{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,4]],"date-time":"2025-11-04T10:44:07Z","timestamp":1762253047781,"version":"3.41.2"},"reference-count":81,"publisher":"The Company of Biologists","issue":"7","license":[{"start":{"date-parts":[[2020,9,3]],"date-time":"2020-09-03T00:00:00Z","timestamp":1599091200000},"content-version":"vor","delay-in-days":430,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["R15DE025960, F32DE027277"],"award-info":[{"award-number":["R15DE025960, F32DE027277"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"publisher","award":["GM127607"],"award-info":[{"award-number":["GM127607"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2019,7,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Cleft palate is a common birth defect, occurring in approximately 1 in 1000 live births worldwide. Known etiological mechanisms of cleft palate include defects within developing palate shelf tissues, defects in mandibular growth and defects in spontaneous fetal mouth movement. Until now, experimental studies directly documenting fetal mouth immobility as an underlying cause of cleft palate have been limited to models lacking neurotransmission. This study extends the range of anomalies directly demonstrated to have fetal mouth movement defects correlated with cleft palate. Here, we show that mouse embryos deficient in retinoic acid (RA) have mispatterned pharyngeal nerves and skeletal elements that block spontaneous fetal mouth movement in utero. Using X-ray microtomography, in utero ultrasound video, ex vivo culture and tissue staining, we demonstrate that proper retinoid signaling and pharyngeal patterning are crucial for the fetal mouth movement needed for palate formation. Embryos with deficient retinoid signaling were generated by stage-specific inactivation of retinol dehydrogenase 10 (Rdh10), a gene crucial for the production of RA during embryogenesis. The finding that cleft palate in retinoid deficiency results from a lack of fetal mouth movement might help elucidate cleft palate etiology and improve early diagnosis in human disorders involving defects of pharyngeal development.<\/jats:p>","DOI":"10.1242\/dmm.039073","type":"journal-article","created":{"date-parts":[[2019,7,4]],"date-time":"2019-07-04T13:28:18Z","timestamp":1562246898000},"source":"Crossref","is-referenced-by-count":12,"title":["RDH10 function is necessary for spontaneous fetal mouth movement that facilitates palate shelf elevation"],"prefix":"10.1242","volume":"12","author":[{"given":"Regina M.","family":"Friedl","sequence":"first","affiliation":[{"name":"University of Louisville School of Dentistry 1 Department of Oral Immunology and Infectious Diseases , , Louisville, KY 40202, \u2008 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Swetha","family":"Raja","sequence":"additional","affiliation":[{"name":"University of Louisville School of Dentistry 1 Department of Oral Immunology and Infectious Diseases , , Louisville, KY 40202, \u2008 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Melissa A.","family":"Metzler","sequence":"additional","affiliation":[{"name":"University of Louisville School of Dentistry 1 Department of Oral Immunology and Infectious Diseases , , Louisville, KY 40202, \u2008 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Niti D.","family":"Patel","sequence":"additional","affiliation":[{"name":"University of Louisville School of Dentistry 1 Department of Oral Immunology and Infectious Diseases , , Louisville, KY 40202, \u2008 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kenneth R.","family":"Brittian","sequence":"additional","affiliation":[{"name":"University of Louisville 2 Department of Medicine, Diabetes and Obesity Center , , Louisville, KY 40202 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Steven P.","family":"Jones","sequence":"additional","affiliation":[{"name":"University