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. 2013 Jul 17;8(7):e69077.
doi: 10.1371/journal.pone.0069077. Print 2013.

Cancer risk in children and adolescents with birth defects: a population-based cohort study

Lorenzo D Botto  1 , Timothy Flood, Julian Little, Mark N Fluchel, Sergey Krikov, Marcia L Feldkamp, Yuan Wu, Rhinda Goedken, Soman Puzhankara, Paul A Romitti
Affiliations

Cancer risk in children and adolescents with birth defects: a population-based cohort study

Lorenzo D Botto et al. PLoS One. .

Abstract

Objective: Birth defects are an increasing health priority worldwide, and the subject of a major 2010 World Health Assembly Resolution. Excess cancer risk may be an added burden in this vulnerable group of children, but studies to date have provided inconsistent findings. This study assessed the risk for cancer in children and young adolescents with major birth defects.

Methods and findings: This retrospective, statewide, population-based, cohort study was conducted in three US states (Utah, Arizona, Iowa). A cohort of 44,151 children and young adolescents (0 through 14 years of age) with selected major, non-chromosomal birth defects or chromosomal anomalies was compared to a reference cohort of 147,940 children without birth defects randomly sampled from each state's births and frequency matched by year of birth. The primary outcome was rate of cancer prior to age 15 years, by type of cancer and type of birth defect. The incidence of cancer was increased 2.9-fold (95% CI, 2.3 to 3.7) in children with birth defects (123 cases of cancer) compared to the reference cohort; the incidence rates were 33.8 and 11.7 per 100,000 person-years, respectively. However, the excess risk varied markedly by type of birth defect. Increased risks were seen in children with microcephaly, cleft palate, and selected eye, cardiac, and renal defects. Cancer risk was not increased with many common birth defects, including hypospadias, cleft lip with or without cleft palate, or hydrocephalus.

Conclusion: Children with some structural, non-chromosomal birth defects, but not others, have a moderately increased risk for childhood cancer. Information on such selective risk can promote more effective clinical evaluation, counseling, and research.

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Conflict of interest statement

Competing Interests: The authors have declared that no competing interests exist.

Figures

Figure 1
Figure 1. Inclusions and exclusions leading to final cohorts in the UTAZIA study of birth defects and cancer.
Figure 2
Figure 2. Cancer-free time curves of reference cohort vs. children with chromosomal anomalies (top panel) and children with non-chromosomal birth defects (middle panel), together with hazard rate for cancer by age in reference cohort vs. children with non-chromosomal birth defects (lower panel).
Figure 3
Figure 3. Cumulative incidence of cancer prior to age 15 years in children with non-chromosomal birth defects and with chromosomal anomalies, UTAZIA study.
Cumulative incidence is expressed as a proportion (0.01 = 1 percent).
See this image and copyright information in PMC

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