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In this study, we chronologically evaluated skull morphology change after abnormal fusion of the sutures and synchondroses in mouse models of syndromic CS for further understanding of the disease. We found fusion of the inter\u2010sphenoid synchondrosis (ISS) in Apert syndrome model mice (<jats:italic>Fgfr2<\/jats:italic><jats:sup><jats:italic>S252W<\/jats:italic>\/+<\/jats:sup>) around 3\u2009weeks old as seen in Crouzon syndrome model mice (<jats:italic>Fgfr2c<\/jats:italic><jats:sup><jats:italic>C342Y<\/jats:italic>\/+<\/jats:sup>). We then examined ontogenic trajectories of CS mouse models after 3\u2009weeks of age using geometric morphometrics analyses. Antero\u2010ventral growth of the face was affected in <jats:italic>Fgfr2<\/jats:italic><jats:sup><jats:italic>S252W<\/jats:italic>\/+<\/jats:sup> and <jats:italic>Fgfr2c<\/jats:italic><jats:sup><jats:italic>C342Y<\/jats:italic>\/+<\/jats:sup> mice, while Saethre\u2013Chotzen syndrome model mice (<jats:italic>Twist1<\/jats:italic><jats:sup>+\/\u2212<\/jats:sup>) did not show the ISS fusion and exhibited a similar growth pattern to that of control littermates. Further analysis revealed that the coronal suture synostosis in the CS mouse models induces only the brachycephalic phenotype as a shared morphological feature. Although previous studies suggest that the fusion of the facial sutures during neonatal period is associated with midface hypoplasia, the present study suggests that the progressive postnatal fusion of the cranial synchondrosis also contributes to craniofacial dysmorphology in mouse models of syndromic CS. These morphological trajectories increase our understanding of the progression of syndromic CS skull growth.<\/jats:p>","DOI":"10.1111\/joa.13790","type":"journal-article","created":{"date-parts":[[2022,11,17]],"date-time":"2022-11-17T17:51:07Z","timestamp":1668707467000},"page":"387-401","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":17,"title":["Synchondrosis fusion contributes to the progression of postnatal craniofacial dysmorphology in syndromic craniosynostosis"],"prefix":"10.1111","volume":"242","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0933-2557","authenticated-orcid":false,"given":"Yukiko","family":"Hoshino","sequence":"first","affiliation":[{"name":"Department of Molecular Craniofacial Embryology Graduate School of Medical and Dental Sciences Tokyo Medical and Dental University (TMDU)  Tokyo Japan"},{"name":"Office of New Drug V, Pharmaceuticals and Medical Devices Agency (PMDA)  Tokyo 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