{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,26]],"date-time":"2026-09-26T16:22:27Z","timestamp":1790439747836,"version":"4.1.0"},"reference-count":66,"publisher":"The Company of Biologists","license":[{"start":{"date-parts":[[2021,5,18]],"date-time":"2021-05-18T00:00:00Z","timestamp":1621296000000},"content-version":"am","delay-in-days":503,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01DE027983"],"award-info":[{"award-number":["R01DE027983"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:p>Irf6 and Esrp1 are important for palate development across vertebrates. In zebrafish, we found that irf6 regulates the expression of esrp1. We detailed overlapping Irf6 and Esrp1\/2 expression in mouse orofacial epithelium. In zebrafish, irf6 and esrp1\/2 share expression in periderm, frontonasal ectoderm, and oral epithelium. Genetic disruption of irf6 and esrp1\/2 in zebrafish resulted in cleft of the anterior neurocranium. The esrp1\/2 mutant also developed cleft of the mouth opening. Lineage tracing of cranial neural crest cells revealed that cleft resulted not from migration defect, but from impaired chondrogenesis. Analysis of aberrant cells within the cleft revealed expression of sox10, col1a1 and irf6 and were adjacent to cells krt4 and krt5 positive. Breeding of mouse Irf6;Esrp1;Esrp2 compound mutants suggested genetic interaction, as the triple homozygote and the Irf6;Esrp1 double homozygote was not observed. Further, Irf6 heterozygosity reduced Esrp1\/2 cleft severity. These studies highlight the complementary analysis of Irf6 and Esrp1\/2 in mouse and zebrafish and captured a unique aberrant cell population expressing sox10, col1a1 and irf6. Future work characterizing this cell population will yield additional insight into cleft pathogenesis.<\/jats:p>","DOI":"10.1242\/dev.194498","type":"journal-article","created":{"date-parts":[[2020,11,24]],"date-time":"2020-11-24T21:16:24Z","timestamp":1606252584000},"source":"Crossref","is-referenced-by-count":22,"title":["An <i>Irf6<\/i>-<i>Esrp1\/2<\/i> regulatory axis controls midface morphogenesis in vertebrates"],"prefix":"10.1242","author":[{"given":"Shannon H.","family":"Carroll","sequence":"first","affiliation":[{"name":"Center for Regenerative Medicine, Massachusetts General Hospital, Boston, MA 02114, USA"},{"name":"Shriners Hospital for Children, Boston, MA 02114, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claudio","family":"Macias Trevino","sequence":"additional","affiliation":[{"name":"Center for Regenerative Medicine, Massachusetts General Hospital, Boston, MA 02114, USA"},{"name":"Harvard Medical School, Boston, MA 02115, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edward B.","family":"Li","sequence":"additional","affiliation":[{"name":"Harvard Medical School, Boston, MA 02115, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenta","family":"Kawasaki","sequence":"additional","affiliation":[{"name":"Center for Regenerative Medicine, Massachusetts General Hospital, Boston, MA 02114, USA"},{"name":"Shriners Hospital for Children, Boston, MA 02114, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nikita","family":"Myers","sequence":"additional","affiliation":[{"name":"Center for Regenerative Medicine, Massachusetts General Hospital, Boston, MA 02114, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shawn A.","family":"Hallett","sequence":"additional","affiliation":[{"name":"Center for Regenerative Medicine, Massachusetts General Hospital, Boston, MA 02114, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nora","family":"Alhazmi","sequence":"additional","affiliation":[{"name":"Harvard School of Dental Medicine, Boston, MA 