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Dental and craniofacial manifestations have not been a focus of case reports to date. We analyzed the craniofacial and dental phenotype of <jats:italic>Crtap<\/jats:italic><jats:sup>\u2212\/\u2212<\/jats:sup> mice by skull measurements, micro\u2010computed tomography (micro\u2010CT), histology, and immunohistochemistry.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p><jats:italic>Crtap<\/jats:italic><jats:sup>\u2212\/\u2212<\/jats:sup> mice exhibited a brachycephalic skull shape with fusion of the nasofrontal suture and facial bones, resulting in mid\u2010face retrusion and a class III dental malocclusion. Loss of CRTAP also resulted in decreased dentin volume and decreased cellular cementum volume, though acellular cementum thickness was increased. Periodontal dysfunction was revealed by decreased alveolar bone volume and mineral density, increased periodontal ligament (PDL) space, ectopic calcification within the PDL, bone\u2010tooth ankylosis, altered immunostaining of extracellular matrix proteins in bone and PDL, increased pSMAD5, and more numerous osteoclasts on alveolar bone surfaces.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p><jats:italic>Crtap<\/jats:italic><jats:sup>\u2212\/\u2212<\/jats:sup> mice serve as a useful model of the dental and craniofacial abnormalities seen in individuals with osteogenesis imperfecta type VII.<\/jats:p><\/jats:sec>","DOI":"10.1002\/dvdy.166","type":"journal-article","created":{"date-parts":[[2020,3,5]],"date-time":"2020-03-05T04:45:32Z","timestamp":1583383532000},"page":"884-897","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":22,"title":["Dental and craniofacial defects in the 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