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This study aimed to identify additional genes contributing to the pathogenesis of BEEC and to verify previously suggested candidate genes. We performed trio\u2010based whole genome sequencing on 19 individuals with BEEC and their unaffected parents; of those, five carried earlier reported microdeletions. The genome data was also filtered in silico for variants in 204 candidate genes selected from databases, publications, and in\u2010house findings. Variants were prioritized based on allele frequency and predicted functional impact. In 8 of the 19 trios, our findings highlight members of the\n                    <jats:italic>ADGR\u2010<\/jats:italic>\n                    gene family as novel candidate genes for BEEC, alongside other implicated genes such as\n                    <jats:italic>TRANK1<\/jats:italic>\n                    ,\n                    <jats:italic>CSNK1E<\/jats:italic>\n                    ,\n                    <jats:italic>IFT122<\/jats:italic>\n                    ,\n                    <jats:italic>SDK1<\/jats:italic>\n                    ,\n                    <jats:italic>SDK2<\/jats:italic>\n                    , and\n                    <jats:italic>KIF19 and propose two more CNVs as risk factors for BEEC<\/jats:italic>\n                    ; on chromosome regions\n                    <jats:italic>1p36 and 16p11.2<\/jats:italic>\n                    . This study identifies novel candidate genes for BEEC within the\n                    <jats:italic>ADGR<\/jats:italic>\n                    gene family. The results also further implicate a complex molecular background of BEEC.\n                  <\/jats:p>","DOI":"10.1002\/ajmga.70074","type":"journal-article","created":{"date-parts":[[2026,2,11]],"date-time":"2026-02-11T04:50:12Z","timestamp":1770785412000},"page":"1286-1305","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Genome Sequencing in 19 Families With Bladder Exstrophy and Epispadias Complex Indicates Involvement of the\n                    <scp>\n                      <i>ADGR<\/i>\n                    <\/scp>\n                    \u2010Gene Family"],"prefix":"10.1002","volume":"200","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6638-4631","authenticated-orcid":false,"given":"Agneta","family":"Nordenskj\u00f6ld","sequence":"first","affiliation":[{"name":"Department of Women's and Children's Health Karolinska Institutet  Stockholm Sweden"},{"name":"Center for Molecular Medicine Karolinska Institutet  Stockholm Sweden"},{"name":"Department of Pediatric Surgery, Astrid Lindgren Children Hospital Karolinska University Hospital  Stockholm Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8339-4783","authenticated-orcid":false,"given":"Samara","family":"Alm","sequence":"additional","affiliation":[{"name":"Department of Women's and Children's Health Karolinska Institutet  Stockholm Sweden"},{"name":"Center for Molecular Medicine Karolinska Institutet  Stockholm Sweden"},{"name":"Department of Urology Ersta Hospital  Stockholm Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3716-4917","authenticated-orcid":false,"given":"Jesper","family":"Eisfeldt","sequence":"additional","affiliation":[{"name":"Center for Molecular Medicine Karolinska Institutet  Stockholm Sweden"},{"name":"Department of Molecular Medicine and Surgery Karolinska Institutet  Stockholm Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0000-4321-8147","authenticated-orcid":false,"given":"Jia","family":"Cao","sequence":"additional","affiliation":[{"name":"Department of Women's and Children's Health Karolinska Institutet  Stockholm Sweden"},{"name":"Center for Molecular Medicine Karolinska Institutet  Stockholm Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4505-338X","authenticated-orcid":false,"given":"Magnus","family":"Anderberg","sequence":"additional","affiliation":[{"name":"Department of Pediatric Surgery Sk\u00e5ne University Hospital  Lund Sweden"},{"name":"Department of Clinical Sciences Lund University  Lund Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gillian","family":"Barker","sequence":"additional","affiliation":[{"name":"Department of Women's and Children's Health Karolinska Institutet  Stockholm Sweden"},{"name":"Department 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