Cancer diagnostic profile in children with structural birth defects: An assessment in 15,000 childhood cancer cases
- 29 May 2020
- Vol. 126 (15), 3483-3492
- https://doi.org/10.1002/cncr.32982
Abstract
Background Birth defects are established risk factors for childhood cancer. Nonetheless, cancer epidemiology in children with birth defects is not well characterized. Methods Using data from population‐based registries in 4 US states, this study compared children with cancer but no birth defects (n = 13,111) with children with cancer and 1 or more nonsyndromic birth defects (n = 1616). The objective was to evaluate cancer diagnostic characteristics, including tumor type, age at diagnosis, and stage at diagnosis. Results Compared with the general population of children with cancer, children with birth defects were diagnosed with more embryonal tumors (26.6% vs 18.7%; q < 0.001), including neuroblastoma (12.5% vs 8.2%; q < 0.001) and hepatoblastoma (5.0% vs 1.3%; q < 0.001), but fewer hematologic malignancies, including acute lymphoblastic leukemia (12.4% vs 24.4%; q < 0.001). In age‐stratified analyses, differences in tumor type were evident among children younger than 1 year and children 1 to 4 years old, but they were attenuated among children 5 years of age or older. The age at diagnosis was younger in children with birth defects for most cancers, including leukemia, lymphoma, astrocytoma, medulloblastoma, ependymoma, embryonal tumors, and germ cell tumors (all q < 0.05). Conclusions The results indicate possible etiologic heterogeneity in children with birth defects, have implications for future surveillance efforts, and raise the possibility of differential cancer ascertainment in children with birth defects. Lay Summary Scientific studies suggest that children with birth defects are at increased risk for cancer. However, these studies have not been able to determine whether important tumor characteristics, such as the type of tumor diagnosed, the age at which the tumor is diagnosed, and the degree to which the tumor has spread at the time of diagnosis, are different for children with birth defects and children without birth defects. This study attempts to answer these important questions. By doing so, it may help scientists and physicians to understand the causes of cancer in children with birth defects and diagnose cancer at earlier stages when it is more treatable.Keywords
Funding Information
- Alex's Lemonade Stand Foundation for Childhood Cancer (Epidemiology Award)
- Cancer Prevention and Research Institute of Texas (RP140258, RP160097, RP170071)
- National Cancer Institute (3P30CA125123‐0851)
This publication has 41 references indexed in Scilit:
- Cancer Risk in Children and Adolescents with Birth Defects: A Population-Based Cohort StudyPLOS ONE, 2013
- Managing the Morbidity Associated with Respiratory Viral Infections in Children with Congenital Heart DiseaseInternational Journal of Pediatrics, 2012
- Molecular subgroups of medulloblastoma: the current consensusActa Neuropathologica, 2011
- What Can We Learn about Disease Etiology from Case-Case Analyses? Lessons from Breast CancerCancer Epidemiology, Biomarkers & Prevention, 2010
- Updated national birth prevalence estimates for selected birth defects in the United States, 2004–2006Birth Defects Research Part A: Clinical and Molecular Teratology, 2010
- Cancer Risk in Children with Birth Defects and in Their Families: A Population Based Cohort Study of 5.2 Million Children from Norway and SwedenCancer Epidemiology, Biomarkers & Prevention, 2008
- National estimates and race/ethnic‐specific variation of selected birth defects in the United States, 1999–2001Birth Defects Research Part A: Clinical and Molecular Teratology, 2006
- Congenital abnormalities and childhood cancerCancer, 2005
- A remark on algorithm 643: FEXACTACM Transactions on Mathematical Software, 1993
- A Network Algorithm for Performing Fisher's Exact Test in r × c Contingency TablesJournal of the American Statistical Association, 1983