of Louisville 2 Department of Medicine, Diabetes and Obesity Center , , Louisville, KY 40202 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1735-8223","authenticated-orcid":false,"given":"Lisa L.","family":"Sandell","sequence":"additional","affiliation":[{"name":"University of Louisville School of Dentistry 1 Department of Oral Immunology and Infectious Diseases , , Louisville, KY 40202, \u2008 USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2019,7,3]]},"reference":[{"key":"2024061302260023900_DMM039073C1","doi-asserted-by":"publisher","first-page":"135","DOI":"10.1002\/dvdy.24476","article-title":"Retinoic acid signaling regulates Krt5 and Krt14 independently of stem cell markers in submandibular salivary gland epithelium","volume":"246","author":"Abashev","year":"2017","journal-title":"Dev. Dyn."},{"key":"2024061302260023900_DMM039073C2","doi-asserted-by":"publisher","first-page":"6496","DOI":"10.1073\/pnas.94.12.6496","article-title":"Cleft palate and decreased brain gamma-aminobutyric acid in mice lacking the 67-kDa isoform of glutamic acid decarboxylase","volume":"94","author":"Asada","year":"1997","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061302260023900_DMM039073C3","doi-asserted-by":"crossref","first-page":"5011","DOI":"10.1242\/dev.126.22.5011","article-title":"Compensatory defects associated with mutations in Hoxa1 restore normal palatogenesis to Hoxa2 mutants","volume":"126","author":"Barrow","year":"1999","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C4","doi-asserted-by":"crossref","first-page":"5103","DOI":"10.1242\/dev.129.22.5103","article-title":"Initiating Hox gene expression: in the early chick neural tube differential sensitivity to FGF and RA signaling subdivides the HoxB genes in two distinct groups","volume":"129","author":"Bel-Vialar","year":"2002","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C5","doi-asserted-by":"publisher","first-page":"149","DOI":"10.1002\/aja.1001200112","article-title":"Morphogenesis of the palate in normal human embryos with special emphasis on the mechanisms involved","volume":"120","author":"Burdi","year":"1967","journal-title":"Am. J. Anat."},{"key":"2024061302260023900_DMM039073C6","doi-asserted-by":"publisher","first-page":"231","DOI":"10.1242\/dev.067082","article-title":"Palatogenesis: morphogenetic and molecular mechanisms of secondary palate development","volume":"139","author":"Bush","year":"2012","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C7","doi-asserted-by":"publisher","first-page":"33","DOI":"10.1016\/j.ydbio.2016.05.010","article-title":"Temporal and spatial requirements for Hoxa3 in mouse embryonic development","volume":"415","author":"Chojnowski","year":"2016","journal-title":"Dev. Biol."},{"key":"2024061302260023900_DMM039073C8","doi-asserted-by":"publisher","first-page":"11451","DOI":"10.1073\/pnas.94.21.11451","article-title":"Cleft palate in mice with a targeted mutation in the gamma-aminobutyric acid-producing enzyme glutamic acid decarboxylase 67","volume":"94","author":"Condie","year":"1997","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061302260023900_DMM039073C9","doi-asserted-by":"publisher","first-page":"344","DOI":"10.1038\/ng1195-344","article-title":"Deficiency of the beta 3 subunit of the type A gamma-aminobutyric acid receptor causes cleft palate in mice","volume":"11","author":"Culiat","year":"1995","journal-title":"Nat. Genet."},{"key":"2024061302260023900_DMM039073C10","doi-asserted-by":"publisher","first-page":"110","DOI":"10.1038\/nrm3932","article-title":"Mechanisms of retinoic acid signalling and its roles in organ and limb development","volume":"16","author":"Cunningham","year":"2015","journal-title":"Nat. Rev. Mol. Cell Biol."