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Here, we provide novel insights into the temporospatial requirement for cilia in SBDs arising from deficiency in\n                    <jats:italic>Ift140<\/jats:italic>\n                    , an intraflagellar transport (IFT) protein regulating ciliogenesis.\n                    <jats:italic>Ift140<\/jats:italic>\n                    -deficient mice exhibit cilia defects accompanied by wide spectrum of SBDs including macrostomia (craniofacial defects), exencephaly, body wall defects, tracheoesophageal fistula (TEF), randomized heart looping, congenital heart defects (CHDs), lung hypoplasia, renal anomalies, and polydactyly. Tamoxifen inducible\n                    <jats:italic>CAGGCre-ER<\/jats:italic>\n                    deletion of a floxed\n                    <jats:italic>Ift140<\/jats:italic>\n                    allele between E5.5 to 9.5 revealed early requirement for\n                    <jats:italic>Ift140<\/jats:italic>\n                    in left-right heart looping regulation, mid to late requirement for cardiac outflow septation and alignment, and late requirement for craniofacial development and body wall closure. Surprisingly, CHD were not observed with 4 Cre drivers targeting different lineages essential for heart development, but craniofacial defects and omphalocele were observed with\n                    <jats:italic>Wnt1-Cre<\/jats:italic>\n                    targeting neural crest and\n                    <jats:italic>Tbx18-Cre<\/jats:italic>\n                    targeting epicardial lineage and rostral sclerotome through which trunk neural crest cells migrate. These findings revealed cell autonomous role of cilia in cranial\/trunk neural crest-mediated craniofacial and body wall closure defects, while non-cell autonomous multi-lineage interactions underlie CHD pathogenesis, revealing unexpected developmental complexity for CHD associated with ciliopathies.\n                  <\/jats:p>","DOI":"10.1371\/journal.pbio.3002425","type":"journal-article","created":{"date-parts":[[2023,12,11]],"date-time":"2023-12-11T13:37:48Z","timestamp":1702301868000},"page":"e3002425","update-policy":"https:\/\/doi.org\/10.1371\/journal.pbio.corrections_policy","source":"Crossref","is-referenced-by-count":1,"title":["Autonomous and non-cell autonomous role of cilia in structural birth defects in mice"],"prefix":"10.1371","volume":"21","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4401-1946","authenticated-orcid":true,"given":"Richard J. 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