{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,28]],"date-time":"2026-05-28T04:28:24Z","timestamp":1779942504838,"version":"3.53.1"},"reference-count":33,"publisher":"Wiley","issue":"10","license":[{"start":{"date-parts":[[2025,3,6]],"date-time":"2025-03-06T00:00:00Z","timestamp":1741219200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["F32\u2010DE030699"],"award-info":[{"award-number":["F32\u2010DE030699"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["K99\u2010DE034476"],"award-info":[{"award-number":["K99\u2010DE034476"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["R01\u2010DE025468"],"award-info":[{"award-number":["R01\u2010DE025468"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Dynamics"],"published-print":{"date-parts":[[2025,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>In vitro modeling is a powerful approach to investigate the pathomechanisms driving human congenital conditions. Here, we use human embryonic stem cells (hESCs) to model Nager and Rodriguez syndromes, two craniofacial conditions characterized by hypoplastic neural crest\u2010derived craniofacial bones, caused by pathogenic variants of SF3B4, a core component of the spliceosome.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We observed that siRNA\u2010mediated knockdown of\n                      <jats:italic>SF3B4<\/jats:italic>\n                      interferes with the production of hESC\u2010derived neural crest cells, as seen by a marked reduction in neural crest gene expression. This phenotype is associated with an increase in neural crest cell apoptosis and premature neuronal differentiation.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Altogether, these results point to a role of SF3B4 in neural crest cell survival, maintenance, and differentiation. We propose that the dysregulation of these processes may contribute to Nager\/Rodriguez syndrome\u2010associated craniofacial defects.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.70009","type":"journal-article","created":{"date-parts":[[2025,3,12]],"date-time":"2025-03-12T01:38:58Z","timestamp":1741743538000},"page":"1160-1169","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Human stem cell model of neural crest cell differentiation reveals a requirement of\n                    <scp>SF3B4<\/scp>\n                    in survival, maintenance, and differentiation"],"prefix":"10.1002","volume":"254","author":[{"given":"Casey","family":"Griffin","sequence":"first","affiliation":[{"name":"Department of Molecular Pathobiology, College of Dentistry New York University  New York 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