},{"key":"2024061302260023900_DMM039073C11","doi-asserted-by":"publisher","first-page":"2931","DOI":"10.1242\/dev.01897","article-title":"Developmental regulation of the Hox genes during axial morphogenesis in the mouse","volume":"132","author":"Deschamps","year":"2005","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C12","doi-asserted-by":"publisher","first-page":"99","DOI":"10.1016\/0378-3782(85)90174-4","article-title":"The emergence of fetal behaviour. II. Quantitative aspects","volume":"12","author":"de Vries","year":"1985","journal-title":"Early Hum. Dev."},{"key":"2024061302260023900_DMM039073C13","doi-asserted-by":"publisher","first-page":"857","DOI":"10.1002\/bies.20080","article-title":"Opposing FGF and retinoid pathways: a signalling switch that controls differentiation and patterning onset in the extending vertebrate body axis","volume":"26","author":"Diez del Corral","year":"2004","journal-title":"BioEssays"},{"key":"2024061302260023900_DMM039073C14","doi-asserted-by":"publisher","first-page":"921","DOI":"10.1016\/j.cell.2008.09.002","article-title":"Retinoic acid synthesis and signaling during early organogenesis","volume":"134","author":"Duester","year":"2008","journal-title":"Cell"},{"key":"2024061302260023900_DMM039073C15","doi-asserted-by":"crossref","first-page":"1123","DOI":"10.1242\/dev.125.6.1123","article-title":"Hoxa1 and Hoxb1 synergize in patterning the hindbrain, cranial nerves and second pharyngeal arch","volume":"125","author":"Gavalas","year":"1998","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C16","doi-asserted-by":"publisher","first-page":"419","DOI":"10.1016\/j.jormas.2018.05.002","article-title":"Pierre Robin sequence: a comprehensive narrative review of the literature over time","volume":"119","author":"Giudice","year":"2018","journal-title":"J. Stomatol. Oral Maxillofac. Surg."},{"key":"2024061302260023900_DMM039073C17","doi-asserted-by":"publisher","first-page":"1087","DOI":"10.1016\/S0002-9394(35)90563-3","article-title":"The relation of vitamin A to anophthalmos in pigs","volume":"18","author":"Hale","year":"1935","journal-title":"Am. J. Ophthamol."},{"key":"2024061302260023900_DMM039073C18","doi-asserted-by":"publisher","first-page":"2039","DOI":"10.1242\/dev.00423","article-title":"A novel role for retinoids in patterning the avian forebrain during presomite stages","volume":"130","author":"Halilagic","year":"2003","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C19","doi-asserted-by":"publisher","first-page":"362","DOI":"10.1016\/j.ydbio.2006.11.021","article-title":"Retinoids control anterior and dorsal properties in the developing forebrain","volume":"303","author":"Halilagic","year":"2007","journal-title":"Dev. Biol."},{"key":"2024061302260023900_DMM039073C20","doi-asserted-by":"publisher","first-page":"177","DOI":"10.1242\/dev.02706","article-title":"Cyp26 enzymes generate the retinoic acid response pattern necessary for hindbrain development","volume":"134","author":"Hernandez","year":"2007","journal-title":"Development"},{"volume-title":"Beobachtungen zur geschichte der nasen und gaumenbildung beim menschlichen embryo","year":"1901","author":"His","key":"2024061302260023900_DMM039073C21"},{"key":"2024061302260023900_DMM039073C22","doi-asserted-by":"publisher","first-page":"4143","DOI":"10.1073\/pnas.94.8.4143","article-title":"Mice devoid of gamma-aminobutyrate type A receptor beta3 subunit have epilepsy, cleft palate, and hypersensitive behavior","volume":"94","author":"Homanics","year":"1997","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061302260023900_DMM039073C23","doi-asserted-by":"publisher","first-page":"e898","DOI":"10.1038\/cddis.2013.424","article-title":"Retinoic acid alters the proliferation and survival of the epithelium and mesenchyme and suppresses Wnt\/beta-catenin signaling in developing cleft palate","volume":"4","author":"Hu","year":"2013","journal-title":"Cell Death Dis."