Detecting gene-environment interactions in human birth defects: Study designs and statistical methods

Birth Defects Res A Clin Mol Teratol. 2015 Aug;103(8):692-702. doi: 10.1002/bdra.23382. Epub 2015 May 23.

Abstract

Background: The National Birth Defects Prevention Study (NBDPS) contains a wealth of information on affected and unaffected family triads, and thus provides numerous opportunities to study gene-environment interactions (G×E) in the etiology of birth defect outcomes. Depending on the research objective, several analytic options exist to estimate G×E effects that use varying combinations of individuals drawn from available triads.

Methods: In this study, we discuss important considerations in the collection of genetic data and environmental exposures.

Results: We will also present several population- and family-based approaches that can be applied to data from the NBDPS including case-control, case-only, family-based trio, and maternal versus fetal effects. For each, we describe the data requirements, applicable statistical methods, advantages, and disadvantages.

Conclusion: A range of approaches can be used to evaluate potentially important G×E effects in the NBDPS. Investigators should be aware of the limitations inherent to each approach when choosing a study design and interpreting results.

Keywords: National Birth Defects Prevention Study; birth defects; case-only; congenital heart defects; family-based study; gene-environment interaction; genetic epidemiology; neural tube defects; transmission disequilibrium test.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Case-Control Studies
  • Congenital Abnormalities / etiology*
  • Female
  • Gene-Environment Interaction*
  • Genetic Predisposition to Disease*
  • Humans
  • Male
  • Models, Statistical*
  • Pedigree
  • Polymorphism, Single Nucleotide
  • Research Design*
  • Risk Factors