Dataset Information

Cancer risk in individuals with major birth defects: large Nordic population based case-control study among children, adolescents, and adults.

ABSTRACT: Objective: To examine associations between birth defects and cancer from birth into adulthood.

Design: Population based nested case-control study.

Setting: Nationwide health registries in Denmark, Finland, Norway, and Sweden.

Participants: 62?295 cancer cases (0-46 years) and 724?542 frequency matched controls (matched on country and birth year), born between 1967 and 2014.

Main outcome measures: Relative risk of cancer in relation to major birth defects, estimated as odds ratios with 99% confidence intervals from logistic regression models.

Results: Altogether, 3.5% (2160/62?295) of cases and 2.2% (15?826/724?542) of controls were born with major birth defects. The odds ratio of cancer for people with major birth defects compared with those without was 1.74 (99% confidence interval 1.63 to 1.84). For individuals with non-chromosomal birth defects, the odds ratio of cancer was 1.54 (1.44 to 1.64); for those with chromosomal anomalies, the odds ratio was 5.53 (4.67 to 6.54). Many structural birth defects were associated with later cancer in the same organ system or anatomical location, such as defects of the eye, nervous system, and urinary organs. The odds ratio of cancer increased with number of defects and decreased with age, for both non-chromosomal and chromosomal anomalies. The odds ratio of cancer in people with any non-chromosomal birth defect was lower in adults (?20 years: 1.21, 1.09 to 1.33) than in adolescents (15-19 years: 1.58, 1.31 to 1.90) and children (0-14 years: 2.03, 1.85 to 2.23). The relative overall cancer risk among adults with chromosomal anomalies was markedly reduced from 11.3 (9.35 to 13.8) in children to 1.50 (1.01 to 2.24). Among adults, skeletal dysplasia (odds ratio 3.54, 1.54 to 8.15), nervous system defects (1.76, 1.16 to 2.65), chromosomal anomalies (1.50, 1.01 to 2.24), genital organs defects (1.43, 1.14 to 1.78), and congenital heart defects (1.28, 1.02 to 1.59) were associated with overall cancer risk.

Conclusions: The increased risk of cancer in individuals with birth defects persisted into adulthood, both for non-chromosomal and chromosomal anomalies. Further studies on the molecular mechanisms involved are warranted.

SUBMITTER: Daltveit DS

PROVIDER: S-EPMC7708828 | biostudies-literature | 2020 Dec

REPOSITORIES: biostudies-literature

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Cancer risk in individuals with major birth defects: large Nordic population based case-control study among children, adolescents, and adults.

Daltveit Dagrun Slettebø DS Klungsøyr Kari K Engeland Anders A Ekbom Anders A Gissler Mika M Glimelius Ingrid I Grotmol Tom T Madanat-Harjuoja Laura L Ording Anne Gulbech AG Sæther Solbjørg Makalani Myrtveit SMM Sørensen Henrik Toft HT Troisi Rebecca R Bjørge Tone T

BMJ (Clinical research ed.) 20201202

<h4>Objective</h4>To examine associations between birth defects and cancer from birth into adulthood.<h4>Design</h4>Population based nested case-control study.<h4>Setting</h4>Nationwide health registries in Denmark, Finland, Norway, and Sweden.<h4>Participants</h4>62 295 cancer cases (0-46 years) and 724 542 frequency matched controls (matched on country and birth year), born between 1967 and 2014.<h4>Main outcome measures</h4>Relative risk of cancer in relation to major birth defects, estimated ...[more]

PMID: 33268348

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Project description:Background: Although major external structural birth defects continue to occur globally, the greatest burden is shouldered by resource-constrained countries with no surveillance systems. To our knowledge, many studies have been published on risk factors for major external structural birth defects, however, limited studies have been published in developing countries. The objective of this study was to identify risk factors for major external structural birth defects among children in Kiambu County, Kenya. Methods: A hospital-based case-control study was used to identify the risk factors for major external structural birth defects. A structured questionnaire was used to gather information retrospectively on maternal exposure to environmental teratogens, multifactorial inheritance, and sociodemographic-environmental factors during the study participants' last pregnancies. Descriptive analyses (means, standard deviations, medians, and ranges) were used to summarize continuous variables, whereas categorical variables were summarized as proportions and percentages in frequency tables. Afterward, logistic regression analyses were conducted to estimate the effects of the predictors on the odds of major external structural birth defects in the country. Results: Women who conceived when residing in Ruiru sub-county (adjusted odds ratio [aOR]: 5.28; 95% CI: 1.68-16.58; P<0.01), and Thika sub-county (aOR: 0.27; 95% CI; 0.076-0.95; P =0.04); and preceding siblings with history of birth defects (aOR: 7.65; 95% CI; 1.46-40.01; P =0.02) were identified as the significant predictors of major external structural birth defects in the county. Conclusions: These findings pointed to MESBDs of genetic, multifactorial inheritance, and sociodemographic-environmental etiology. Thus, we recommend regional defect-specific surveillance programs, public health preventive measures, and treatment strategies to understand the epidemiology and economic burden of these defects in Kenya. We specifically recommend the integration of clinical genetic services with routine reproductive health services because of potential maternal genetic predisposition in the region.

Dataset Information

Cancer risk in individuals with major birth defects: large Nordic population based case-control study among children, adolescents, and adults.

Publications

Cancer risk in individuals with major birth defects: large Nordic population based case-control study among children, adolescents, and adults.

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