02115, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justin","family":"Cotney","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut Health, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Russ P.","family":"Carstens","sequence":"additional","affiliation":[{"name":"Department of Medicine, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA 19104, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6385-7448","authenticated-orcid":false,"given":"Eric C.","family":"Liao","sequence":"additional","affiliation":[{"name":"Center for Regenerative Medicine, Massachusetts General Hospital, Boston, MA 02114, USA"},{"name":"Shriners Hospital for Children, Boston, MA 02114, USA"},{"name":"Harvard Medical School, Boston, MA 02115, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2020,1,1]]},"reference":[{"key":"2021042700462128800_DEV194498C1","doi-asserted-by":"publisher","first-page":"1457","DOI":"10.1002\/dvdy.24338","article-title":"Recent insights into the morphological diversity in the amniote primary and secondary palates","volume":"244","author":"Abramyan","year":"2015","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C2","doi-asserted-by":"publisher","first-page":"2800","DOI":"10.12688\/f1000research.9503.1","article-title":"Genetic factors influencing risk to orofacial clefts: today's challenges and tomorrow's opportunities","volume":"5","author":"Beaty","year":"2016","journal-title":"F1000Res"},{"key":"2021042700462128800_DEV194498C3","doi-asserted-by":"publisher","first-page":"e08954","DOI":"10.7554\/eLife.08954.029","article-title":"The splicing regulators Esrp1 and Esrp2 direct an epithelial splicing program essential for mammalian development","volume":"4","author":"Bebee","year":"2015","journal-title":"Elife"},{"key":"2021042700462128800_DEV194498C4","doi-asserted-by":"publisher","first-page":"991","DOI":"10.1002\/dvdy.24431","article-title":"Ablation of the epithelial-specific splicing factor Esrp1 results in ureteric branching defects and reduced nephron number","volume":"245","author":"Bebee","year":"2016","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C5","doi-asserted-by":"publisher","first-page":"13710","DOI":"10.1073\/pnas.1110931108","article-title":"Developmental factor IRF6 exhibits tumor suppressor activity in squamous cell carcinomas","volume":"108","author":"Botti","year":"2011","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2021042700462128800_DEV194498C6","doi-asserted-by":"publisher","first-page":"1799","DOI":"10.1038\/s41467-017-01961-y","article-title":"Evolutionary recruitment of flexible Esrp-dependent splicing programs into diverse embryonic morphogenetic processes","volume":"8","author":"Burguera","year":"2017","journal-title":"Nat. Commun."},{"key":"2021042700462128800_DEV194498C7","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":"2021042700462128800_DEV194498C8","doi-asserted-by":"publisher","first-page":"270","DOI":"10.1002\/ajmg.a.33702","article-title":"Cranial neural crest cells on the move: their roles in craniofacial development","volume":"155A","author":"Cordero","year":"2011","journal-title":"Am. J. Med. Genet. A"},{"key":"2021042700462128800_DEV194498C9","doi-asserted-by":"publisher","first-page":"1143","DOI":"10.1016\/j.ajhg.2018.04.009","article-title":"Mutations in the epithelial cadherin-p120-catenin complex cause mendelian non-syndromic cleft lip with or without cleft palate","volume":"102","author":"Cox","year":"2018","journal-title":"Am. J. Hum. Genet."},{"key":"2021042700462128800_DEV194498C10","doi-asserted-by":"publisher","first-page":"447","DOI":"10.1111\/j.1469-7580.2005.00485.x","article-title":"Patterning the neural crest derivatives during development of the vertebrate head: insights from avian studies","volume":"207","author":"Creuzet","year":"2005","journal-title":"J. Anat."