{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,22]],"date-time":"2026-10-22T19:31:49Z","timestamp":1792697509354,"version":"4.5.0"},"reference-count":49,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2014,7,10]],"date-time":"2014-07-10T00:00:00Z","timestamp":1404950400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/3.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["DC009410"],"award-info":[{"award-number":["DC009410"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Developmental Dynamics"],"published-print":{"date-parts":[[2014,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    <jats:styled-content>Background:<\/jats:styled-content>\n                    Heterozygous mutations in the chromatin remodeling gene\n                    <jats:italic>CHD7<\/jats:italic>\n                    cause CHARGE syndrome, a developmental disorder with variable craniofacial dysmorphisms and respiratory difficulties. The molecular etiologies of these malformations are not well understood. Homozygous\n                    <jats:italic>Chd7<\/jats:italic>\n                    null mice die by E11, whereas\n                    <jats:italic>\n                      Chd7\n                      <jats:sup>Gt\/+<\/jats:sup>\n                    <\/jats:italic>\n                    heterozygous null mice are a viable and excellent model of CHARGE. We explored skeletal phenotypes in\n                    <jats:italic>\n                      Chd7\n                      <jats:sup>Gt\/+<\/jats:sup>\n                    <\/jats:italic>\n                    and\n                    <jats:italic>Chd7<\/jats:italic>\n                    conditional knockout mice, using\n                    <jats:italic>Foxg1\u2010Cre<\/jats:italic>\n                    to delete\n                    <jats:italic>Chd7<\/jats:italic>\n                    (\n                    <jats:italic>Foxg1<\/jats:italic>\n                    \u2010CKO) in the developing eye, ear, nose, pharyngeal pouch, forebrain, and gut and\n                    <jats:italic>Wnt1\u2010Cre<\/jats:italic>\n                    (\n                    <jats:italic>Wnt1<\/jats:italic>\n                    \u2010CKO) to delete\n                    <jats:italic>Chd7<\/jats:italic>\n                    in migrating neural crest cells.\n                    <jats:styled-content>Results:<\/jats:styled-content>\n                    <jats:italic>Foxg1<\/jats:italic>\n                    \u2010CKO mice exhibited postnatal respiratory distress and death, dysplasia of the eye, concha, and frontal bone, hypoplastic maxillary shelves and nasal epithelia, and reduced tracheal rings.\n                    <jats:italic>Wnt1\u2010<\/jats:italic>\n                    CKO mice exhibited frontal and occipital bone dysplasia, hypoplasia of the maxillary shelves and mandible, and cleft palate. In contrast, heterozygous\n                    <jats:italic>\n                      Chd7\n                      <jats:sup>Gt\/+<\/jats:sup>\n                    <\/jats:italic>\n                    mice had apparently normal skeletal development.\n                    <jats:styled-content>Conclusions:<\/jats:styled-content>\n                    Conditional deletion of\n                    <jats:italic>Chd7<\/jats:italic>\n                    in ectodermal and endodermal derivatives (\n                    <jats:italic>Foxg1\u2010Cre<\/jats:italic>\n                    ) or migrating neural crest cells (\n                    <jats:italic>Wnt1\u2010Cre<\/jats:italic>\n                    ) results in varied and more severe craniofacial defects than in\n                    <jats:italic>\n                      Chd7\n                      <jats:sup>Gt\/+<\/jats:sup>\n                    <\/jats:italic>\n                    mice. These studies indicate that CHD7 has an important, dosage\u2010dependent role in development of several different craniofacial tissues.\n                    <jats:italic>Developmental Dynamics 243:1055\u20131066, 2014<\/jats:italic>\n                    . \u00a9 2014 The Authors Developmental Dynamics published by Wiley Periodicals, Inc. on behalf of American Association of Anatomists.\n                  <\/jats:p>","DOI":"10.1002\/dvdy.24156","type":"journal-article","created":{"date-parts":[[2014,6,26]],"date-time":"2014-06-26T13:19:11Z","timestamp":1403788751000},"page":"1055-1066","source":"Crossref","is-referenced-by-count":44,"title":["The chromatin remodeling protein CHD7, mutated in CHARGE syndrome, is necessary for proper craniofacial and tracheal development"],"prefix":"10.1002","volume":"243","author":[{"given":"Ethan D.","family":"Sperry","sequence":"first","affiliation":[{"name":"Department of Human Genetics The University of Michigan Ann Arbor Michigan"},{"name":"The 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