ArticlePloS one2013
Cancer risk in children and adolescents with birth defects: a population-based cohort study.
Article in PloS one, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 44 papers, 4 of them syntheses that pooled it.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
44 citing papers in PubMed, 4 syntheses or guidelines pooled it, 84 citations in OpenAlex.
- Risk of Cancer in Patients with Congenital Heart Disease: A Systematic Review and Meta-Analysis.Cardiology · 2025Pooled it
- Genes and microRNAs associated with mouse cleft palate: A systematic review and bioinformatics analysis.Mechanisms of development · 2018Pooled it
- Age-specific global epidemiology of hydrocephalus: Systematic review, metanalysis and global birth surveillance.PloS one · 2018Pooled it
- Pooled it
- Increased Risk of Sarcomas in Children With Congenital Anomalies: Findings From the Genetic Overlap Between Anomalies and Cancer in Kids (GOBACK) Registry Linkage Study.Pediatric blood & cancer · 2026Article
- Cancer Diagnoses in Patients With Cleft Lip and/or Palate: Increased Childhood Cancer Occurrence.The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association · 2026Article
- Risk of Childhood Cancer in Children with Congenital Anomalies and Their Impact on Survival: A Population-Based Registry Approach.Current oncology (Toronto, Ont.) · 2025Article
- Co-occurrence of congenital anomalies and childhood brain tumors in 22 million live births.Neuro-oncology · 2025Article
- Adult-onset medulloblastoma presenting in the setting of untreated spina bifida: illustrative case.Journal of neurosurgery. Case lessons · 2025Article
- Environmental risk factors of Wilms tumour: A systematic review and meta-analysis.EJC paediatric oncology · 2024Review
- Cancer risk in individuals with polydactyly: a Swedish population-based cohort study.British journal of cancer · 2024Article
- Article
- Role of non-chromosomal birth defects on the risk of developing childhood Hodgkin lymphoma: A Children's Oncology Group study.Pediatric blood & cancer · 2024Article
- Nonchromosomal birth defects and risk of childhood acute leukemia: An assessment in 15 000 leukemia cases and 46 000 controls from the Childhood Cancer and Leukemia International Consortium.International journal of cancer · 2024Article
- Surgically Correctable Congenital Anomalies: Reducing Morbidity and Mortality in the First 8000 Days of Life.World journal of surgery · 2023Review
- Associations between birth defects with neural crest cell origins and pediatric embryonal tumors.Cancer · 2023Article
- Article
- The genetic factors contributing to the risk of cleft lip-cleft palate and their clinical utility.Oral and maxillofacial surgery · 2023Review
- Identification of risk variants related to malignant tumors in children with birth defects by whole genome sequencing.Biomarker research · 2022Article
- The risks of birth defects and childhood cancer with conception by assisted reproductive technology.Human reproduction (Oxford, England) · 2022Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
10 authors at 4 institutions in 2 countries.
Funding
Abstract
objectiveBirth 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
findingsThis 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.
conclusionChildren 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.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.