{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,23]],"date-time":"2026-04-23T00:56:26Z","timestamp":1776905786678,"version":"3.51.2"},"reference-count":45,"publisher":"Wiley","issue":"22","license":[{"start":{"date-parts":[[2024,7,11]],"date-time":"2024-07-11T00:00:00Z","timestamp":1720656000000},"content-version":"am","delay-in-days":366,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#am"},{"start":{"date-parts":[[2023,7,11]],"date-time":"2023-07-11T00:00:00Z","timestamp":1689033600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100010210","name":"Defense Human Resources Activity","doi-asserted-by":"publisher","award":["W81XWH\u201020\u20101\u20100567"],"award-info":[{"award-number":["W81XWH\u201020\u20101\u20100567"]}],"id":[{"id":"10.13039\/100010210","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["acsjournals.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Cancer"],"published-print":{"date-parts":[[2023,11,15]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>There are few assessments evaluating associations between birth defects with neural crest cell developmental origins (BDNCOs) and embryonal tumors, which are characterized by undifferentiated cells having a molecular profile similar to neural crest cells. The effect of BDNCOs on embryonal tumors was estimated to explore potential shared etiologic pathways and genetic origins.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>With the use of a multistate, registry\u2010linkage cohort study, BDNCO\u2013embryonal tumor associations were evaluated by generating hazard ratios (HRs) and 95% confidence intervals (CIs) with Cox regression models. BDNCOs consisted of ear, face, and neck defects, Hirschsprung disease, and a selection of congenital heart defects. Embryonal tumors included neuroblastoma, nephroblastoma, and hepatoblastoma. Potential HR modification (HRM) was investigated by infant sex, maternal race\/ethnicity, maternal age, and maternal education.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The risk of embryonal tumors among those with BDNCOs was 0.09% (co\u2010occurring <jats:italic>n<\/jats:italic>\u00a0=\u00a0105) compared to 0.03% (95% CI, 0.03%\u20130.04%) among those without a birth defect. Children with BDNCOs were 4.2 times (95% CI, 3.5\u20135.1 times) as likely to be diagnosed with an embryonal tumor compared to children born without a birth defect. BDNCOs were strongly associated with hepatoblastoma (HR, 16.1; 95% CI, 11.3\u201322.9), and the HRs for neuroblastoma (3.1; 95% CI, 2.3\u20134.2) and nephroblastoma (2.9; 95% CI, 1.9\u20134.4) were elevated. There was no notable HRM by the aforementioned factors.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Children with BDNCOs are more likely to develop embryonal tumors compared to children without a birth defect. Disruptions of shared developmental pathways may contribute to both phenotypes, which could inform future genomic assessments and cancer surveillance strategies of these conditions.<\/jats:p><\/jats:sec>","DOI":"10.1002\/cncr.34952","type":"journal-article","created":{"date-parts":[[2023,7,11]],"date-time":"2023-07-11T09:46:50Z","timestamp":1689068810000},"page":"3595-3602","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Associations between birth defects with neural crest cell origins and pediatric embryonal tumors"],"prefix":"10.1002","volume":"129","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0044-4336","authenticated-orcid":false,"given":"Eugene C.","family":"Wong","sequence":"first","affiliation":[{"name":"Department of Epidemiology Gillings School of Global Public Health The University of North Carolina at Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0978-5863","authenticated-orcid":false,"given":"Philip J.","family":"Lupo","sequence":"additional","affiliation":[{"name":"Section of Hematology\u2010Oncology Department of Pediatrics Baylor College of Medicine  Houston Texas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tania A.","family":"Desrosiers","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Gillings School of Global Public Health The University of North Carolina at Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hazel B.","family":"Nichols","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Gillings School of Global Public Health The University of North Carolina at Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Susan M.","family":"Smith","sequence":"additional","affiliation":[{"name":"Department of Nutrition Gillings School of Global Public Health The University of North Carolina at Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Charles","family":"Poole","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Gillings School of Global Public Health The University of North Carolina at Chapel Hill  Chapel Hill North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mark","family":"Canfield","sequence":"additional","affiliation":[{"name":"Birth Defects Epidemiology and Surveillance Branch Texas Department of State Health Services  Austin Texas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Charles","family":"Shumate","sequence":"additional","affiliation":[{"name":"Birth Defects Epidemiology and Surveillance Branch Texas Department of State Health Services  Austin Texas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tiffany M.","family":"Chambers","sequence":"additional","affiliation":[{"name":"Section of Hematology\u2010Oncology Department of Pediatrics Baylor College of Medicine  Houston Texas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6674-9562","authenticated-orcid":false,"given":"Jeremy M.","family":"Schraw","sequence":"additional","affiliation":[{"name":"Section of Hematology\u2010Oncology Department of Pediatrics Baylor College of Medicine  Houston Texas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wendy N.","family":"Nembhard","sequence":"additional","affiliation":[{"name":"Department of Epidemiology Fay W. Boozman College of Public Health University of Arkansas for Medical Sciences and Arkansas Center for Birth Defects Research and Prevention  Little Rock Arkansas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mahsa M.","family":"Yazdy","sequence":"additional","affiliation":[{"name":"Massachusetts Center for Birth Defects Research and Prevention Massachusetts Department of Public Health  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eirini","family":"Nestoridi","sequence":"additional","affiliation":[{"name":"Massachusetts Center for Birth Defects Research and Prevention Massachusetts Department of Public Health  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amanda E.","family":"Janitz","sequence":"additional","affiliation":[{"name":"Department of Biostatistics and Epidemiology Hudson College of Public Health University of Oklahoma Health Sciences Center  Oklahoma City Oklahoma 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