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Complete loss of function of this gene is not compatible with life. On the other hand, missense and copy number variants of <jats:italic>CHD1<\/jats:italic> can result in intellectual disabilities and craniofacial malformations in human patients including cleft palate and Pilarowski\u2013Bjornsson Syndrome. We have used the aquatic developmental model organism <jats:italic>Xenopus laevis<\/jats:italic>, to determine a specific role for Chd1 in such cranioafcial disorders. Protein and gene knockdown techniques in <jats:italic>Xenopus<\/jats:italic>, including antisense oligos and mosaic Crispr\/Cas9\u2010mediated mutagenesis, recapitulated the craniofacial defects observed in humans. Further analysis indicated that embryos deficient in Chd1 had defects in cranial neural crest development and jaw cartilage morphology. Additionally, flow cytometry and immunohistochemistry revealed that decreased Chd1 resulted in increased in apoptosis in the developing head. Together, these experiments demonstrate that Chd1 is critical for fundamental processes and cell survival in craniofacial development. We also presented evidence that Chd1 is regulated by retinoic acid signaling during craniofacial development. Expression levels of <jats:italic>chd1<\/jats:italic> mRNA, specifically in the head, were increased by RAR agonist exposure and decreased upon antagonist treatment. Subphenotypic levels of an RAR antagonist and Chd1 morpholinos synergized to result in orofacial defects. Further, RAR DNA binding sequences (RAREs) were detected in <jats:italic>chd1<\/jats:italic> regulatory regions by bioinformatic analysis. In summary, by combining human genetics and experiments in an aquatic model we now have a better understanding of the role of CHD1 in craniofacial disorders.<\/jats:p>","DOI":"10.1002\/dvg.23394","type":"journal-article","created":{"date-parts":[[2020,9,18]],"date-time":"2020-09-18T07:31:52Z","timestamp":1600414312000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Using an aquatic model, <scp><i>Xenopus laevis<\/i><\/scp>, to uncover the role of chromodomain 1 in craniofacial disorders"],"prefix":"10.1002","volume":"59","author":[{"given":"Brent H.","family":"Wyatt","sequence":"first","affiliation":[{"name":"Department of Biology Virginia Commonwealth University  Richmond Virginia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas O.","family":"Raymond","sequence":"additional","affiliation":[{"name":"Department of Biology Virginia Commonwealth University  Richmond Virginia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lisa A.","family":"Lansdon","sequence":"additional","affiliation":[{"name":"Department of Biology University of Iowa  Iowa City Iowa USA"},{"name":"Department of Pathology and Laboratory Medicine Children's Mercy Hospital  Kansas City Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Benjamin W.","family":"Darbro","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey C.","family":"Murray","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John Robert","family":"Manak","sequence":"additional","affiliation":[{"name":"Department of Biology University of Iowa  Iowa City Iowa USA"},{"name":"Department of Pediatrics University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1427-9140","authenticated-orcid":false,"given":"Amanda J. 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