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The phenotypically distinct syndromes suggest that these TZ proteins have differing functions. However, mutations in a single TZ gene can result in multiple syndromes, suggesting that the phenotype is influenced by modifier genes. We performed a comprehensive analysis of ten zebrafish TZ mutants, including mks1, tmem216, tmem67, rpgrip1l, cc2d2a, b9d2, cep290, tctn1, nphp1 and nphp4, as well as mutants in ift88 and ift172. Our data indicate that variations in phenotypes exist between different TZ mutants, supporting different tissue-specific functions of these TZ genes. Further, we observed phenotypic variations within progeny of a single TZ mutant, reminiscent of multiple disease syndromes being associated with mutations in one gene. In some mutants, the dynamics of the phenotype became complex with transitory phenotypes that are corrected over time. We also demonstrated that multiple-guide-derived CRISPR\/Cas9 F0 \u2018crispant\u2019 embryos recapitulate zygotic null phenotypes, and rapidly identified ciliary phenotypes in 11 cilia-associated gene candidates (ankfn1, ccdc65, cfap57, fhad1, nme7, pacrg, saxo2, c1orf194, ttc26, zmynd12 and cfap52).<\/jats:p>","DOI":"10.1242\/dmm.049568","type":"journal-article","created":{"date-parts":[[2022,12,19]],"date-time":"2022-12-19T09:31:50Z","timestamp":1671442310000},"source":"Crossref","is-referenced-by-count":29,"title":["Variable phenotypes and penetrance between and within different zebrafish ciliary transition zone mutants"],"prefix":"10.1242","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3413-5208","authenticated-orcid":false,"given":"Jun","family":"Wang","sequence":"first","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , Birmingham, AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Holly R.","family":"Thomas","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , Birmingham, AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert G.","family":"Thompson","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , Birmingham, AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephanie C.","family":"Waldrep","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , Birmingham, AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph","family":"Fogerty","sequence":"additional","affiliation":[{"name":"Cole Eye Institute, Cleveland Clinic 2 Department of Ophthalmic Research , , Cleveland, OH 44106 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ping","family":"Song","sequence":"additional","affiliation":[{"name":"Cole Eye Institute, Cleveland Clinic 2 Department of Ophthalmic Research , , Cleveland, OH 44106 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhang","family":"Li","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham 3 Department of Cell, Developmental and Integrative Biology , , AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yongjie","family":"Ma","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , Birmingham, AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peu","family":"Santra","sequence":"additional","affiliation":[{"name":"SUNY Upstate Medical University 4 Department of Cell and Developmental Biology , , Syracuse, NY 13210 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jonathan D.","family":"Hoover","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , Birmingham, AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nan Cher","family":"Yeo","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , Birmingham, AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Iain A.","family":"Drummond","sequence":"additional","affiliation":[{"name":"Davis Center for Aging and Regeneration, Mount Desert Island Biological Laboratory 5 , 159 Old Bar Harbor Road, Bar Harbor, ME 04609 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bradley K.","family":"Yoder","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham 3 Department of Cell, Developmental and Integrative Biology , , AL 35294 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jeffrey D.","family":"Amack","sequence":"additional","affiliation":[{"name":"SUNY Upstate Medical University 4 Department of Cell and Developmental Biology , , Syracuse, NY 13210 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brian","family":"Perkins","sequence":"additional","affiliation":[{"name":"Cole Eye Institute, Cleveland Clinic 2 Department of Ophthalmic Research , , Cleveland, OH 44106 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9084-5098","authenticated-orcid":false,"given":"John M.","family":"Parant","sequence":"additional","affiliation":[{"name":"University of Alabama at Birmingham School of Medicine 1 Department of Pharmacology and Toxicology , , 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