},{"key":"2024061302260023900_DMM039073C24","first-page":"185","article-title":"[Study on etiology of retinoic acid-induced cleft palate in mouse]","volume":"38","author":"Huang","year":"2003","journal-title":"Zhonghua Kou Qiang Yi Xue Za Zhi"},{"key":"2024061302260023900_DMM039073C25","doi-asserted-by":"publisher","first-page":"245","DOI":"10.1242\/dmm.012484","article-title":"Dysphagia and disrupted cranial nerve development in a mouse model of DiGeorge (22q11) deletion syndrome","volume":"7","author":"Karpinski","year":"2014","journal-title":"Dis. Model Mech."},{"key":"2024061302260023900_DMM039073C26","doi-asserted-by":"publisher","first-page":"1268","DOI":"10.1093\/hmg\/ddx031","article-title":"Rdh10 loss-of-function and perturbed retinoid signaling underlies the etiology of choanal atresia","volume":"26","author":"Kurosaka","year":"2017","journal-title":"Hum. Mol. Genet."},{"key":"2024061302260023900_DMM039073C27","doi-asserted-by":"publisher","first-page":"329","DOI":"10.1016\/j.ydbio.2015.09.024","article-title":"Hard to swallow: developmental biological insights into pediatric dysphagia","volume":"409","author":"LaMantia","year":"2016","journal-title":"Dev. Biol."},{"key":"2024061302260023900_DMM039073C28","doi-asserted-by":"publisher","first-page":"2344","DOI":"10.1242\/dev.134577","article-title":"Golgb1 regulates protein glycosylation and is crucial for mammalian palate development","volume":"143","author":"Lan","year":"2016","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C29","doi-asserted-by":"publisher","first-page":"75","DOI":"10.1054\/ijom.2000.0009","article-title":"Complete bony fusion of the mandible to the zygomatic complex and maxillary tuberosity: case report and review","volume":"30","author":"Laster","year":"2001","journal-title":"Int. J. Oral Maxillofac. Surg."},{"key":"2024061302260023900_DMM039073C30","doi-asserted-by":"publisher","first-page":"402","DOI":"10.1006\/meth.2001.1262","article-title":"Analysis of relative gene expression data using real-time quantitative PCR and the 2(-Delta Delta C(T)) Method","volume":"25","author":"Livak","year":"2001","journal-title":"Methods"},{"key":"2024061302260023900_DMM039073C31","doi-asserted-by":"crossref","first-page":"2723","DOI":"10.1242\/dev.120.10.2723","article-title":"Function of the retinoic acid receptors (RARs) during development (I). Craniofacial and skeletal abnormalities in RAR double mutants","volume":"120","author":"Lohnes","year":"1994","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C32","doi-asserted-by":"publisher","first-page":"33","DOI":"10.1016\/0925-4773(95)00488-2","article-title":"Compound mutants for retinoic acid receptor (RAR) beta and RAR alpha 1 reveal developmental functions for multiple RAR beta isoforms","volume":"55","author":"Luo","year":"1996","journal-title":"Mech. Dev."},{"key":"2024061302260023900_DMM039073C33","doi-asserted-by":"publisher","first-page":"591","DOI":"10.1016\/j.gde.2004.07.012","article-title":"Retinoic acid signalling in the development of branchial arches","volume":"14","author":"Mark","year":"2004","journal-title":"Curr. Opin. Genet. Dev."},{"key":"2024061302260023900_DMM039073C34","doi-asserted-by":"publisher","first-page":"15071","DOI":"10.1038\/nrdp.2015.71","article-title":"22q11.2 deletion syndrome","volume":"1","author":"McDonald-McGinn","year":"2015","journal-title":"Nat. Rev. Dis. Primers"},{"key":"2024061302260023900_DMM039073C35","doi-asserted-by":"publisher","first-page":"619","DOI":"10.1016\/S0092-8674(01)00247-1","article-title":"TBX1 is responsible for cardiovascular defects in velo-cardio-facial\/DiGeorge syndrome","volume":"104","author":"Merscher","year":"2001","journal-title":"Cell"},{"key":"2024061302260023900_DMM039073C36","doi-asserted-by":"publisher","first-page":"812","DOI":"10.3390\/nu8120812","article-title":"Enzymatic metabolism of vitamin a in