},{"key":"2021042700462128800_DEV194498C11","doi-asserted-by":"publisher","first-page":"68","DOI":"10.1038\/jid.2012.269","article-title":"Interferon regulatory factor 6 promotes differentiation of the periderm by activating expression of Grainyhead-like 3","volume":"133","author":"de La Garza","year":"2013","journal-title":"J. Invest. Dermatol."},{"key":"2021042700462128800_DEV194498C12","doi-asserted-by":"publisher","first-page":"167","DOI":"10.1038\/nrg2933","article-title":"Cleft lip and palate: understanding genetic and environmental influences","volume":"12","author":"Dixon","year":"2011","journal-title":"Nat. Rev. Genet."},{"key":"2021042700462128800_DEV194498C13","doi-asserted-by":"publisher","first-page":"1333","DOI":"10.1097\/SCS.0b013e318260f20b","article-title":"Embryonic fate map of first pharyngeal arch structures in the sox10: kaede zebrafish transgenic model","volume":"23","author":"Dougherty","year":"2012","journal-title":"J. Craniofac. Surg."},{"key":"2021042700462128800_DEV194498C14","doi-asserted-by":"publisher","first-page":"76","DOI":"10.1242\/dev.080473","article-title":"Distinct requirements for wnt9a and irf6 in extension and integration mechanisms during zebrafish palate morphogenesis","volume":"140","author":"Dougherty","year":"2013","journal-title":"Development"},{"key":"2021042700462128800_DEV194498C15","doi-asserted-by":"publisher","first-page":"897","DOI":"10.1002\/dvdy.24566","article-title":"Zebrafish models of orofacial clefts","volume":"246","author":"Duncan","year":"2017","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C16","doi-asserted-by":"publisher","first-page":"7129","DOI":"10.1038\/s41598-017-06310-z","article-title":"Intercellular genetic interaction between Irf6 and Twist1 during craniofacial development","volume":"7","author":"Fakhouri","year":"2017","journal-title":"Sci. Rep."},{"key":"2021042700462128800_DEV194498C17","doi-asserted-by":"publisher","first-page":"627","DOI":"10.1016\/j.devcel.2011.08.005","article-title":"A conserved Pbx-Wnt-p63-Irf6 regulatory module controls face morphogenesis by promoting epithelial apoptosis","volume":"21","author":"Ferretti","year":"2011","journal-title":"Dev. Cell"},{"key":"2021042700462128800_DEV194498C18","doi-asserted-by":"publisher","first-page":"e98186","DOI":"10.1371\/journal.pone.0098186","article-title":"Efficient mutagenesis by Cas9 protein-mediated oligonucleotide insertion and large-scale assessment of single-guide RNAs","volume":"9","author":"Gagnon","year":"2014","journal-title":"PLoS ONE"},{"key":"2021042700462128800_DEV194498C19","doi-asserted-by":"publisher","first-page":"459","DOI":"10.1016\/bs.ctdb.2015.07.003","article-title":"From bench to bedside and back: improving diagnosis and treatment of craniofacial malformations utilizing animal models","volume":"115","author":"Goodwin","year":"2015","journal-title":"Curr. Top. Dev. Biol."},{"key":"2021042700462128800_DEV194498C20","doi-asserted-by":"publisher","first-page":"e56270","DOI":"10.1371\/journal.pone.0056270","article-title":"Cell-autonomous and non-cell-autonomous roles for IRF6 during development of the tongue","volume":"8","author":"Goudy","year":"2013","journal-title":"PLoS ONE"},{"key":"2021042700462128800_DEV194498C21","doi-asserted-by":"publisher","first-page":"37","DOI":"10.1016\/S0070-2153(08)00602-9","article-title":"The etiopathogenesis of cleft lip and cleft palate: usefulness and caveats of mouse models","volume":"84","author":"Gritli-Linde","year":"2008","journal-title":"Curr. Top. Dev. Biol."