{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,28]],"date-time":"2026-08-28T05:19:22Z","timestamp":1787894362106,"version":"build-2784847793"},"reference-count":47,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2025,12,23]],"date-time":"2025-12-23T00:00:00Z","timestamp":1766448000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2025,12,23]],"date-time":"2025-12-23T00:00:00Z","timestamp":1766448000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100003287","name":"Rally Foundation","doi-asserted-by":"publisher","award":["23CND05"],"award-info":[{"award-number":["23CND05"]}],"id":[{"id":"10.13039\/100003287","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000054","name":"National Cancer Institute","doi-asserted-by":"publisher","award":["1R01CA284531"],"award-info":[{"award-number":["1R01CA284531"]}],"id":[{"id":"10.13039\/100000054","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100009633","name":"Eunice Kennedy Shriver National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["R01HD112081"],"award-info":[{"award-number":["R01HD112081"]}],"id":[{"id":"10.13039\/100009633","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000005","name":"U.S. Department of Defense","doi-asserted-by":"publisher","award":["CA190214"],"award-info":[{"award-number":["CA190214"]}],"id":[{"id":"10.13039\/100000005","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004917","name":"Cancer Prevention and Research Institute of Texas","doi-asserted-by":"publisher","award":["RP210037"],"award-info":[{"award-number":["RP210037"]}],"id":[{"id":"10.13039\/100004917","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2026,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Cancer risk in children with VACTERL, a nonrandom co\u2010occurrence of \u2265\u20093 defects (vertebral, anal, cardiac, tracheoesophogeal fistula, renal, and limb), remains unclear. We evaluated this association in a population\u2010based study. We analyzed data from the Genetic Overlap Between Anomalies and Cancer in Kids (GOBACK) Study, a US registry linkage cohort. VACTERL was defined as the presence of \u2265\u20093 associated defects. Cox regression was applied to estimate hazard ratios (HRs) and 95% confidence intervals (CIs) for cancer risk before age 18 in children with VACTERL compared to children without birth defects. Kaplan\u2013Meier analyses were used to estimate cumulative incidence of cancer in each group. Of 21,224,742 births, 2288 met VACTERL criteria; 8 developed cancer, 5 (63%) of whom were diagnosed with embryonal tumors. Children with VACTERL had a significantly increased cancer risk (HR\u2009=\u20093.0, 95% CI: 1.5\u20136.0), particularly for embryonal tumors (HR\u2009=\u20096.9, 95% CI: 2.9\u201316.5), relative to unaffected children. Cancer incidence was 421.3 (95% CI: 181.9, 830.0) per million person\u2010years for VACTERL versus 133.4 (95% CI: 131.8\u2013135.0) for unaffected children. Children with VACTERL may face increased cancer risk. Shared developmental or epigenetic mechanisms may underlie both conditions, highlighting efforts to identify subgroups that may benefit from targeted surveillance.<\/jats:p>","DOI":"10.1002\/ajmga.70031","type":"journal-article","created":{"date-parts":[[2025,12,23]],"date-time":"2025-12-23T11:13:26Z","timestamp":1766488406000},"page":"1004-1011","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["A Population\u2010Based Assessment of Cancer Risk in Children With\n                    <scp>VACTERL<\/scp>"],"prefix":"10.1002","volume":"200","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3233-5967","authenticated-orcid":false,"given":"Ji Yun","family":"Tark","sequence":"first","affiliation":[{"name":"Section of Epidemiology and Population Sciences, Department of Medicine Baylor College of Medicine  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexander","family":"Renwick","sequence":"additional","affiliation":[{"name":"Section of Epidemiology and Population Sciences, Department of Medicine Baylor College of Medicine  Houston Texas USA"},{"name":"Department of Statistics Rice University  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5210-7219","authenticated-orcid":false,"given":"Giorgio","family":"Tettamanti","sequence":"additional","affiliation":[{"name":"Unit of Epidemiology, Institute of Environmental 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\/0000-0002-8191-5477","authenticated-orcid":false,"given":"Rachel D.","family":"Harris","sequence":"additional","affiliation":[{"name":"Division of Hematology\/Oncology, Department of Pediatrics Baylor College of Medicine  Houston Texas USA"},{"name":"St. Jude Children's Research Hospital  Memphis Tennessee USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tania A.","family":"Desrosiers","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Gillings School of Public Health, University of North Carolina at Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew F.","family":"Olshan","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Gillings School of Public Health, University of North Carolina at Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2690-385X","authenticated-orcid":false,"given":"Amanda E.","family":"Janitz","sequence":"additional","affiliation":[{"name":"Department of Biostatistics and Epidemiology Hudson College of Public Health, University of Oklahoma Health Campus  Oklahoma City Oklahoma USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael E.","family":"Scheurer","sequence":"additional","affiliation":[{"name":"Division of Hematology\/Oncology, Department of Pediatrics Baylor College of Medicine  Houston Texas USA"},{"name":"Department of Pediatrics School of Medicine, Emory University  Atlanta Georgia USA"},{"name":"Aflac Cancer and Blood Disorders Center at Children's Healthcare of Atlanta  Atlanta Georgia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2884-2835","authenticated-orcid":false,"given":"Charles J.","family":"Shumate","sequence":"additional","affiliation":[{"name":"Birth Defects Epidemiology and Surveillance Branch Texas Department of State Health Services  Austin Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Angela E.","family":"Scheuerle","sequence":"additional","affiliation":[{"name":"Department of Pediatrics University of Texas Southwestern Medical Center  Dallas Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sharon E.","family":"Plon","sequence":"additional","affiliation":[{"name":"Division of Hematology\/Oncology, Department of Pediatrics Baylor College of Medicine  Houston Texas USA"},{"name":"Department of Molecular and Human Genetics Baylor College of Medicine  