developing vertebrate embryos","volume":"8","author":"Metzler","year":"2016","journal-title":"Nutrients"},{"key":"2024061302260023900_DMM039073C37","doi-asserted-by":"publisher","first-page":"dev164822","DOI":"10.1242\/dev.164822","article-title":"RDH10-mediated retinol metabolism and RARalpha-mediated retinoic acid signaling are required for submandibular salivary gland initiation","volume":"145","author":"Metzler","year":"2018","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C38","doi-asserted-by":"publisher","first-page":"194","DOI":"10.1097\/MOO.0b013e328325a538","article-title":"Updates on pediatric feeding and swallowing problems","volume":"17","author":"Miller","year":"2009","journal-title":"Curr. Opin Otolaryngol. Head Neck Surg."},{"key":"2024061302260023900_DMM039073C39","doi-asserted-by":"publisher","first-page":"263","DOI":"10.1002\/(SICI)1096-9926(199812)58:6&lt;263::AID-TERA8&gt;3.0.CO;2-A","article-title":"Retinoic acid-induced thymic abnormalities in the mouse are associated with altered pharyngeal morphology, thymocyte maturation defects, and altered expression of Hoxa3 and Pax1","volume":"58","author":"Mulder","year":"1998","journal-title":"Teratology"},{"key":"2024061302260023900_DMM039073C40","doi-asserted-by":"publisher","first-page":"1572","DOI":"10.1097\/SCS.0b013e31822e5ea6","article-title":"Commentary on role of apoptosis in retinoic Acid-induced cleft palate","volume":"22","author":"Nelson","year":"2011","journal-title":"J. Craniofac. Surg."},{"key":"2024061302260023900_DMM039073C41","doi-asserted-by":"publisher","first-page":"541","DOI":"10.1038\/nrg2340","article-title":"Retinoic acid in development: towards an integrated view","volume":"9","author":"Niederreither","year":"2008","journal-title":"Nat. Rev. Genet."},{"key":"2024061302260023900_DMM039073C42","doi-asserted-by":"publisher","first-page":"444","DOI":"10.1038\/7788","article-title":"Embryonic retinoic acid synthesis is essential for early mouse post-implantation development","volume":"21","author":"Niederreither","year":"1999","journal-title":"Nat. Genet."},{"key":"2024061302260023900_DMM039073C43","doi-asserted-by":"publisher","first-page":"2525","DOI":"10.1242\/dev.00463","article-title":"The regional pattern of retinoic acid synthesis by RALDH2 is essential for the development of posterior pharyngeal arches and the enteric nervous system","volume":"130","author":"Niederreither","year":"2003","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C44","doi-asserted-by":"publisher","first-page":"e9758","DOI":"10.1371\/journal.pone.0009758","article-title":"Cleft palate is caused by CNS dysfunction in Gad1 and Viaat knockout mice","volume":"5","author":"Oh","year":"2010","journal-title":"PLoS ONE"},{"key":"2024061302260023900_DMM039073C45","doi-asserted-by":"publisher","first-page":"42","DOI":"10.1016\/j.taap.2007.02.019","article-title":"Involvement of apoptotic cell death and cell cycle perturbation in retinoic acid-induced cleft palate in mice","volume":"221","author":"Okano","year":"2007","journal-title":"Toxicol. Appl. Pharmacol."},{"key":"2024061302260023900_DMM039073C46","doi-asserted-by":"publisher","first-page":"1744","DOI":"10.1002\/dvdy.23862","article-title":"The regulation of endogenous retinoic acid level through CYP26B1 is required for elevation of palatal shelves","volume":"241","author":"Okano","year":"2012","journal-title":"Dev. Dyn."},{"key":"2024061302260023900_DMM039073C47","doi-asserted-by":"publisher","first-page":"1008","DOI":"10.1002\/bdra.20735","article-title":"Updated national birth prevalence estimates for selected birth defects in the United States, 2004\u20132006","volume":"88","author":"Parker","year":"2010","journal-title":"Birth Defects Res. A Clin. Mol. Teratol."