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With over 180 syndromic associations, reaching genetic diagnoses and understanding variations in underlying cellular mechanisms remains a challenge. Variants of FGFR2 are highly associated with craniosynostosis and warrant further investigation. Using the missense mutation FGFR2<jats:sup>W290R<\/jats:sup>, an effective mouse model of Crouzon syndrome, craniofacial features were analyzed using geometric morphometrics across developmental time (E10.5\u2014adulthood, <jats:italic>n<\/jats:italic>\u2009=\u2009665 total). Given the interrelationship between the cranial vault and basicranium in craniosynostosis patients, the basicranium and synchondroses were analyzed in perinates. Embryonic time points showed minimal significant shape differences. However, hetero\u2010 and homozygous mutant perinates and adults showed significant differences in shape and size of the cranial vault, face, and basicranium, which were associated with cranial doming and shortening of the basicranium and skull. Although there were also significant shape and size differences associated with the basicranial bones and clear reductions in basicranial ossification in cleared whole\u2010mount samples, there were no significant alterations in chondrocyte cell shape, size, or orientation along the spheno\u2010occipital synchondrosis. Finally, shape differences in the cranial vault and basicranium were interrelated at perinatal stages. These results point toward the possibility that facial shape phenotypes in craniosynostosis may result in part from pleiotropic effects of the causative mutations rather than only from the secondary consequences of the sutural defects, indicating a novel direction of research that may shed light on the etiology of the broad changes in craniofacial morphology observed in craniosynostosis syndromes.<\/jats:p>","DOI":"10.1002\/ar.25398","type":"journal-article","created":{"date-parts":[[2024,2,27]],"date-time":"2024-02-27T00:25:50Z","timestamp":1708993550000},"page":"1944-1971","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Dosage\u2010dependent effects of <scp>FGFR2<sup>W290R<\/sup><\/scp> mutation on craniofacial shape and cellular dynamics of the basicranial synchondroses"],"prefix":"10.1002","volume":"308","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4081-1073","authenticated-orcid":false,"given":"Heather 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