regulates axonal growth in cortical neurons through microtubule destabilization"],"prefix":"10.1111","volume":"161","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1903-5169","authenticated-orcid":false,"given":"Pamela","family":"Meneses Iack","sequence":"first","affiliation":[{"name":"Instituto de Ci\u00eancias Biom\u00e9dicas Laborat\u00f3rio de Neuroplasticidade Centro de Ci\u00eancias da Sa\u00fade Bloco F Universidade Federal do Rio de Janeiro Cidade Universit\u00e1ria  Rio de Janeiro RJ Brazil"},{"name":"Instituto de Biof\u00edsica Carlos Chagas Filho Laborat\u00f3rio de Neuroqu\u00edmica Centro de Ci\u00eancias da Sa\u00fade Universidade Federal do Rio de Janeiro Bloco C Cidade Universit\u00e1ria  Rio de Janeiro RJ Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8014-9854","authenticated-orcid":false,"given":"Danielle","family":"Ray\u00eae","sequence":"additional","affiliation":[{"name":"Instituto de Ci\u00eancias Biom\u00e9dicas Laborat\u00f3rio de Neuroplasticidade Centro de Ci\u00eancias da Sa\u00fade Bloco F Universidade Federal do Rio de Janeiro Cidade Universit\u00e1ria  Rio de Janeiro RJ Brazil"},{"name":"Department of Ophthalmology and Visual Sciences Albert Einstein College of Medicine  New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8989-4009","authenticated-orcid":false,"given":"Roberto","family":"Lent","sequence":"additional","affiliation":[{"name":"Instituto de Ci\u00eancias Biom\u00e9dicas Laborat\u00f3rio de Neuroplasticidade Centro de Ci\u00eancias da Sa\u00fade Bloco F Universidade Federal do Rio de Janeiro Cidade Universit\u00e1ria  Rio de Janeiro RJ Brazil"},{"name":"D\u2019Or Institute for Research and Education  Rio de Janeiro RJ Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1135-4149","authenticated-orcid":false,"given":"Victor 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