},{"key":"2024061302260023900_DMM039073C48","doi-asserted-by":"publisher","first-page":"532","DOI":"10.1597\/05-085","article-title":"The hyoid bone in malay infants with cleft lip and palate","volume":"43","author":"Rajion","year":"2006","journal-title":"Cleft Palate Craniofac. J."},{"key":"2024061302260023900_DMM039073C49","doi-asserted-by":"publisher","first-page":"843","DOI":"10.1242\/dev.065938","article-title":"Retinoic acid signalling during development","volume":"139","author":"Rhinn","year":"2012","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C50","doi-asserted-by":"publisher","first-page":"351","DOI":"10.1242\/dev.02204","article-title":"Retinaldehyde dehydrogenase 2 (RALDH2)-mediated retinoic acid synthesis regulates early mouse embryonic forebrain development by controlling FGF and sonic hedgehog signaling","volume":"133","author":"Ribes","year":"2006","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C51","doi-asserted-by":"publisher","first-page":"928","DOI":"10.1002\/dvdy.20268","article-title":"Retinoic acid down-regulates Tbx1 expression in vivo and in vitro","volume":"232","author":"Roberts","year":"2005","journal-title":"Dev. Dyn."},{"key":"2024061302260023900_DMM039073C52","doi-asserted-by":"publisher","first-page":"1333","DOI":"10.1101\/gad.5.8.1333","article-title":"Expression of a retinoic acid response element-hsplacZ transgene defines specific domains of transcriptional activity during mouse embryogenesis","volume":"5","author":"Rossant","year":"1991","journal-title":"Genes Dev."},{"key":"2024061302260023900_DMM039073C53","doi-asserted-by":"publisher","first-page":"686","DOI":"10.1161\/CIRCRESAHA.109.205732","article-title":"Decreased levels of embryonic retinoic acid synthesis accelerate recovery from arterial growth delay in a mouse model of DiGeorge syndrome","volume":"106","author":"Ryckebusch","year":"2010","journal-title":"Circ. Res."},{"key":"2024061302260023900_DMM039073C54","doi-asserted-by":"publisher","first-page":"1113","DOI":"10.1101\/gad.1533407","article-title":"RDH10 is essential for synthesis of embryonic retinoic acid and is required for limb, craniofacial, and organ development","volume":"21","author":"Sandell","year":"2007","journal-title":"Genes Dev."},{"key":"2024061302260023900_DMM039073C55","doi-asserted-by":"publisher","first-page":"844","DOI":"10.1002\/dvg.22344","article-title":"Whole mount nuclear fluorescent imaging: convenient documentation of embryo morphology","volume":"50","author":"Sandell","year":"2012","journal-title":"Genesis"},{"key":"2024061302260023900_DMM039073C56","doi-asserted-by":"publisher","first-page":"e30698","DOI":"10.1371\/journal.pone.0030698","article-title":"RDH10 oxidation of Vitamin A is a critical control step in synthesis of retinoic acid during mouse embryogenesis","volume":"7","author":"Sandell","year":"2012","journal-title":"PLoS ONE"},{"key":"2024061302260023900_DMM039073C57","doi-asserted-by":"publisher","first-page":"378","DOI":"10.1007\/s00246-009-9613-0","article-title":"22q11 deletion syndrome: a role for TBX1 in pharyngeal and cardiovascular development","volume":"31","author":"Scambler","year":"2010","journal-title":"Pediatr. Cardiol."},{"key":"2024061302260023900_DMM039073C58","doi-asserted-by":"crossref","first-page":"2755","DOI":"10.1242\/dev.128.14.2755","article-title":"Local retinoid signaling coordinates forebrain and facial morphogenesis by maintaining FGF8 and SHH","volume":"128","author":"Schneider","year":"2001","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C59","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1016\/j.ydbio.2007.10.018","article-title":"A nutritional model of late embryonic vitamin A deficiency produces defects in organogenesis at a high penetrance and reveals new roles for the vitamin in skeletal development","volume":"316","author":"See","year":"2008","journal-title":"Dev. Biol."