Jeremy M. Schraw , Ji Yun Tark , Tania A. Desrosiers , Tiffany M. Chambers , Charles J. Shumate , Wendy N. Nembhard , Mahsa M. Yazdy , Eirini Nestoridi , Mary Frances Wedekind Malone , Theodore W. Laetsch , Brigitte C. Widemann , Amanda E. Janitz , Jean Paul Tanner , Russell S. Kirby , Jason L. Salemi , Logan G. Spector , Chad D. Huff , Sharon E. Plon , Philip J. Lupo
{"title":"Risk of carcinomas among children and adolescents with birth defects","authors":"Jeremy M. Schraw , Ji Yun Tark , Tania A. Desrosiers , Tiffany M. Chambers , Charles J. Shumate , Wendy N. Nembhard , Mahsa M. Yazdy , Eirini Nestoridi , Mary Frances Wedekind Malone , Theodore W. Laetsch , Brigitte C. Widemann , Amanda E. Janitz , Jean Paul Tanner , Russell S. Kirby , Jason L. Salemi , Logan G. Spector , Chad D. Huff , Sharon E. Plon , Philip J. Lupo","doi":"10.1016/j.canep.2025.102748","DOIUrl":null,"url":null,"abstract":"<div><h3>Background</h3><div>Birth defects are associated with childhood cancer, but little is known regarding pediatric carcinomas, a group of especially rare tumors.</div></div><div><h3>Methods</h3><div>We used Cox proportional hazards regression to estimate the hazard ratio (HR) and 95 % confidence interval (CI) for any carcinoma, as well as thyroid, hepatocellular, and renal carcinoma specifically, up to 18 years of age among children with major, non-syndromic anomalies or chromosomal/genetic syndromes, relative to unaffected children.</div></div><div><h3>Results</h3><div>Our registry-linkage study included nine states and 21,933,476 children between 1990 and 2018: 641,827 with non-syndromic anomalies, and 49,619 with syndromes. Carcinomas were diagnosed in 833 children, including 35 with non-syndromic anomalies and eight with syndromes. The hazard of carcinoma was increased both among children with non-syndromic anomalies (HR: 1.7, CI: 1.2–2.4; N = 35) and syndromes (HR: 4.7, CI: 2.3–9.5; N = 7). Hepatocellular carcinoma was associated with non-syndromic anomalies (HR: 4.6, CI: 2.2–9.7; N = 8) and syndromes (HR: 8.0, CI: 1.1–58.1; N < 5). The hazard of renal carcinoma was markedly increased in children with tuberous sclerosis (HR 59.6, CI: 23.7–149.5; N = 5), a known cause of renal cancer. Thyroid carcinoma was not associated with non-syndromic anomalies or syndromes.</div></div><div><h3>Conclusion</h3><div>Birth defects are associated with hepatocellular and renal carcinoma in children.</div></div>","PeriodicalId":56322,"journal":{"name":"Cancer Epidemiology","volume":"95 ","pages":"Article 102748"},"PeriodicalIF":2.4000,"publicationDate":"2025-01-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Cancer Epidemiology","FirstCategoryId":"3","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S1877782125000074","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"ONCOLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Background
Birth defects are associated with childhood cancer, but little is known regarding pediatric carcinomas, a group of especially rare tumors.
Methods
We used Cox proportional hazards regression to estimate the hazard ratio (HR) and 95 % confidence interval (CI) for any carcinoma, as well as thyroid, hepatocellular, and renal carcinoma specifically, up to 18 years of age among children with major, non-syndromic anomalies or chromosomal/genetic syndromes, relative to unaffected children.
Results
Our registry-linkage study included nine states and 21,933,476 children between 1990 and 2018: 641,827 with non-syndromic anomalies, and 49,619 with syndromes. Carcinomas were diagnosed in 833 children, including 35 with non-syndromic anomalies and eight with syndromes. The hazard of carcinoma was increased both among children with non-syndromic anomalies (HR: 1.7, CI: 1.2–2.4; N = 35) and syndromes (HR: 4.7, CI: 2.3–9.5; N = 7). Hepatocellular carcinoma was associated with non-syndromic anomalies (HR: 4.6, CI: 2.2–9.7; N = 8) and syndromes (HR: 8.0, CI: 1.1–58.1; N < 5). The hazard of renal carcinoma was markedly increased in children with tuberous sclerosis (HR 59.6, CI: 23.7–149.5; N = 5), a known cause of renal cancer. Thyroid carcinoma was not associated with non-syndromic anomalies or syndromes.
Conclusion
Birth defects are associated with hepatocellular and renal carcinoma in children.
期刊介绍:
Cancer Epidemiology is dedicated to increasing understanding about cancer causes, prevention and control. The scope of the journal embraces all aspects of cancer epidemiology including:
• Descriptive epidemiology
• Studies of risk factors for disease initiation, development and prognosis
• Screening and early detection
• Prevention and control
• Methodological issues
The journal publishes original research articles (full length and short reports), systematic reviews and meta-analyses, editorials, commentaries and letters to the editor commenting on previously published research.