},{"key":"2021042700462128800_DEV194498C22","doi-asserted-by":"publisher","first-page":"851","DOI":"10.1242\/dev.01705","article-title":"New insights into craniofacial morphogenesis","volume":"132","author":"Helms","year":"2005","journal-title":"Development"},{"key":"2021042700462128800_DEV194498C23","doi-asserted-by":"publisher","first-page":"1749","DOI":"10.1242\/dev.00397","article-title":"A zone of frontonasal ectoderm regulates patterning and growth in the face","volume":"130","author":"Hu","year":"2003","journal-title":"Development"},{"key":"2021042700462128800_DEV194498C24","doi-asserted-by":"publisher","first-page":"1335","DOI":"10.1038\/ng1903","article-title":"Abnormal skin, limb and craniofacial morphogenesis in mice deficient for interferon regulatory factor 6 (Irf6)","volume":"38","author":"Ingraham","year":"2006","journal-title":"Nat. Genet."},{"key":"2021042700462128800_DEV194498C25","doi-asserted-by":"publisher","first-page":"1220","DOI":"10.1242\/dev.089615","article-title":"Smad4-Irf6 genetic interaction and TGFbeta-mediated IRF6 signaling cascade are crucial for palatal fusion in mice","volume":"140","author":"Iwata","year":"2013","journal-title":"Development"},{"key":"2021042700462128800_DEV194498C26","doi-asserted-by":"publisher","first-page":"1152","DOI":"10.1002\/dvdy.20646","article-title":"Development of the upper lip: morphogenetic and molecular mechanisms","volume":"235","author":"Jiang","year":"2006","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C27","doi-asserted-by":"publisher","first-page":"63","DOI":"10.1002\/bdra.20430","article-title":"Mouse genetic models of cleft lip with or without cleft palate","volume":"82","author":"Juriloff","year":"2008","journal-title":"Birth Defects Res. A Clin. Mol. Teratol"},{"key":"2021042700462128800_DEV194498C28","doi-asserted-by":"publisher","first-page":"D1284","DOI":"10.1093\/nar\/gkx1188","article-title":"JASPAR 2018: update of the open-access database of transcription factor binding profiles and its web framework","volume":"46","author":"Khan","year":"2018","journal-title":"Nucleic Acids Res."},{"key":"2021042700462128800_DEV194498C29","doi-asserted-by":"publisher","first-page":"283","DOI":"10.1016\/0168-9525(89)90103-0","article-title":"Genetics and early development of zebrafish","volume":"5","author":"Kimmel","year":"1989","journal-title":"Trends Genet."},{"key":"2021042700462128800_DEV194498C30","doi-asserted-by":"publisher","first-page":"120","DOI":"10.1007\/978-0-387-46954-6_7","article-title":"Cranial neural crest and development of the head skeleton","volume":"589","author":"Knight","year":"2006","journal-title":"Adv. Exp. Med. Biol."},{"key":"2021042700462128800_DEV194498C31","doi-asserted-by":"publisher","first-page":"1441","DOI":"10.1002\/dvdy.20598","article-title":"Developmental expression analysis of the mouse and chick orthologues of IRF6: the gene mutated in Van der Woude syndrome","volume":"235","author":"Knight","year":"2006","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C32","doi-asserted-by":"publisher","first-page":"285","DOI":"10.1038\/ng985","article-title":"Mutations in IRF6 cause Van der Woude and popliteal pterygium syndromes","volume":"32","author":"Kondo","year":"2002","journal-title":"Nat. Genet."},{"key":"2021042700462128800_DEV194498C33","doi-asserted-by":"publisher","first-page":"220","DOI":"10.1002\/dvdy.24341","article-title":"Toward an orofacial gene regulatory network","volume":"245","author":"Kousa","year":"2016","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C34","doi-asserted-by":"publisher","first-page":"670","DOI":"10.1002\/dvdy.24537","article-title":"IRF6 expression in basal epithelium partially rescues Irf6 knockout mice","volume":"246","author":"Kousa","year":"2017","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C35","doi-asserted-by":"publisher","first-page":"1726","DOI":"10.1093\/hmg\/ddz010","article-title":"The TFAP2A-IRF6-GRHL3 genetic pathway is conserved in neurulation","volume":"28","author":"Kousa","year":"2019","journal-title":"Hum. Mol. Genet."