},{"key":"2024061302260023900_DMM039073C60","doi-asserted-by":"publisher","first-page":"2611","DOI":"10.1242\/dev.01845","article-title":"Shifting boundaries of retinoic acid activity control hindbrain segmental gene expression","volume":"132","author":"Sirbu","year":"2005","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C61","doi-asserted-by":"publisher","first-page":"10440","DOI":"10.1074\/jbc.M112.432286","article-title":"Mice with Tak1 deficiency in neural crest lineage exhibit cleft palate associated with abnormal tongue development","volume":"288","author":"Song","year":"2013","journal-title":"J. Biol. Chem."},{"key":"2024061302260023900_DMM039073C62","doi-asserted-by":"crossref","first-page":"14","DOI":"10.1093\/oso\/9780195139068.003.0002","article-title":"Palatogenesis: closure of the secondary palate","volume-title":"Cleft Lip and Palate: From Origin to Treatment","author":"Sperber","year":"2002"},{"key":"2024061302260023900_DMM039073C63","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1111\/j.1460-9568.2004.03860.x","article-title":"Development of spontaneous mouth\/tongue movement and related neural activity, and their repression in fetal mice lacking glutamate decarboxylase 67","volume":"21","author":"Tsunekawa","year":"2005","journal-title":"Eur. J. Neurosci."},{"key":"2024061302260023900_DMM039073C64","doi-asserted-by":"publisher","first-page":"661","DOI":"10.1038\/nature05541","article-title":"Restoration of p53 function leads to tumour regression in vivo","volume":"445","author":"Ventura","year":"2007","journal-title":"Nature"},{"key":"2024061302260023900_DMM039073C65","doi-asserted-by":"publisher","first-page":"1763","DOI":"10.1073\/pnas.0437920100","article-title":"Decreased embryonic retinoic acid synthesis results in a DiGeorge syndrome phenotype in newborn mice","volume":"100","author":"Vermot","year":"2003","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061302260023900_DMM039073C66","first-page":"745","article-title":"Comparison of hyoid bone position among cleft lip palate and normal subjects","volume":"24","author":"Wahaj","year":"2014","journal-title":"J. Coll. Physicians Surg. Pak."},{"key":"2024061302260023900_DMM039073C67","doi-asserted-by":"publisher","first-page":"191","DOI":"10.1002\/tera.1420020304","article-title":"Correlation of embryonic movement with palate closure in mice","volume":"2","author":"Walker","year":"1969","journal-title":"Teratology"},{"key":"2024061302260023900_DMM039073C68","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1016\/j.neuroscience.2017.06.057","article-title":"Altered neurobiological function of brainstem hypoglossal neurons in DiGeorge\/22q11.2 Deletion Syndrome","volume":"359","author":"Wang","year":"2017","journal-title":"Neuroscience"},{"key":"2024061302260023900_DMM039073C69","doi-asserted-by":"publisher","first-page":"66","DOI":"10.2307\/3348002","article-title":"The importance of prenatal diet","volume":"23","author":"Warkany","year":"1945","journal-title":"The Milbank Memorial Fund Quarterly"},{"key":"2024061302260023900_DMM039073C70","doi-asserted-by":"publisher","first-page":"882","DOI":"10.1001\/archpedi.1943.02010180058007","article-title":"Congenital malformations induced in rats by maternal nutritional deficiency: Iv. cleft palate","volume":"65","author":"Warkany","year":"1943","journal-title":"Am. J. Dis. Child."},{"key":"2024061302260023900_DMM039073C71","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1111\/j.1601-0825.2009.01588.x","article-title":"The impact of orofacial clefts on quality of life and healthcare use and costs","volume":"16","author":"Wehby","year":"2010","journal-title":"Oral Dis."