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It is essential for the induction of mesonephric and metanephric tubules, the regulation of renal tubule morphogenesis, and the regulation of renal progenitor cell expansion and differentiation. To our knowledge,\n                    <jats:italic>WNT9B<\/jats:italic>\n                    has not been associated with renal defects in humans; however,\n                    <jats:italic>WNT9B<\/jats:italic>\n                    <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    mice have renal agenesis\/hypoplasia and reproductive tract abnormalities. We report four individuals from two unrelated consanguineous families with bilateral renal agenesis\/hypoplasia\/dysplasia and homozygous variants in\n                    <jats:italic>WNT9B<\/jats:italic>\n                    . The proband from Family 1 has bilateral renal cystic dysplasia and chronic kidney disease. He has two deceased siblings who presented with bilateral renal hypoplasia\/agenesis. The three affected family members were homozygous for a missense variant in\n                    <jats:italic>WNT9B<\/jats:italic>\n                    (NM_003396.2: c.949G&gt;A\/p.(Gly317Arg)). The proband from Family 2 has renal hypoplasia\/dysplasia, chronic kidney disease, and is homozygous for a nonsense variant in\n                    <jats:italic>WNT9B<\/jats:italic>\n                    (NM_003396.2: c.11dupC\/p.(Pro5Alafs*52)). Two of her siblings died in the neonatal period, one confirmed to be in the context of oligohydramnios. The proband's unaffected brother is also homozygous for the nonsense variant in\n                    <jats:italic>WNT9B<\/jats:italic>\n                    , suggesting nonpenetrance. We propose a novel association of\n                    <jats:italic>WNT9B<\/jats:italic>\n                    and renal anomalies in humans. Further study is needed to delineate the contribution of\n                    <jats:italic>WNT9B<\/jats:italic>\n                    to genitourinary anomalies in humans.\n                  <\/jats:p>","DOI":"10.1002\/ajmg.a.62398","type":"journal-article","created":{"date-parts":[[2021,6,19]],"date-time":"2021-06-19T03:29:16Z","timestamp":1624073356000},"page":"3005-3011","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["Homozygous\n                    <scp>\n                      <i>WNT9B<\/i>\n                    <\/scp>\n                    variants in two families with bilateral renal agenesis\/hypoplasia\/dysplasia"],"prefix":"10.1002","volume":"185","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2834-6973","authenticated-orcid":false,"given":"Gabrielle","family":"Lemire","sequence":"first","affiliation":[{"name":"Children's Hospital of Eastern Ontario (CHEO) Research Institute, University of Ottawa  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