Association Between Birth Defects and Cancer Risk Among Children and Adolescents in a Population-Based Assessment of 10 Million Live Births
Association Between Birth Defects and Cancer Risk Among Children and Adolescents in a Population-Based Assessment of 10 Million Live Births
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DOI：
10.1001/jamaoncol.2019.1215
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发表时间：
2019-08-01
期刊：
影响因子：
28.4
通讯作者：
Plon, Sharon E.
中科院分区：
文献类型：
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作者：
Lupo, Philip J.;Schraw, Jeremy M.;Plon, Sharon E.
出生缺陷影响大约1/33的儿童。已知一些出生缺陷与儿童癌症密切相关（例如，21三体和急性白血病）。然而，在以前的研究中，由于样本量不足，对出生缺陷儿童癌症风险的全面评估受到限制。目的通过增加非染色体出生缺陷的数量来确定特定的出生缺陷-儿童癌症（BD-CC）关联并表征儿童癌症风险。设计、设置和样本这项多州、基于人群的登记关联研究汇集了德克萨斯州、阿肯色州、密歇根州和北卡罗来纳州1992年1月1日至2013年12月31日出生的10181074名儿童的出生、出生缺陷和癌症的全州数据。儿童被随访至18岁，以诊断癌症。数据检索时间为2016年9月26日至2017年9月21日，数据分析时间为2017年9月2日至2019年3月21日。暴露出生缺陷诊断（染色体异常和非染色体出生缺陷）由全州，基于人口的出生缺陷登记处记录。主要结果和测量18岁之前的癌症诊断，如州癌症登记处所记录的。考克斯回归模型用于产生风险比（HR）和95% CI，以评估BD-CC相关性以及非染色体缺陷数量与癌症风险之间的相关性。结果18岁以前，染色体异常儿童患癌症的可能性是无任何出生缺陷儿童的11.6倍（95%CI，10.4-12.9），而非染色体出生缺陷儿童患癌症的可能性是无任何出生缺陷儿童的2.5倍（95%CI，2.4-2.6）。越来越多的非染色体出生缺陷与相应的癌症风险增加有关。与没有出生缺陷的儿童相比，有4个或更多重大出生缺陷的儿童被诊断患有癌症的可能性高5.9倍（95%CI，5.3-6.4）。在对72种BD-CC类型的分析中，40种HR具有统计学意义（校正P
ImportanceBirth defects affect approximately 1 in 33 children. Some birth defects are known to be strongly associated with childhood cancer (eg, trisomy 21 and acute leukemia). However, comprehensive evaluations of childhood cancer risk in those with birth defects have been limited in previous studies by insufficient sample sizes. ObjectivesTo identify specific birth defect-childhood cancer (BD-CC) associations and characterize cancer risk in children by increasing number of nonchromosomal birth defects. Design, Setting, and ParticipantsThis multistate, population-based registry linkage study pooled statewide data on births, birth defects, and cancer from Texas, Arkansas, Michigan, and North Carolina on 10181074 children born from January 1, 1992, to December 31, 2013. Children were followed up to 18 years of age for a diagnosis of cancer. Data were retrieved between September 26, 2016, and September 21, 2017, and data analysis was performed from September 2, 2017, to March 21, 2019. ExposuresBirth defects diagnoses (chromosomal anomalies and nonchromosomal birth defects) recorded by statewide, population-based birth defects registries. Main Outcomes and MeasuresCancer diagnosis before age 18 years, as recorded in state cancer registries. Cox regression models were used to generate hazard ratios (HRs) and 95% CIs to evaluate BD-CC associations and the association between number of nonchromosomal defects and cancer risk. ResultsCompared with children without any birth defects, children with chromosomal anomalies were 11.6 (95% CI, 10.4-12.9) times more likely to be diagnosed with cancer, whereas children with nonchromosomal birth defects were 2.5 (95% CI, 2.4-2.6) times more likely to be diagnosed with cancer before 18 years of age. An increasing number of nonchromosomal birth defects was associated with a corresponding increase in the risk of cancer. Children with 4 or more major birth defects were 5.9 (95% CI, 5.3-6.4) times more likely to be diagnosed with cancer compared with those without a birth defect. In the analysis of 72 specific BD-CC patterns, 40 HRs were statistically significant (adjusted P