},{"key":"2024061302260023900_DMM039073C72","doi-asserted-by":"crossref","first-page":"1553","DOI":"10.1242\/dev.127.8.1553","article-title":"Retinoid signaling is essential for patterning the endoderm of the third and fourth pharyngeal arches","volume":"127","author":"Wendling","year":"2000","journal-title":"Development"},{"key":"2024061302260023900_DMM039073C73","doi-asserted-by":"publisher","first-page":"13459","DOI":"10.1073\/pnas.95.23.13459","article-title":"Defects in embryonic hindbrain development and fetal resorption resulting from vitamin A deficiency in the rat are prevented by feeding pharmacological levels of all-trans-retinoic acid","volume":"95","author":"White","year":"1998","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2024061302260023900_DMM039073C74","doi-asserted-by":"publisher","first-page":"263","DOI":"10.1006\/dbio.2000.9635","article-title":"Vitamin A deficiency results in the dose-dependent acquisition of anterior character and shortening of the caudal hindbrain of the rat embryo","volume":"220","author":"White","year":"2000","journal-title":"Dev. Biol."},{"key":"2024061302260023900_DMM039073C75","doi-asserted-by":"publisher","first-page":"189","DOI":"10.1002\/aja.1000920202","article-title":"An analysis of the syndrome of malformations induced by maternal vitamin a deficiency. Effects of restoration of vitamin a at various times during gestation","volume":"92","author":"Wilson","year":"1953","journal-title":"Am. J. Anat."},{"key":"2024061302260023900_DMM039073C76","doi-asserted-by":"publisher","first-page":"575","DOI":"10.1016\/j.neuron.2006.04.016","article-title":"A shared vesicular carrier allows synaptic corelease of GABA and glycine","volume":"50","author":"Wojcik","year":"2006","journal-title":"Neuron"},{"year":"2007","author":"World Health Organization","key":"2024061302260023900_DMM039073C77"},{"key":"2024061302260023900_DMM039073C78","doi-asserted-by":"publisher","first-page":"673","DOI":"10.1016\/0003-9969(72)90193-8","article-title":"Myoneural maturation and function of the foetal rat tongue at the time of secondary palate closure","volume":"17","author":"Wragg","year":"1972","journal-title":"Arch. Oral Biol."},{"key":"2024061302260023900_DMM039073C79","doi-asserted-by":"publisher","first-page":"477","DOI":"10.3349\/ymj.2000.41.4.477","article-title":"Development of the lip and palate in staged human embryos and early fetuses","volume":"41","author":"Yoon","year":"2000","journal-title":"Yonsei Med. J."},{"key":"2024061302260023900_DMM039073C80","doi-asserted-by":"publisher","first-page":"154","DOI":"10.3389\/fphys.2017.00154","article-title":"Differences in oral structure and tissue interactions during mouse vs. human palatogenesis: implications for the translation of findings from mice","volume":"8","author":"Yu","year":"2017","journal-title":"Front. Physiol."},{"key":"2024061302260023900_DMM039073C81","doi-asserted-by":"publisher","first-page":"630","DOI":"10.1161\/CIRCRESAHA.109.215319","article-title":"DiGeorge syndrome, Tbx1, and retinoic acid signaling come full circle","volume":"106","author":"Yutzey","year":"2010","journal-title":"Circ. Res."}],"container-title":["Disease Models &amp; Mechanisms"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1242\/dmm.039073","content-type":"unspecified","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.039073\/3483980\/dmm039073.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.039073\/3483980\/dmm039073.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,13]],"date-time":"2024-06-13T02:26:53Z","timestamp":1718245613000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dmm\/article\/12\/7\/dmm039073\/3366\/RDH10-function-is-necessary-for-spontaneous-fetal"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,7,1]]},"references-count":81,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2019,7,1]]}},"URL":"https:\/\/doi.org\/10.1242\/dmm.039073","relation":{},"ISSN":["1754-8411","1754-8403"],"issn-type":[{"type":"electronic","value":"1754-8411"},{"type":"print","value":"1754-8403"}],"subject":[],"published":{"date-parts":[[2019,7,1]]},"article-number":"dmm039073"}}