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Sutures are a critical connective tissue that regulates bone growth; their aberrant fusion results in abnormal shapes of the head and face. The molecular and cellular mechanisms have been investigated for a long time, but knowledge gaps remain between genetic mutations and mechanisms of pathogenesis for craniosynostosis. We previously demonstrated that the augmentation of bone morphogenetic protein (BMP) signaling through constitutively active BMP type 1A receptor (caBmpr1a) in neural crest cells (NCCs) caused the development of premature fusion of the anterior frontal suture, leading to craniosynostosis in mice. In this study, we demonstrated that ectopic cartilage forms in sutures prior to premature fusion in\n                    <jats:italic>caBmpr1a<\/jats:italic>\n                    mice. The ectopic cartilage is subsequently replaced by bone nodules leading to premature fusion with similar but unique fusion patterns between two neural crest\u2010specific transgenic Cre mouse lines,\n                    <jats:italic>P0\u2010Cre<\/jats:italic>\n                    and\n                    <jats:italic>Wnt1\u2010Cre<\/jats:italic>\n                    mice, which coincides with patterns of premature fusion in each line. Histologic and molecular analyses suggest that endochondral ossification in the affected sutures. Both in vitro and in vivo observations suggest a greater chondrogenic capacity and reduced osteogenic capability of neural crest progenitor cells in mutant lines. These results suggest that the augmentation of BMP signaling alters the cell fate of cranial NCCs toward a chondrogenic lineage to prompt endochondral ossification to prematurely fuse cranial sutures. By comparing\n                    <jats:italic>P0\u2010Cre;caBmpr1a<\/jats:italic>\n                    and\n                    <jats:italic>Wnt1\u2010Cre;caBmpr1a<\/jats:italic>\n                    mice at the stage of neural crest formation, we found more cell death of cranial NCCs in\n                    <jats:italic>P0\u2010Cre;caBmpr1a<\/jats:italic>\n                    than\n                    <jats:italic>Wnt1\u2010Cre;caBmpr1a<\/jats:italic>\n                    mice at the developing facial primordia. These findings may provide a platform for understanding why mutations of broadly expressed genes result in the premature fusion of limited sutures. \u00a9 2022 The Authors.\n                    <jats:italic>JBMR Plus<\/jats:italic>\n                    published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research.\n                  <\/jats:p>","DOI":"10.1002\/jbm4.10716","type":"journal-article","created":{"date-parts":[[2022,12,30]],"date-time":"2022-12-30T05:16:57Z","timestamp":1672377417000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Augmentation of\n                    <scp>BMP<\/scp>\n                    Signaling in Cranial Neural Crest Cells Leads to Premature Cranial Sutures Fusion through Endochondral Ossification in Mice"],"prefix":"10.1093","volume":"7","author":[{"given":"Hiroki","family":"Ueharu","sequence":"first","affiliation":[{"name":"Department of Biologic and Materials Sciences &amp; Prosthodontics University Michigan School of Dentistry  Ann Arbor Michigan USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haichun","family":"Pan","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences &amp; Prosthodontics University Michigan School of Dentistry  Ann Arbor Michigan USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xia","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences &amp; Prosthodontics University Michigan School of Dentistry  Ann Arbor Michigan USA"},{"name":"Department of Oral Pathology Hospital of Stomatology, Jilin University  Changchun China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mamoru","family":"Ishii","sequence":"additional","affiliation":[{"name":"Department of Biochemistry &amp; Molecular Medicine Keck School of Medicine of University of Southern California  Los Angeles California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica","family":"Pongetti","sequence":"additional","affiliation":[{"name":"Department of Orthodontics and Pediatric Dentistry University Michigan School of Dentistry  Ann Arbor Michigan USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anshul K.","family":"Kulkarni","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences &amp; Prosthodontics University Michigan School of Dentistry  Ann Arbor Michigan USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Folasade E.","family":"Adegbenro","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences &amp; Prosthodontics University Michigan School of Dentistry  Ann Arbor Michigan USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jaden","family":"Wurn","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences &amp; Prosthodontics University Michigan School of Dentistry  Ann Arbor Michigan USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert E.","family":"Maxson","sequence":"additional","affiliation":[{"name":"Department of Biochemistry &amp; Molecular Medicine Keck School of Medicine of University of Southern California  Los Angeles California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hongchen","family":"Sun","sequence":"additional","affiliation":[{"name":"Department of Oral Pathology Hospital of Stomatology, Jilin University  Changchun China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3115-1437","authenticated-orcid":false,"given":"Yoshihiro","family":"Komatsu","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Texas Health Science Center  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2338-6992","authenticated-orcid":false,"given":"Honghao","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences &amp; 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