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Here, we applied a comprehensive bioinformatic approach on ChIP\u2010seq, RNA\u2010seq, and gene expression array studies to identify novel transcriptional targets of HNF1\u03b2 explaining the kidney phenotype of HNF1\u03b2 patients. We identified BAR\/IMD Domain Containing Adaptor Protein 2 Like 2 (\n                    <jats:italic>BAIAP2L2<\/jats:italic>\n                    ), as a novel transcriptional target of HNF1\u03b2 and validated direct transcriptional activation of the\n                    <jats:italic>BAIAP2L2<\/jats:italic>\n                    promoter by a reporter luciferase assay. Using mass spectrometry analysis, we show that BAIAP2L2 binds to other members of the I\u2010BAR domain\u2010containing family: BAIAP2 and BAIAP2L1. Subsequently, the role of BAIAP2L2 in maintaining epithelial cell integrity in the kidney was assessed using\n                    <jats:italic>Baiap2l2<\/jats:italic>\n                    knockout cell and mouse models. Kidney epithelial cells lacking functional BAIAP2L2 displayed normal F\u2010actin distribution at cell\u2013cell contacts and formed polarized three\u2010dimensional spheroids with a lumen. In vivo,\n                    <jats:italic>Baiap2l2<\/jats:italic>\n                    knockout mice displayed normal kidney and colon tissue morphology and serum and urine electrolyte concentrations were not affected. Altogether, our study is the first to characterize the function of BAIAP2L2 in the kidney in vivo and we report that mice lacking BAIAP2L2 exhibit normal electrolyte homeostasis and tissue morphology under physiological conditions.\n                  <\/jats:p>","DOI":"10.1096\/fj.202201121r","type":"journal-article","created":{"date-parts":[[2022,12,15]],"date-time":"2022-12-15T10:02:23Z","timestamp":1671098543000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["<scp>HNF1\u03b2<\/scp>\n                    \u2010associated cyst development and electrolyte disturbances are not explained by\n                    <i>BAIAP2L2<\/i>\n                    expression"],"prefix":"10.1096","volume":"37","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2838-8636","authenticated-orcid":false,"given":"Lotte E.","family":"Tholen","sequence":"first","affiliation":[{"name":"Department of Physiology, Radboud Institute for Molecular Life Sciences Radboud University Medical Center  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1934-6450","authenticated-orcid":false,"given":"Heidi","family":"Schigt","sequence":"additional","affiliation":[{"name":"Department of Physiology, Radboud Institute for Molecular Life Sciences Radboud University Medical Center  Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sanne G. 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