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Currently, with the advances in genetic screening strategies, an increasing number of novel variants and their roles in orofacial diseases have been explored. Zebrafish is recognized as a powerful animal model, and its homologous genes and similar oral structure and development process provide an ideal platform for studying the contributions of genetic and environmental factors to human craniofacial malformations. Here, we reviewed zebrafish models for the study of craniomaxillofacial developmental anomalies, such as human nonsyndromic cleft lip with or without an affected palate and jaw and tooth developmental anomalies. Due to its potential for gene expression and regulation research, zebrafish may provide new perspectives for understanding craniomaxillofacial diseaseand its treatment.<\/jats:p>","DOI":"10.1002\/bdr2.2014","type":"journal-article","created":{"date-parts":[[2022,4,19]],"date-time":"2022-04-19T06:52:02Z","timestamp":1650351122000},"page":"583-595","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Application of zebrafish in the study of craniomaxillofacial developmental anomalies"],"prefix":"10.1002","volume":"114","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7649-5201","authenticated-orcid":false,"given":"Kang","family":"Li","sequence":"first","affiliation":[{"name":"Jiangsu Province Key Laboratory of Oral Diseases Nanjing Medical University  Nanjing China"},{"name":"Jiangsu Province Engineering Research Center of Stomatological Translational Medicine Nanjing Medical University  Nanjing 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