Spina bifida and pediatric cancers

Abstract

Spina bifida has been reported to co-occur with pediatric cancer, but comprehensive evaluations remained elusive. We investigated this co-occurrence in two large, population-based studies in Taiwan (N = 1900 cancer cases, 2,077,137 controls) and Denmark (N = 5508 cases, 137,700 controls). Analyses in Denmark were restricted to the period before prenatal diagnostics became available (2004) and pregnancy terminations of fetuses with birth defects became more common. Using national patient and cancer registries, we linked spina bifida and cancer diagnoses among cases and non-cases. The risk of spina bifida among all cancer cases was increased and similar in Denmark [odds ratio (OR)=8.4, 95% confidence interval (CI) 5.1-13.8] and Taiwan (OR = 8.5, 95% CI 4.0-17.8), particularly for central nervous system (CNS) tumors (Denmark: OR = 16.3, 95% CI 8.1-33.0; Taiwan: OR = 26.6, 95% CI 8.5, 83.1), including benign CNS tumors (Denmark: OR = 41.5, 95% CI 21.2, 81.4). These findings suggest the need for comprehensive investigation of shared risk factors in the link between spina bifida and pediatric cancer.

Keywords:

• Birth defects
• central nervous system tumors
• childhood cancer epidemiology
• folate
• spina bifida

Declaration of interests statement

The authors report no conflicts of interest.

Additional information

Funding

Authors would like to thank Drs. Negar Omidakhsh and Hsin-Yun Tsai for their help with the manuscript. The Danish study was supported by the US National Institutes of Health (R03ES021643, R21CA175959). The Taiwan study was supported by the Alex’s Lemonade Stand Foundation (grant 17-01882). Ms. He was supported by funds from the Tobacco-Related Disease Research Program of the University of California, Grant Number T29DT0485. Dr. Federman was supported by NIH/NCATS Grant # UL1TR001881.