2021
DOI: 10.1101/2021.02.27.433115
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Single-cell analysis identifies a key role forHhipin murine coronal suture development

Abstract: Craniofacial development depends on proper formation and maintenance of sutures between adjacent bones of the skull. Within sutures, bone growth occurs through proliferation and differentiation of osteoprogenitors to osteoblasts within the osteogenic front at the edge of each bone, and suture mesenchyme maintains the separation between them. Many genes have been found that cause suture dysgenesis when mutated, particularly of the coronal suture. Such genes are largely studied in isolation, and there is little … Show more

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“…Dermal bones of the skull arise individually in association with chondrocranial cartilages (2,4) but are ultimately linked with other bones by sutures. Sutures are joints that facilitate circumferential growth of the skull through the coupled functions of suture mesenchyme that keeps adjacent bone separate, differentiating osteoblasts occupying a zone between the mesenchyme and osteogenic bony fronts, and addition of unmineralized matrix along the osteogenic fronts of individual bones (6)(7)(8)(9). In craniosynostosis, osteoblasts that mineralize the suture before the completion of brain growth alter subsequent skull growth patterns and produce abnormal head shapes (10).…”
Section: Introductionmentioning
confidence: 99%
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“…Dermal bones of the skull arise individually in association with chondrocranial cartilages (2,4) but are ultimately linked with other bones by sutures. Sutures are joints that facilitate circumferential growth of the skull through the coupled functions of suture mesenchyme that keeps adjacent bone separate, differentiating osteoblasts occupying a zone between the mesenchyme and osteogenic bony fronts, and addition of unmineralized matrix along the osteogenic fronts of individual bones (6)(7)(8)(9). In craniosynostosis, osteoblasts that mineralize the suture before the completion of brain growth alter subsequent skull growth patterns and produce abnormal head shapes (10).…”
Section: Introductionmentioning
confidence: 99%
“…Approximately 1 in 2,000-2,500 children of all ethnic groups are born with craniosynostosis conditions (12,13) and though variants of many genes are associated with these disorders (8,(13)(14)(15)(16)(17)(18)(19)(20), alteration to the function of fibroblast growth factor receptor 2 (FGFR2) results in the more common craniosynostosis syndromes of Apert, Crouzon, and Pfeiffer. The Fgfr2c C342Y/+ Crouzon syndrome mouse model carries a cysteine to tyrosine substitution at amino acid 342 (Cys342Tyr; C342Y) in Fgfr2 equivalent to the FGFR2 mutation most often associated with Crouzon syndrome.…”
Section: Introductionmentioning
confidence: 99%
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