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Disruption of this pathway in mice leads to a series of structural malformations, but the exact role and critical period of the Hh pathway in the early development of the cranial base have been rarely described.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Embryos exposed to vismodegib from E7.5, E9.5, and E10.5 had a higher percentage of cranial base fenestra. The peak incidence of hypoplasia in sphenoid winglets and severe craniosynostosis in cranial base synchondroses was observed when vismodegib was administered between E9.5 and E10.5. Cranial base craniosynostosis results from accelerating terminal differentiation of chondrocytes and premature osteogenesis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>We define the critical periods for the induction of cranial base deformity by vismodegib administration at a meticulous temporal resolution. Our findings suggest that the Hh pathway may play a vital role in the early development of the cranial base. This research also establishes a novel and easy\u2010to\u2010establish mouse model of synostosis in the cranial base using a commercially available pathway\u2010selective inhibitor.<\/jats:p><\/jats:sec>","DOI":"10.1002\/dvdy.270","type":"journal-article","created":{"date-parts":[[2020,11,9]],"date-time":"2020-11-09T17:46:55Z","timestamp":1604944015000},"page":"527-541","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Defining the critical period of hedgehog pathway <scp>inhibitor\u2010induced<\/scp> cranial base dysplasia in mice"],"prefix":"10.1002","volume":"250","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1370-7550","authenticated-orcid":false,"given":"Jiangping","family":"Chen","sequence":"first","affiliation":[{"name":"Fujian Key Laboratory of Oral Diseases &amp; Stomatological Key lab of Fujian College and University School and Hospital of Stomatology, Fujian Medical University  Fuzhou Fujian 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