{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,5]],"date-time":"2026-07-05T05:58:29Z","timestamp":1783231109648,"version":"3.54.6"},"reference-count":49,"publisher":"The Company of Biologists","issue":"11","license":[{"start":{"date-parts":[[2023,11,28]],"date-time":"2023-11-28T00:00:00Z","timestamp":1701129600000},"content-version":"vor","delay-in-days":27,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["HD093758"],"award-info":[{"award-number":["HD093758"]}],"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":["HD067244"],"award-info":[{"award-number":["HD067244"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Baylor College of Medicine"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,11,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Sonic hedgehog (Shh) signaling is the morphogen signaling that regulates embryonic craniofacial and neural tube development. G\u00a0protein-coupled receptor 161 (Gpr161) is a negative regulator of Shh signaling, and its inactivation in mice results in embryo lethality associated with craniofacial defects and neural tube defects. However, the structural defects of later embryonic stages and cell lineages underlying abnormalities have not been well characterized due to the limited lifespan of Gpr161 null mice. We found that embryos with Pax3 lineage-specific deletion of Gpr161 presented with tectal hypertrophy (anterior dorsal neuroepithelium), cranial vault and facial bone hypoplasia (cranial neural crest), vertebral abnormalities (somite) and the closed form of spina bifida (posterior dorsal neuroepithelium). In particular, the closed form of spina bifida was partly due to reduced Pax3 and Cdx4 gene expression in the posterior dorsal neural tubes of Gpr161 mutant embryos with decreased Wnt signaling, whereas Shh signaling was increased. We describe a previously unreported role for Gpr161 in the development of posterior neural tubes and confirm its role in cranial neural crest- and somite-derived skeletogenesis and midbrain morphogenesis in mice.<\/jats:p>","DOI":"10.1242\/dmm.050277","type":"journal-article","created":{"date-parts":[[2023,10,27]],"date-time":"2023-10-27T04:03:19Z","timestamp":1698379399000},"source":"Crossref","is-referenced-by-count":5,"title":["<i>Pax3<\/i>\n                    lineage-specific deletion of\n                    <i>Gpr161<\/i>\n                    is associated with spinal neural tube and craniofacial malformations during embryonic development"],"prefix":"10.1242","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8667-3887","authenticated-orcid":false,"given":"Sung-Eun","family":"Kim","sequence":"first","affiliation":[{"name":"Dell Pediatric Research Institute, Dell Medical School, University of Texas at Austin 1 Department of Pediatrics , , Austin, TX 78723 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pooja J.","family":"Chothani","sequence":"additional","affiliation":[{"name":"Dell Pediatric Research Institute, Dell Medical School, University of Texas at Austin 1 Department of Pediatrics , , Austin, TX 78723 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rehana","family":"Shaik","sequence":"additional","affiliation":[{"name":"Dell Pediatric Research Institute, Dell Medical School, University of Texas at Austin 1 Department of Pediatrics , , Austin, TX 78723 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Westley","family":"Pollard","sequence":"additional","affiliation":[{"name":"Dell Pediatric Research Institute, Dell Medical School, University of Texas at Austin 1 Department of Pediatrics , , Austin, TX 78723 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard H.","family":"Finnell","sequence":"additional","affiliation":[{"name":"Dell Pediatric Research Institute, Dell Medical School, University of Texas at Austin 1 Department of Pediatrics , , Austin, TX 78723 , USA"},{"name":"Center for Precision Environmental Health 2 , Department of Molecular and Cellular Biology , , Houston, TX 77030 , USA"},{"name":"Baylor College of Medicine 2 , Department of Molecular and Cellular Biology , , Houston, TX 77030 , USA"},{"name":"Baylor College of Medicine 3 Departments of Molecular and Human Genetics and Medicine , , Houston, TX 77030 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2023,11,28]]},"reference":[{"key":"2023112808441450100_DMM050277C1","doi-asserted-by":"publisher","first-page":"1304","DOI":"10.1016\/S0960-9822(00)80052-4","article-title":"Inhibition of sonic hedgehog signaling in vivo results in craniofacial neural crest cell death","volume":"9","author":"Ahlgren","year":"1999","journal-title":"Curr. 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