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Quantitative and qualitative parameters were compared between OI type III and type IV groups (unpaired <jats:italic>t<\/jats:italic> test) and the unaffected population (<jats:italic>Z<\/jats:italic>\u2010score). 3D evaluation revealed a high prevalence of craniocervical abnormalities, craniofacial asymmetries, and nasal septum deviation in both OI groups. Mean airway dimensions were comparable to the non\u2010affected population norms, except for 5 individuals who had insufficient airway dimensions. In 2D, the maxilla was retrognathic and hypoplastic, and the mandibular position was convergent with respect to the face, resulting in mandibular prognathism and face height reduction. The 2D trends were more pronounced in OI type III, whereas the 3D craniocervical and airway abnormalities were common in both types. This study illustrates the prevalence of craniofacial and airway anomalies in OI that occur along with facial deformities are not associated with postcranial phenotype and OI type, are apparent only in 3D evaluation, and are likely to influence treatment strategy. For OI patients, a team effort involving a dentist, orthodontist, neurologist, and ear\u2010nose\u2010throat (ENT) practitioner is recommended for successful management of craniofacial deformities. \u00a9 2018 The Authors <jats:italic>JBMR Plus<\/jats:italic> published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.<\/jats:p><\/jats:sec>","DOI":"10.1002\/jbm4.10124","type":"journal-article","created":{"date-parts":[[2018,11,16]],"date-time":"2018-11-16T20:49:37Z","timestamp":1542401377000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":16,"title":["Cone\u2010Beam Computed Tomography of Osteogenesis Imperfecta Types III and IV: Three\u2010Dimensional Evaluation of Craniofacial Features and Upper Airways"],"prefix":"10.1093","volume":"3","author":[{"given":"Natalie","family":"Reznikov","sequence":"first","affiliation":[{"name":"Faculty of Dentistry McGill University Montreal 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