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Many patients with craniofacial phenotypes, such as orofacial clefting and craniosynostosis, also present with limb defects, including polydactyly, syndactyly, brachydactyly, or ectrodactyly. The gene regulatory networks governing craniofacial and limb development initially seem distinct from one another, and yet these birth defects frequently occur together. Both developmental processes are highly conserved among vertebrates, and zebrafish have emerged as an advantageous model due to their high fecundity, relative ease of genetic manipulation, and transparency during development. Here we summarize studies that have used zebrafish models to study human syndromes that present with both craniofacial and limb phenotypes. We discuss the highly conserved processes of craniofacial and limb\/fin development and describe recent zebrafish studies that have explored the function of genes associated with human syndromes with phenotypes in both structures. We attempt to identify commonalities between the two to help explain why craniofacial and limb anomalies often occur together.<\/jats:p>","DOI":"10.1002\/dvg.23407","type":"journal-article","created":{"date-parts":[[2021,1,4]],"date-time":"2021-01-04T12:25:54Z","timestamp":1609763154000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":20,"title":["The power of zebrafish models for understanding the co\u2010occurrence of craniofacial and limb disorders"],"prefix":"10.1002","volume":"59","author":[{"given":"Brittany T.","family":"Truong","sequence":"first","affiliation":[{"name":"Human Medical Genetics &amp; Genomics Graduate Program University of Colorado Denver Anschutz Medical Campus  Aurora Colorado USA"},{"name":"Department of Craniofacial Biology University of Colorado Denver Anschutz Medical Campus  Aurora Colorado USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3003-6042","authenticated-orcid":false,"given":"Kristin B.","family":"Artinger","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Biology University of Colorado Denver Anschutz Medical Campus  Aurora Colorado USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2021,1,4]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.00191"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-7580.2012.01526.x"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1534-5807(02)00163-6"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.modgep.2005.03.002"},{"issue":"8","key":"e_1_2_8_6_1","first-page":"984","article-title":"A population\u2010based study of Craniosynostosis in metropolitan Atlanta, 1989\u20102003","volume":"146","author":"Boulet S. 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