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Its development relies on a series of steps including neural progenitor cell proliferation, neuronal migration, axonal outgrowth and brain wiring. Disruption of these steps leads to cortical malformations, often associated with intellectual disability and epilepsy. We have generated a new resource to shed further light on subcortical heterotopia, a malformation characterized by abnormal neuronal position. We describe here the generation and characterization of a knockout (<jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content>) mouse model for <jats:italic>Eml<\/jats:italic>1, a microtubule\u2010associated protein showing mutations in human ribbon\u2010like subcortical heterotopia. As previously reported for a spontaneous mouse mutant showing a mutation in <jats:italic>Eml<\/jats:italic>1, we observe severe cortical heterotopia in the <jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content>. We also observe abnormal progenitor cells in early corticogenesis, likely to be the origin of the defects. <jats:italic>EML<\/jats:italic>1 <jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content> mice on the C57<jats:styled-content style=\"fixed-case\">BL<\/jats:styled-content>\/6N genetic background also appear to present a wider phenotype than the original mouse mutant, showing additional brain anomalies, such as corpus callosum abnormalities. We compare the anatomy of male and female mice and also study heterozygote animals. This new resource will help unravel roles for <jats:italic>Eml<\/jats:italic>1 in brain development and tissue architecture, as well as the mechanisms leading to severe subcortical heterotopia.<\/jats:p>","DOI":"10.1111\/joa.13013","type":"journal-article","created":{"date-parts":[[2019,6,7]],"date-time":"2019-06-07T09:15:09Z","timestamp":1559898909000},"page":"637-650","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":27,"title":["The neuroanatomy of <i>Eml<\/i>1 knockout mice, a model of subcortical heterotopia"],"prefix":"10.1111","volume":"235","author":[{"given":"Stephan C.","family":"Collins","sequence":"first","affiliation":[{"name":"Institut de G\u00e9n\u00e9tique et de Biologie Mol\u00e9culaire et Cellulaire Illkirch France"},{"name":"Centre National de la Recherche Scientifique UMR7104 Illkirch France"},{"name":"Institut National de la Sant\u00e9 et de la Recherche M\u00e9dicale, U964 Illkirch 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