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In a previous study, expression screening identified a cohort of genes showing UPC\u2010enriched expression including <jats:italic>D17H6S56E\u20105<\/jats:italic>, <jats:italic>Hs3st3a1<\/jats:italic>, <jats:italic>Hs3st3b1<\/jats:italic>, and <jats:italic>Tmem59l<\/jats:italic>. Each of these is also enriched in branch tips of assembling airways of the developing lungs. Here, we used Crispr\u2010CAS9 directed gene editing to mutate each of these targets to address their potential role(s) in UPC programs.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Single (<jats:italic>D17H6S56E\u20105<\/jats:italic> and <jats:italic>Tmem59<\/jats:italic>l) and double (<jats:italic>Hs3st3a1<\/jats:italic> and <jats:italic>Hs3st3b1<\/jats:italic>) mutants were viable, fertile, and displayed varying frequencies of ureter duplications and no overt lung phenotype. Ureter duplications arise spontaneously through multiple outgrowths of the ureteric bud at the onset of kidney development. <jats:italic>Tmem59l<\/jats:italic> mutants and <jats:italic>Hs3st3a1<\/jats:italic>\/<jats:italic>Hs3st3b1<\/jats:italic> compound mutants showed a weakly penetrant, but statistically significant increase in duplicated ureters compared to C57BL6\/J and SW wild\u2010type mouse strains.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p><jats:italic>Tmem59l<\/jats:italic> and <jats:italic>Hs3st3a1<\/jats:italic>\/<jats:italic>Hs3st3b1<\/jats:italic> activities contribute to the regulatory programs restricting ureteric outgrowth in the developing mouse kidney. However, the low penetrance of the observed phenotype precludes a detailed analysis of their specific actions.<\/jats:p><\/jats:sec>","DOI":"10.1002\/dvdy.157","type":"journal-article","created":{"date-parts":[[2020,2,4]],"date-time":"2020-02-04T09:44:02Z","timestamp":1580809442000},"page":"765-774","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Mutational analysis of genes with ureteric progenitor cell\u2010specific expression in branching morphogenesis of the mouse kidney"],"prefix":"10.1002","volume":"249","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3540-2950","authenticated-orcid":false,"given":"Elisabeth A.","family":"Rutledge","sequence":"first","affiliation":[{"name":"Department of Stem Cell Biology and Regenerative Medicine, Eli and Edythe Broad\u2010CIRM Center for Regenerative Medicine and Stem Cell Research W.M. Keck School of Medicine of the University of Southern California  Los Angeles California"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andrew P.","family":"McMahon","sequence":"additional","affiliation":[{"name":"Department of Stem Cell Biology and Regenerative Medicine, Eli and Edythe Broad\u2010CIRM Center for Regenerative Medicine and Stem Cell Research W.M. 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