},{"key":"2021042700462128800_DEV194498C36","doi-asserted-by":"publisher","first-page":"2417","DOI":"10.1016\/j.celrep.2018.10.097","article-title":"Esrp1-regulated splicing of Arhgef11 isoforms is required for epithelial tight junction integrity","volume":"25","author":"Lee","year":"2018","journal-title":"Cell Rep."},{"key":"2021042700462128800_DEV194498C37","doi-asserted-by":"publisher","first-page":"dev187369","DOI":"10.1242\/dev.187369","article-title":"Cleft lip and cleft palate in Esrp1 knockout mice is associated with alterations in epithelial-mesenchymal crosstalk","volume":"147","author":"Lee","year":"2020","journal-title":"Development"},{"key":"2021042700462128800_DEV194498C38","doi-asserted-by":"publisher","first-page":"2535","DOI":"10.1002\/ajmg.a.36133","article-title":"Search for genetic modifiers of IRF6 and genotype-phenotype correlations in Van der Woude and popliteal pterygium syndromes","volume":"161A","author":"Leslie","year":"2013","journal-title":"Am. J. Med. Genet. A"},{"key":"2021042700462128800_DEV194498C39","doi-asserted-by":"publisher","first-page":"e1007009","DOI":"10.1371\/journal.pgen.1007009","article-title":"Rapid functional analysis of computationally complex rare human IRF6 gene variants using a novel zebrafish model","volume":"13","author":"Li","year":"2017","journal-title":"PLoS Genet."},{"key":"2021042700462128800_DEV194498C40","doi-asserted-by":"publisher","first-page":"dev174888","DOI":"10.1242\/dev.174888","article-title":"The molecular anatomy of mammalian upper lip and primary palate fusion at single cell resolution","volume":"146","author":"Li","year":"2019","journal-title":"Development"},{"key":"2021042700462128800_DEV194498C41","doi-asserted-by":"publisher","first-page":"353","DOI":"10.1038\/nrg2091","article-title":"Animal models of human disease: zebrafish swim into view","volume":"8","author":"Lieschke","year":"2007","journal-title":"Nat. Rev. Genet."},{"key":"2021042700462128800_DEV194498C42","doi-asserted-by":"publisher","first-page":"3477","DOI":"10.1073\/pnas.051614698","article-title":"Down-regulation of dendritic spine and glutamic acid decarboxylase 67 expressions in the reelin haploinsufficient heterozygous reeler mouse","volume":"98","author":"Liu","year":"2001","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2021042700462128800_DEV194498C43","doi-asserted-by":"publisher","first-page":"766","DOI":"10.1093\/hmg\/ddv614","article-title":"Irf6 directly regulates Klf17 in zebrafish periderm and Klf4 in murine oral epithelium, and dominant-negative KLF4 variants are present in patients with cleft lip and palate","volume":"25","author":"Liu","year":"2016","journal-title":"Hum. Mol. Genet."},{"key":"2021042700462128800_DEV194498C44","doi-asserted-by":"publisher","first-page":"13","DOI":"10.3389\/fcell.2019.00013","article-title":"Expanding the CRISPR toolbox in zebrafish for studying development and disease","volume":"7","author":"Liu","year":"2019","journal-title":"Front. Cell Dev. Biol."},{"key":"2021042700462128800_DEV194498C45","doi-asserted-by":"publisher","first-page":"dev157628","DOI":"10.1242\/dev.157628","article-title":"Face morphogenesis is promoted by Pbx-dependent EMT via regulation of Snail1 during frontonasal prominence fusion","volume":"145","author":"Losa","year":"2018","journal-title":"Development"},{"key":"2021042700462128800_DEV194498C46","doi-asserted-by":"publisher","first-page":"263","DOI":"10.1146\/annurev-genom-090711-163729","article-title":"The evolution of human genetic studies of cleft lip and cleft palate","volume":"13","author":"Marazita","year":"2012","journal-title":"Annu. Rev. Genomics Hum. Genet."