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However, most of their targets were limited to non\u2010syndromic diseases that exhibit abnormalities in a small fraction of tissues or organs in the body. This is likely because of the complexity of interpreting the phenotypic variations resulting from somatic mosaic mutations generated in the founder animals (crispants). In this study, we attempted to model the syndromic disease campomelic dysplasia (CD) by generating <jats:italic>sox9<\/jats:italic> crispants in <jats:italic>X. tropicalis<\/jats:italic>. The resulting crispants failed to form neural crest cells at neurula stages and exhibited various combinations of jaw, gill, ear, heart, and gut defects at tadpole stages, recapitulating part of the syndromic phenotype of CD patients. Genotyping of the crispants with a variety of allelic series of mutations suggested that the heart and gut defects depend primarily on frame\u2010shift mutations expected to be null, whereas the jaw, gill, and ear defects could be induced not only by such mutations but also by in\u2010frame deletion mutations expected to delete part of the jawed vertebrate\u2010specific domain from the encoded Sox9 protein. These results demonstrate that <jats:italic>Xenopus<\/jats:italic> crispants are useful for investigating the phenotype\u2013genotype relationships behind syndromic diseases and examining the tissue\u2010specific role of each functional domain within a single protein, providing novel insights into vertebrate jaw evolution.<\/jats:p>","DOI":"10.1111\/dgd.12884","type":"journal-article","created":{"date-parts":[[2023,7,28]],"date-time":"2023-07-28T15:48:17Z","timestamp":1690559297000},"page":"481-497","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Phenotype\u2013genotype relationships in <i>Xenopus sox9<\/i> crispants provide insights into campomelic dysplasia and vertebrate jaw evolution"],"prefix":"10.1111","volume":"65","author":[{"ORCID":"https:\/\/orcid.org\/0009-0005-4828-6874","authenticated-orcid":false,"given":"Nusrat","family":"Hossain","sequence":"first","affiliation":[{"name":"Amphibian Research Center, 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