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U.S.A."],"published-print":{"date-parts":[[2019,11,5]]},"abstract":"<jats:title>Significance<\/jats:title>\n                  <jats:p>\n                    Thyroid hormone deficiencies are the most common preventable causes of intellectual disability. We report that mutations in the thyroid hormone receptor \u03b11 gene (\n                    <jats:italic>THRA<\/jats:italic>\n                    ) that result in intellectual disability also reduce brain size. Using human\n                    <jats:italic>THRA<\/jats:italic>\n                    mutation stem cell models, we studied the impact of\n                    <jats:italic>THRA<\/jats:italic>\n                    mutations on human brain development by combining quantitative lineage analysis, gene expression analyses, and novel assays of neuroepithelium formation. We found that\n                    <jats:italic>THRA<\/jats:italic>\n                    regulates the balance between progenitor self-renewal and neurogenesis, and thus overall brain size. Importantly, these in vitro results are consistent with in vivo evidence from magnetic resonance imaging of people with these mutations, advancing our understanding of thyroid hormone action in human brain development.\n                  <\/jats:p>","DOI":"10.1073\/pnas.1908762116","type":"journal-article","created":{"date-parts":[[2019,10,18]],"date-time":"2019-10-18T19:30:08Z","timestamp":1571427008000},"page":"22754-22763","update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":46,"title":["Mutations in thyroid hormone receptor \u03b11 cause premature neurogenesis and progenitor cell depletion in human cortical development"],"prefix":"10.1073","volume":"116","author":[{"given":"Teresa G.","family":"Krieger","sequence":"first","affiliation":[{"name":"Gurdon Institute, University of Cambridge, Cambridge CB2 1QN, United Kingdom;"},{"name":"Cavendish Laboratory, University of Cambridge, Cambridge CB3 0HE, United Kingdom;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carla M.","family":"Moran","sequence":"additional","affiliation":[{"name":"Wellcome Trust-MRC Institute of Metabolic Science, University of Cambridge, Cambridge CB2 0QQ, United Kingdom;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alberto","family":"Frangini","sequence":"additional","affiliation":[{"name":"Gurdon Institute, University of Cambridge, Cambridge CB2 1QN, United Kingdom;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"W. 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