{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,16]],"date-time":"2026-01-16T12:18:21Z","timestamp":1768565901333,"version":"3.49.0"},"reference-count":46,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2019,12,17]],"date-time":"2019-12-17T00:00:00Z","timestamp":1576540800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100007619","name":"Osteology Foundation","doi-asserted-by":"publisher","award":["YG 15\u2010244"],"award-info":[{"award-number":["YG 15\u2010244"]}],"id":[{"id":"10.13039\/501100007619","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The Anatomical Record"],"published-print":{"date-parts":[[2020,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Sclerosteosis, a rare autosomal recessive genetic disorder caused by a mutation of the <jats:italic>Sost<\/jats:italic> gene, manifests in the facial skeleton by gigantism, facial distortion, mandibular prognathism, cranial nerve palsy, and, in extreme cases, compression of the medulla oblongata. Mice lacking sclerostin reflect some symptoms of sclerosteosis, but this is the first report of the effect on the facial skeleton. We used geometric morphometrics (GMM) to analyze the deformations of the murine facial skeleton from the wild\u2010type to the <jats:italic>Sost<\/jats:italic> gene knockout. Landmark coordinates were obtained by surface reconstructions from micro\u2010computed tomography. Centroid size, principal component scores in shape space and form space, and asymmetry were computed by the standard GMM formulas, and dental and skeletal jaw lengths were examined as ratios. We show here that, compared to wild type controls, mice lacking <jats:italic>Sost<\/jats:italic> have larger centroid size (effect size, <jats:italic>p<\/jats:italic>\u2010value: 4.59, &lt;.001), higher mean asymmetry (1.14, .065), dental and skeletal mandibular prognathism (1.36, .010 and 5.92, &lt;.001), a smaller foramen magnum (\u22121.71, .015), and calvaria that are more highly curved (form space <jats:italic>p<\/jats:italic> = 4.09, .002; shape space <jats:italic>p<\/jats:italic> = 12.82, .002). These features of mice lacking sclerostin largely correspond to the changes of the facial skeleton observed in sclerosteosis. This alignment further supports claims that the <jats:italic>Sost<\/jats:italic> gene plays a fundamental role in bony facial development in rodents and humans alike.<\/jats:p>","DOI":"10.1002\/ar.24318","type":"journal-article","created":{"date-parts":[[2019,11,14]],"date-time":"2019-11-14T23:54:26Z","timestamp":1573775666000},"page":"2295-2308","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Anatomical similarity between the <i>Sost<\/i>\u2010knockout mouse and sclerosteosis in humans"],"prefix":"10.1002","volume":"303","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8954-0024","authenticated-orcid":false,"given":"Uwe Y.","family":"Schwarze","sequence":"first","affiliation":[{"name":"Department of Oral Biology, School of Dentistry Medical University of Vienna  Vienna Austria"},{"name":"Austrian Cluster for Tissue Regeneration  Vienna Austria"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Toni","family":"Dobsak","sequence":"additional","affiliation":[{"name":"Austrian Cluster for Tissue Regeneration  Vienna Austria"},{"name":"Department of Oral Surgery Medical University of Vienna  Vienna Austria"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5400-9009","authenticated-orcid":false,"given":"Reinhard","family":"Gruber","sequence":"additional","affiliation":[{"name":"Department of Oral Biology, School of Dentistry Medical University of Vienna  Vienna Austria"},{"name":"Austrian Cluster for Tissue Regeneration  Vienna Austria"},{"name":"Department of Periodontology University of Bern  Bern Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fred L.","family":"Bookstein","sequence":"additional","affiliation":[{"name":"Department of Anthropology University of Vienna  Vienna Austria"},{"name":"Department of Statistics University of Washington  Seattle Washington"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2019,12,17]]},"reference":[{"key":"e_1_2_9_2_1","first-page":"248","article-title":"Classification of malocclusion. 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