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chad D.","family":"Huff","sequence":"additional","affiliation":[{"name":"Department of Epidemiology University of Texas MD Anderson Cancer Center  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3285-4281","authenticated-orcid":false,"given":"Ann","family":"Nordgren","sequence":"additional","affiliation":[{"name":"Department of Molecular Medicine and Surgery Karolinska Institutet  Stockholm Sweden"},{"name":"Department of Clinical Genetics and Genomics Sahlgrenska University Hospital  Gothenburg Sweden"},{"name":"Department of Laboratory Medicine Institute of Biomedicine, Sahlgrenska Academy, University of Gothenburg  Gothenburg Sweden"},{"name":"Clinical Genetics and Genomics, Karolinska University Hospital  Solna Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0405-8133","authenticated-orcid":false,"given":"Barbara","family":"Luke","sequence":"additional","affiliation":[{"name":"Department of Obstetrics, Gynecology and Reproductive Biology College of Human Medicine, Michigan State University  East Lansing Michigan USA"},{"name":"Population, Policy, and Practice Research and Teaching Department Great Ormond Street Institute of Child Health, University College London  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Philip J.","family":"Lupo","sequence":"additional","affiliation":[{"name":"Division of Hematology\/Oncology, Department of Pediatrics Baylor College of Medicine  Houston Texas USA"},{"name":"Department of Pediatrics School of Medicine, Emory University  Atlanta Georgia USA"},{"name":"Aflac Cancer and Blood Disorders Center at Children's Healthcare of Atlanta  Atlanta Georgia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6674-9562","authenticated-orcid":false,"given":"Jeremy M.","family":"Schraw","sequence":"additional","affiliation":[{"name":"Division of Hematology\/Oncology, Department of Pediatrics Baylor College of Medicine  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,12,23]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.11046"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.beha.2014.10.002"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.37637"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.12688\/f1000research.13213.1"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-018-04540-x"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1097\/MCD.0b013e328358243c"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2005.10.087"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19970711)71:1<8::AID-AJMG2>3.0.CO;2-V"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2025-265123"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.3390\/jcm11175056"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10552-016-0807-5"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00441773"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.2147\/CLEP.S147188"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.20327"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30853"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2024.07.005"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(99)05264-2"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41416-022-01961-w"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1201"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.71.5.815"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamanetworkopen.2020.22927"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10815-021-02095-3"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1093\/humrep\/deaa272"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoncol.2019.1215"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1001\/jama.2007.66"},{"key":"e_1_2_9_27_1","volume-title":"Certification Criteria","author":"North American Association of Central Cancer Registries (NAACCR)","year":"2021"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0179006"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1203\/pdr.0b013e318039d978"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(73)80024-1"},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19960628)63:4<529::AID-AJMG4>3.0.CO;2-J"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1038\/ki.2013.417"},{"key":"e_1_2_9_33_1","doi-asserted-by":"publisher","DOI":"10.1002\/cncr.32982"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.canep.2025.102748"},{"key":"e_1_2_9_35_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1616361"},{"key":"e_1_2_9_36_1","doi-asserted-by":"publisher","DOI":"10.1159\/000346301"},{"key":"e_1_2_9_37_1","doi-asserted-by":"publisher","DOI":"10.1159\/000345406"},{"key":"e_1_2_9_38_1","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-6-56"},{"key":"e_1_2_9_39_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31664"},{"key":"e_1_2_9_40_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.35638"},{"key":"e_1_2_9_41_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33572"},{"key":"e_1_2_9_42_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamapediatrics.2019.0392"},{"key":"e_1_2_9_43_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pcl.2014.09.013"},{"key":"e_1_2_9_44_1","doi-asserted-by":"publisher","DOI":"10.1159\/000346192"},{"key":"e_1_2_9_45_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.1773"},{"key":"e_1_2_9_46_1","doi-asserted-by":"publisher","DOI":"10.1002\/bdr2.1686"},{"key":"e_1_2_9_47_1","doi-asserted-by":"publisher","DOI":"10.1001\/archpedi.1986.02140170051027"},{"key":"e_1_2_9_48_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamanetworkopen.2022.30157"}],"container-title":["American Journal of Medical Genetics Part A"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmga.70031","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/ajmga.70031","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmga.70031","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,28]],"date-time":"2026-08-28T04:44:38Z","timestamp":1787892278000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmga.70031"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,23]]},"references-count":47,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2026,5]]}},"alternative-id":["10.1002\/ajmga.70031"],"URL":"https:\/\/doi.org\/10.1002\/ajmga.70031","archive":["Portico"],"relation":{},"ISSN":["1552-4825","1552-4833"],"issn-type":[{"value":"1552-4825","type":"print"},{"value":"1552-4833","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,23]]},"assertion":[{"value":"2025-10-02","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-12-08","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-12-23","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}