{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T06:09:56Z","timestamp":1778047796929,"version":"3.51.4"},"reference-count":56,"publisher":"The Company of Biologists","issue":"21","license":[{"start":{"date-parts":[[2025,10,30]],"date-time":"2025-10-30T00:00:00Z","timestamp":1761782400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01DE032332"],"award-info":[{"award-number":["R01DE032332"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01DE027983"],"award-info":[{"award-number":["R01DE027983"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,11,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>The pituitary gland produces several hormones that regulate growth, metabolism, stress response, reproduction and homeostasis. Congenital hypopituitarism is a deficiency in one or more pituitary hormones and encompasses a spectrum of clinical conditions. The pituitary has a complex embryonic origin, with the oral ectoderm contributing the anterior lobe, and the neural ectoderm generating the posterior lobe. Pituitary abnormalities and growth deficiencies are associated with cleft palate; however, the developmental genetic connection between pituitary and orofacial cleft malformations remains to be determined. The epithelial RNA splicing regulators Esrp1 and Esrp2 (Esrp1\/2) are required for orofacial development in zebrafish, mice and humans, and loss of function of these genes results in a cleft palate. Here, we present a detailed developmental analysis of the genetic requirement for Esrp1\/2 in pituitary morphogenesis in mouse and zebrafish. Further, we describe an individual with cleft palate and hypopituitarism who harbors a nucleotide variant in the RNA-binding domain of ESRP2. The discovery of this key function for Esrp1\/2 in pituitary formation has significant fundamental and clinical implications for understanding congenital hypopituitarism and craniofacial anomalies.<\/jats:p>","DOI":"10.1242\/dev.204636","type":"journal-article","created":{"date-parts":[[2025,10,20]],"date-time":"2025-10-20T09:15:00Z","timestamp":1760951700000},"source":"Crossref","is-referenced-by-count":1,"title":["Genetic requirement for\n                    <i>Esrp1<\/i>\n                    and\n                    <i>Esrp2<\/i>\n                    in vertebrate pituitary morphogenesis"],"prefix":"10.1242","volume":"152","author":[{"given":"Shannon H.","family":"Carroll","sequence":"first","affiliation":[{"name":"Center for Craniofacial Innovation 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"Children's Hospital of Philadelphia 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"Shriners Hospital for Children 2 , Tampa, FL 33607 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sogand","family":"Schafer","sequence":"additional","affiliation":[{"name":"Center for Craniofacial Innovation 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"Children's Hospital of Philadelphia 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ariella S.","family":"Richman","sequence":"additional","affiliation":[{"name":"Center for Craniofacial Innovation 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"Children's Hospital of Philadelphia 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peng","family":"Wang","sequence":"additional","affiliation":[{"name":"Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia 3 , Philadelphia, PA 19104 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mian Umair","family":"Ahsan","sequence":"additional","affiliation":[{"name":"Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia 3 , Philadelphia, PA 19104 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lisa","family":"Tsay","sequence":"additional","affiliation":[{"name":"Center for Craniofacial Innovation 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"Children's Hospital of Philadelphia 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kai","family":"Wang","sequence":"additional","affiliation":[{"name":"Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia 3 , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"University of Pennsylvania 4 Department of Pathology and Laboratory Medicine , , Philadelphia, PA 19104 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6385-7448","authenticated-orcid":false,"given":"Eric C.","family":"Liao","sequence":"additional","affiliation":[{"name":"Center for Craniofacial Innovation 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"Children's Hospital of Philadelphia 1 , Division of Plastic and Reconstructive Surgery, Department of Surgery , , Philadelphia, PA 19104 ,","place":["USA"]},{"name":"Shriners Hospital for Children 2 , Tampa, FL 33607 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2025,10,30]]},"reference":[{"key":"2025103006580294700_DEV204636C1","doi-asserted-by":"publisher","first-page":"696","DOI":"10.3906\/sag-1303-89","article-title":"Endocrine abnormalities of patients with cleft lip and\/or cleft palate during the neonatal period","volume":"44","author":"Akin","year":"2014","journal-title":"Turk J. 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