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Natl. Acad. Sci. U.S.A."],"published-print":{"date-parts":[[2017,10,24]]},"abstract":"<jats:title>Significance<\/jats:title>\n                  <jats:p>Several chromatin-modifying enzymes are mutated in human craniofacial disorders. These factors function genome-wide to regulate accessibility and expression of extensive gene sets. Therefore, understanding chromatin-modifier function requires identification of responsible cellular origins and genomic characterization of phenotypes in primary cells. We now combine reporter flow cytometry with low cell number genomics to identify neural crest stem-cell factors as molecular targets of UTX in Kabuki syndrome. UTX demethylates histones to regulate some gene expression, but many UTX-bound regions are subject to novel mechanisms of transcriptional regulation. This study identifies UTX cellular and molecular targets in craniofacial development and this methodology is broadly adaptable to study genome distribution of other chromatin factors in neural crest disorders.<\/jats:p>","DOI":"10.1073\/pnas.1705011114","type":"journal-article","created":{"date-parts":[[2017,10,9]],"date-time":"2017-10-09T15:01:16Z","timestamp":1507561276000},"update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":98,"title":["UTX-guided neural crest function underlies craniofacial features of Kabuki syndrome"],"prefix":"10.1073","volume":"114","author":[{"given":"Karl B.","family":"Shpargel","sequence":"first","affiliation":[{"name":"Department of Genetics, University of North Carolina, Chapel Hill, NC 27599-7264;"},{"name":"Lineberger Comprehensive Cancer Center, University of North Carolina, Chapel Hill, NC 27599-7264;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joshua","family":"Starmer","sequence":"additional","affiliation":[{"name":"Department of Genetics, University of North Carolina, Chapel Hill, NC 27599-7264;"},{"name":"Lineberger Comprehensive Cancer Center, University of North Carolina, Chapel Hill, NC 27599-7264;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chaochen","family":"Wang","sequence":"additional","affiliation":[{"name":"Laboratory of Endocrinology and Receptor Biology, National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, MD 20892"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7442-5138","authenticated-orcid":false,"given":"Kai","family":"Ge","sequence":"additional","affiliation":[{"name":"Laboratory of Endocrinology and Receptor Biology, National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, MD 20892"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0792-835X","authenticated-orcid":false,"given":"Terry","family":"Magnuson","sequence":"additional","affiliation":[{"name":"Department of Genetics, University of North Carolina, Chapel Hill, NC 27599-7264;"},{"name":"Lineberger Comprehensive Cancer Center, University of North Carolina, Chapel Hill, NC 27599-7264;"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"341","published-online":{"date-parts":[[2017,10,9]]},"reference":[{"key":"e_1_3_4_1_2","doi-asserted-by":"crossref","first-page":"409","DOI":"10.1016\/S0046-8177(74)80021-3","article-title":"Neurocristopathies\u2013Unifying concept of disease arising in neural crest maldevelopment","volume":"5","author":"Bolande RP","year":"1974","unstructured":"RP Bolande, Neurocristopathies\u2013Unifying concept of disease arising in neural crest maldevelopment. 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