},{"key":"2021042700462128800_DEV194498C47","doi-asserted-by":"publisher","first-page":"610","DOI":"10.2144\/000112619","article-title":"Method for isolation of PCR-ready genomic DNA from zebrafish tissues","volume":"43","author":"Meeker","year":"2007","journal-title":"BioTechniques"},{"key":"2021042700462128800_DEV194498C48","doi-asserted-by":"publisher","first-page":"W401","DOI":"10.1093\/nar\/gku410","article-title":"CHOPCHOP: a CRISPR\/Cas9 and TALEN web tool for genome editing","volume":"42","author":"Montague","year":"2014","journal-title":"Nucleic Acids Res."},{"key":"2021042700462128800_DEV194498C49","doi-asserted-by":"publisher","first-page":"196","DOI":"10.1038\/nchembio.1430","article-title":"An optogenetic gene expression system with rapid activation and deactivation kinetics","volume":"10","author":"Motta-Mena","year":"2014","journal-title":"Nat. Chem. Biol."},{"key":"2021042700462128800_DEV194498C50","doi-asserted-by":"publisher","first-page":"113","DOI":"10.1016\/j.biopsych.2019.07.032","article-title":"Brain development in school-age and adolescent girls: effects of turner syndrome, estrogen therapy, and genomic imprinting","volume":"87","author":"O'donoghue","year":"2020","journal-title":"Biol. Psychiatry"},{"key":"2021042700462128800_DEV194498C51","doi-asserted-by":"publisher","first-page":"2281","DOI":"10.1038\/nprot.2013.143","article-title":"Genome engineering using the CRISPR-Cas9 system","volume":"8","author":"Ran","year":"2013","journal-title":"Nat. Protoc."},{"key":"2021042700462128800_DEV194498C52","first-page":"259","article-title":"Ectodermal dysplasia: case with an unusual combination of dental features","volume":"41","author":"Reid","year":"1986","journal-title":"J. Oral. Med."},{"key":"2021042700462128800_DEV194498C53","doi-asserted-by":"publisher","first-page":"261","DOI":"10.1016\/j.ydbio.2010.11.012","article-title":"Ectodermal Wnt\/beta-catenin signaling shapes the mouse face","volume":"349","author":"Reid","year":"2011","journal-title":"Dev. Biol."},{"key":"2021042700462128800_DEV194498C54","doi-asserted-by":"publisher","first-page":"1329","DOI":"10.1038\/ng1894","article-title":"Irf6 is a key determinant of the keratinocyte proliferation-differentiation switch","volume":"38","author":"Richardson","year":"2006","journal-title":"Nat. Genet."},{"key":"2021042700462128800_DEV194498C55","doi-asserted-by":"publisher","first-page":"1314","DOI":"10.1002\/ajmg.a.33980","article-title":"Genomic strategy identifies a missense mutation in WD-repeat domain 65 (WDR65) in an individual with Van der Woude syndrome","volume":"155A","author":"Rorick","year":"2011","journal-title":"Am. J. Med. Genet. A"},{"key":"2021042700462128800_DEV194498C56","doi-asserted-by":"publisher","first-page":"230","DOI":"10.1038\/nature14580","article-title":"Genetic compensation induced by deleterious mutations but not gene knockdowns","volume":"524","author":"Rossi","year":"2015","journal-title":"Nature"},{"key":"2021042700462128800_DEV194498C57","doi-asserted-by":"publisher","first-page":"249","DOI":"10.1016\/j.ydbio.2008.10.031","article-title":"Maternal interferon regulatory factor 6 is required for the differentiation of primary superficial epithelia in danio and Xenopus embryos","volume":"325","author":"Sabel","year":"2009","journal-title":"Dev. Biol."},{"key":"2021042700462128800_DEV194498C58","doi-asserted-by":"publisher","first-page":"W599","DOI":"10.1093\/nar\/gkm349","article-title":"Zinc Finger Targeter (ZiFiT): an engineered zinc finger\/target site design tool","volume":"35","author":"Sander","year":"2007","journal-title":"Nucleic Acids Res."