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In addition to autism, patients with putative loss of function variants in DYRK1A exhibit microcephaly, intellectual disability, developmental delay, and\/or congenital anomalies of the kidney and urinary tract. DYRK1A is also located within the critical region for Down syndrome; therefore, understanding the role of DYRK1A in brain development is crucial for understanding the pathobiology of multiple developmental disorders. To characterize the function of this gene, we used the diploid frog, Xenopus tropicalis. We discover that Dyrk1a is expressed in ciliated tissues, localizes to ciliary axonemes and basal bodies, and is required for ciliogenesis. We also demonstrate that Dyrk1a localizes to mitotic spindles and that its inhibition leads to decreased forebrain size, abnormal cell cycle progression, and cell death during brain development. These findings provide hypotheses about potential mechanisms of pathobiology and underscore the utility of X. tropicalis as a model system for understanding neurodevelopmental disorders.<\/jats:p>","DOI":"10.1242\/dev.189290","type":"journal-article","created":{"date-parts":[[2020,5,28]],"date-time":"2020-05-28T13:46:16Z","timestamp":1590673576000},"source":"Crossref","is-referenced-by-count":36,"title":["Neurodevelopmental disorder risk gene <i>DYRK1A<\/i> is required for ciliogenesis and brain size in <i>Xenopus<\/i> embryos"],"prefix":"10.1242","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8404-3291","authenticated-orcid":false,"given":"Helen Rankin","family":"Willsey","sequence":"first","affiliation":[{"name":"Department of Psychiatry, Weill Institute for Neurosciences, Quantitative Biosciences Institute, University of California San Francisco, San Francisco, CA, USA"},{"name":"Department of Molecular and Cell Biology, University of California, Berkeley, Berkeley, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuxiao","family":"Xu","sequence":"additional","affiliation":[{"name":"Department of Psychiatry, Weill Institute for Neurosciences, Quantitative Biosciences Institute, University of California San Francisco, San Francisco, CA, USA"},{"name":"Department of Molecular and Cell Biology, University of California, Berkeley, Berkeley, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amanda","family":"Everitt","sequence":"additional","affiliation":[{"name":"Department of Psychiatry, Institute for Neurodegenerative Diseases, Weill Institute for Neurosciences, Quantitative Biosciences Institute, University of California San Francisco, San Francisco, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeanselle","family":"Dea","sequence":"additional","affiliation":[{"name":"Department of Psychiatry, Weill Institute for Neurosciences, Quantitative Biosciences Institute, University of California San Francisco, San Francisco, CA, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cameron R. 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