{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,28]],"date-time":"2026-02-28T04:41:19Z","timestamp":1772253679193,"version":"3.50.1"},"reference-count":31,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2017,10,6]],"date-time":"2017-10-06T00:00:00Z","timestamp":1507248000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01DE019527"],"award-info":[{"award-number":["R01DE019527"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["R01DE020843"],"award-info":[{"award-number":["R01DE020843"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["The Anatomical Record"],"published-print":{"date-parts":[[2018,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:p>EvC syndrome is a type of autosomal\u2010recessive chondrodysplasia. Previous case studies in patients suggest abnormal craniofacial development, in addition to dwarfism and tooth abnormalities. To investigate how craniofacial development is affected in EvC patients, surface models were generated from micro\u2010CT scans of control mice, <jats:italic>Evc2<\/jats:italic> global mutant mice and <jats:italic>Evc2<\/jats:italic> neural crest\u2010specific mutant mice. The anatomic landmarks were placed on the surface model to assess the morphological abnormalities in the <jats:italic>Evc2<\/jats:italic> mutants. Through analyzing the linear and angular measurements between landmarks, we identified a smaller overall skull, shorter nasal bone, shorter frontal bone, and shorter cranial base in the <jats:italic>Evc2<\/jats:italic> global mutants. By comparing neural crest\u2010specific <jats:italic>Evc2<\/jats:italic> mutants with control mice, we demonstrated that the abnormalities within the mid\u2010facial regions are not accounted for by the <jats:italic>Evc2<\/jats:italic> mutation within these regions. Additionally, we also identified disproportionate length to width ratios in the <jats:italic>Evc2<\/jats:italic> mutants at all levels from anterior to posterior of the skull. Overall, this study demonstrates a more comprehensive analysis on the craniofacial morphological abnormalities in EvC syndrome and provides the developmental insight to appreciate the impact of <jats:italic>Evc2<\/jats:italic> mutation within the neural crest cells on multiple aspects of skull deformities. Anat Rec, 2017. \u00a9 2017 Wiley Periodicals, Inc. Anat Rec, 301:46\u201355, 2018. \u00a9 2017 Wiley Periodicals, Inc.<\/jats:p>","DOI":"10.1002\/ar.23692","type":"journal-article","created":{"date-parts":[[2017,9,26]],"date-time":"2017-09-26T12:12:42Z","timestamp":1506427962000},"page":"46-55","source":"Crossref","is-referenced-by-count":18,"title":["The Role of Ellis\u2010Van Creveld 2(<i>EVC2<\/i>) in Mice During Cranial Bone Development"],"prefix":"10.1002","volume":"301","author":[{"given":"Edwin K.","family":"Kwon","sequence":"first","affiliation":[{"name":"Department of Biologic and Materials Sciences, School of Dentistry University of Michigan Michigan"},{"name":"Department of Orthodontics and Pediatric Dentistry, School of Dentistry University of Michigan Michigan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ke'ale","family":"Louie","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences, School of Dentistry University of Michigan Michigan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anshul","family":"Kulkarni","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences, School of Dentistry University of Michigan Michigan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marilia","family":"Yatabe","sequence":"additional","affiliation":[{"name":"Department of Orthodontics and Pediatric Dentistry, School of Dentistry University of Michigan Michigan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Antonio Carlos de Oliveira","family":"Ruellas","sequence":"additional","affiliation":[{"name":"Department of Orthodontics and Pediatric Dentistry, School of Dentistry University of Michigan Michigan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Taylor N.","family":"Snider","sequence":"additional","affiliation":[{"name":"Department of Biologic and Materials Sciences, School of Dentistry University of Michigan Michigan"},{"name":"Department of Orthodontics and Pediatric Dentistry, School of Dentistry University of Michigan Michigan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yoshiyuki","family":"Mochida","sequence":"additional","affiliation":[{"name":"Department of Molecular and Cell Biology, Henry M. 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