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

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

doi:10.1371/journal.pone.0069077

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

Journal article

Open access

Peer reviewed

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

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

PloS one, Vol.8(7), pp.e69077-e69077

2013

DOI: 10.1371/journal.pone.0069077

PMCID: PMC3714243

PMID: 23874873

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Abstract

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. 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. 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.

Neoplasms - complications

Congenital Abnormalities - epidemiology

Congenital Abnormalities - genetics

Neoplasms - genetics

Humans

Risk Factors

Adolescent

Chromosome Aberrations

Child

Population Surveillance

Neoplasms - epidemiology

Cohort Studies

Details

Title: Subtitle: Cancer risk in children and adolescents with birth defects: a population-based cohort study
Creators: Lorenzo D Botto - Division of Medical Genetics, Department of Pediatrics, University of Utah, Salt Lake City, Utah, USA. Lorenzo.botto@hsc.utah.edu
Timothy Flood
Julian Little
Mark N Fluchel
Sergey Krikov
Marcia L Feldkamp
Yuan Wu - University of Iowa
Rhinda Goedken
Soman Puzhankara
Paul A Romitti
Resource Type: Journal article
Publication Details: PloS one, Vol.8(7), pp.e69077-e69077
DOI: 10.1371/journal.pone.0069077
PMID: 23874873
PMCID: PMC3714243
NLM abbreviation: PLoS One
ISSN: 1932-6203
eISSN: 1932-6203
Publisher: United States
Grant note: P30 ES005605 / NIEHS NIH HHS HHSN261201000026C / PHS HHS MM-1079-09/09 / PHS HHS
Language: English
Date published: 2013
Academic Unit: Epidemiology; Biostatistics
Record Identifier: 9983995162802771

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