},{"key":"2021042700462128800_DEV194498C59","doi-asserted-by":"publisher","first-page":"101","DOI":"10.1186\/jbiol205","article-title":"Fishing for the signals that pattern the face","volume":"8","author":"Schilling","year":"2009","journal-title":"J. Biol."},{"key":"2021042700462128800_DEV194498C60","doi-asserted-by":"publisher","first-page":"2204","DOI":"10.1002\/dvdy.22713","article-title":"Examination of a palatogenic gene program in zebrafish","volume":"240","author":"Swartz","year":"2011","journal-title":"Dev. Dyn."},{"key":"2021042700462128800_DEV194498C61","doi-asserted-by":"publisher","first-page":"59","DOI":"10.1038\/nprot.2007.514","article-title":"High-resolution in situ hybridization to whole-mount zebrafish embryos","volume":"3","author":"Thisse","year":"2008","journal-title":"Nat. Protoc."},{"key":"2021042700462128800_DEV194498C62","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1016\/j.ydbio.2016.01.017","article-title":"The old and new face of craniofacial research: How animal models inform human craniofacial genetic and clinical data","volume":"415","author":"Van Otterloo","year":"2016","journal-title":"Dev. Biol."},{"key":"2021042700462128800_DEV194498C63","doi-asserted-by":"publisher","first-page":"591","DOI":"10.1016\/j.molcel.2009.01.025","article-title":"ESRP1 and ESRP2 are epithelial cell-type-specific regulators of FGFR2 splicing","volume":"33","author":"Warzecha","year":"2009","journal-title":"Mol. Cell"},{"key":"2021042700462128800_DEV194498C64","doi-asserted-by":"publisher","first-page":"14364","DOI":"10.1038\/ncomms14364","article-title":"Genome-wide analyses of non-syndromic cleft lip with palate identify 14 novel loci and genetic heterogeneity","volume":"8","author":"Yu","year":"2017","journal-title":"Nat. Commun."},{"key":"2021042700462128800_DEV194498C65","doi-asserted-by":"publisher","first-page":"107","DOI":"10.1159\/000322486","article-title":"Genetic causes of nonsyndromic cleft lip with or without cleft palate","volume":"70","author":"Yuan","year":"2011","journal-title":"Adv. Otorhinolaryngol."},{"key":"2021042700462128800_DEV194498C66","doi-asserted-by":"publisher","first-page":"769","DOI":"10.1056\/NEJMoa032909","article-title":"Interferon regulatory factor 6 (IRF6) gene variants and the risk of isolated cleft lip or palate","volume":"351","author":"Zucchero","year":"2004","journal-title":"N. Engl. J. Med."}],"container-title":["Development"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1242\/dev.194498","content-type":"unspecified","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/journals.biologists.com\/dev\/article-pdf\/doi\/10.1242\/dev.194498\/1970063\/dev194498.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/journals.biologists.com\/dev\/article-pdf\/doi\/10.1242\/dev.194498\/2065155\/dev_194498v1.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"http:\/\/journals.biologists.com\/dev\/article-pdf\/doi\/10.1242\/dev.194498\/2065155\/dev_194498v1.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,5,18]],"date-time":"2021-05-18T22:51:19Z","timestamp":1621378279000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dev\/article\/doi\/10.1242\/dev.194498\/267004\/An-Irf6-Esrp1-2-regulatory-axis-controls-midface"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,1,1]]},"references-count":66,"URL":"https:\/\/doi.org\/10.1242\/dev.194498","relation":{},"ISSN":["1477-9129","0950-1991"],"issn-type":[{"value":"1477-9129","type":"electronic"},{"value":"0950-1991","type":"print"}],"subject":[],"published":{"date-parts":[[2020,1,1]]},"article-number":"dev.194498"}}