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Alterations to the face are common phenotypes in FASD. While alcohol exposure is the underlying cause of FASD, many variables modify the outcomes of such exposures. Genetic risk is one such variable, yet we still have a limited understanding of the nature of the genetic loci mediating susceptibility to FASD.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We employed ENU\u2010based random mutagenesis in zebrafish to identify mutations that enhanced the teratogenicity of ethanol (EtOH). F3 embryos obtained from 126 inbred F2 families were exposed to 1% EtOH in the medium (approximately 41\u00a0mM tissue levels). Zebrafish stained with Alcian Blue and Alizarin Red were screened for qualitative alterations to the craniofacial skeleton between 4 and 7\u00a0days postfertilization (dpf).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>In all, we recovered 6 EtOH\u2010sensitive mutants, 5 from the genetic screen itself and one as a background mutation in one of our wild\u2010type lines. Each mutant has a unique EtOH\u2010induced phenotype relative to the other mutant lines. All but 1 mutation appears to be recessive in nature, and only 1 mutant, au29, has apparent craniofacial defects in the absence of EtOH. To validate the genetic screen, we genetically mapped au29 and found that it carries a mutation in a previously uncharacterized gene, si:dkey\u201088l16.3.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>The phenotypes of these EtOH\u2010sensitive mutants differ from those in previous characterizations of gene\u2013EtOH interactions. Thus, each mutant is likely to provide novel insights into EtOH teratogenesis. Given that most of these mutants only have craniofacial defects in the presence of EtOH and our mapping of au29, it is also likely that many of the mutants will be previously uncharacterized. Collectively, our findings point to the importance of unbiased genetic screens in the identification, and eventual characterization, of risk alleles for FASD.<\/jats:p><\/jats:sec>","DOI":"10.1111\/acer.14240","type":"journal-article","created":{"date-parts":[[2019,11,19]],"date-time":"2019-11-19T18:32:08Z","timestamp":1574188328000},"page":"56-65","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Novel Ethanol\u2010Sensitive Mutants Identified in an F3 Forward Genetic Screen"],"prefix":"10.1111","volume":"44","author":[{"given":"Mary E.","family":"Swartz","sequence":"first","affiliation":[{"name":"Department of Molecular Biosciences Waggoner Center for Alcohol and Addiction Research Austin Texas"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charles Ben","family":"Lovely","sequence":"additional","affiliation":[{"name":"Department of Molecular Biosciences Waggoner Center for Alcohol and Addiction 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