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Genome\u2010wide association and sequencing studies have identified genes linked to human skeletal diseases. Gene editing of zebrafish models allows researchers to further examine the link between genotype and phenotype, with the long\u2010term goal of improving diagnosis and treatment. While current automated tools enable rapid and in\u2010depth phenotyping of the axial skeleton, characterizing the effects of mutations on the craniofacial skeleton has been more challenging. The objective of this study was to evaluate a semi\u2010automated screening tool can be used to quantify craniofacial variations in zebrafish models using four genes that have been associated with human skeletal diseases (\n                    <jats:italic>meox1<\/jats:italic>\n                    ,\n                    <jats:italic>plod2<\/jats:italic>\n                    ,\n                    <jats:italic>sost<\/jats:italic>\n                    , and\n                    <jats:italic>wnt16<\/jats:italic>\n                    ) as test cases. We used traditional landmarks to ground truth our dataset and pseudolandmarks to quantify variation across the 3D cranial skeleton between the groups (somatic crispant, germline mutant, and control fish). The proposed pipeline identified variation between the crispant or mutant fish and control fish for four genes. Variation in phenotypes parallel human craniofacial symptoms for two of the four genes tested. This study demonstrates the potential as well as the limitations of our pipeline as a screening tool to examine multi\u2010dimensional phenotypes associated with the zebrafish craniofacial skeleton.\n                  <\/jats:p>","DOI":"10.1111\/joa.13847","type":"journal-article","created":{"date-parts":[[2023,3,1]],"date-time":"2023-03-01T20:55:42Z","timestamp":1677704142000},"page":"66-77","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Examining craniofacial variation among crispant and mutant zebrafish models of human skeletal diseases"],"prefix":"10.1111","volume":"243","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8639-6795","authenticated-orcid":false,"given":"Kelly M.","family":"Diamond","sequence":"first","affiliation":[{"name":"Department of Biology Rhodes College Tennessee Memphis USA"},{"name":"Center for Developmental Biology and Regenerative Medicine Seattle Children's Research Institute Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Abigail E.","family":"Burtner","sequence":"additional","affiliation":[{"name":"Department of Biology University of Washington Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daanya","family":"Siddiqui","sequence":"additional","affiliation":[{"name":"Department of Biology University of Washington Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kurtis","family":"Alvarado","sequence":"additional","affiliation":[{"name":"Department of Biology University of Washington Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7663-3187","authenticated-orcid":false,"given":"Sanford","family":"Leake","sequence":"additional","affiliation":[{"name":"Department of Biology University of Washington Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9514-7774","authenticated-orcid":false,"given":"Sara","family":"Rolfe","sequence":"additional","affiliation":[{"name":"Center for Developmental Biology and Regenerative Medicine Seattle Children's Research Institute Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0418-6354","authenticated-orcid":false,"given":"Chi","family":"Zhang","sequence":"additional","affiliation":[{"name":"Center for Developmental Biology and Regenerative Medicine Seattle Children's Research Institute Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9760-3761","authenticated-orcid":false,"given":"Ronald Young","family":"Kwon","sequence":"additional","affiliation":[{"name":"Department of Orthopaedics and Sports Medicine University of Washington Seattle Washington USA"},{"name":"Institute for Stem Cell and Regenerative Medicine University of Washington Seattle Washington USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7921-9018","authenticated-orcid":false,"given":"A. 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