Implications of small effect sizes of individual genetic variants on the design and interpretation of genetic association studies of complex diseases.

Accumulated evidence from searching for candidate gene-disease associations of complex diseases can offer some insights as the field moves toward discovery-oriented approaches with massive genome-wide testing. Meta-analyses of 50 non-human lymphocyte antigen gene-disease associations with documented overall statistical significance (752 studies) show summary odds ratios with a median of 1.43 (interquartile range, 1.28-1.65). Many different biases may operate in this field, for both single studies and meta-analyses, and these biases could invalidate some of these seemingly "validated" associations. Studies with a sample size of >500 show a median odds ratio of only 1.15. The median sample size required to detect the observed summary effects in each population addressed in the 752 studies is estimated to be 3,535 (interquartile range, 1,936-9,119 for cases and controls combined). These estimates are steeply inflated in the presence of modest bias. Population heterogeneity, as well as gene-gene and gene-environment interactions, could steeply increase these estimates and may be difficult to address even by very large biobanks and observational cohorts. The one visible solution is for a large number of teams to join forces on the same research platforms. These collaborative studies ideally should be designed up front to also assess more complex gene-gene and gene-environment interactions.

[1]  Paolo Vineis,et al.  A network of investigator networks in human genome epidemiology. , 2005, American journal of epidemiology.

[2]  Tim Sprosen,et al.  UK Biobank: from concept to reality. , 2005, Pharmacogenomics.

[3]  P. Donnelly,et al.  Genome-wide strategies for detecting multiple loci that influence complex diseases , 2005, Nature Genetics.

[4]  M. Khoury,et al.  How many genes underlie the occurrence of common complex diseases in the population? , 2005, International journal of epidemiology.

[5]  N. Laird,et al.  Meta-analysis in clinical trials. , 1986, Controlled clinical trials.

[6]  Thomas A Trikalinos,et al.  'Racial' differences in genetic effects for complex diseases , 2004, Nature Genetics.

[7]  J. Haines,et al.  Effects of age, sex, and ethnicity on the association between apolipoprotein E genotype and Alzheimer disease. A meta-analysis. APOE and Alzheimer Disease Meta Analysis Consortium. , 1997, JAMA.

[8]  Timothy R. Rebbeck,et al.  Assessing the function of genetic variants in candidate gene association studies , 2004, Nature Reviews Genetics.

[9]  Thomas A Trikalinos,et al.  Genetic associations in large versus small studies: an empirical assessment , 2003, The Lancet.

[10]  D. Hunter Gene–environment interactions in human diseases , 2005, Nature Reviews Genetics.

[11]  Francis S. Collins,et al.  The case for a US prospective cohort study of genes and environment , 2004, Nature.

[12]  E. Lander,et al.  Meta-analysis of genetic association studies supports a contribution of common variants to susceptibility to common disease , 2003, Nature Genetics.

[13]  D. Clayton,et al.  Genetic association studies , 2005, The Lancet.

[14]  Lyle J Palmer,et al.  Genetic Epidemiology 4 Shaking the tree : mapping complex disease genes with linkage disequilibrium , 2022 .

[15]  W. Gauderman Sample size requirements for association studies of gene-gene interaction. , 2002, American journal of epidemiology.

[16]  Paolo Vineis,et al.  A road map for efficient and reliable human genome epidemiology , 2006, Nature Genetics.

[17]  Nathaniel Rothman,et al.  Assessing the probability that a positive report is false: an approach for molecular epidemiology studies. , 2004, Journal of the National Cancer Institute.

[18]  Muin J Khoury,et al.  The human genome project is complete. How do we develop a handle for the pump? , 2003, American journal of epidemiology.

[19]  John P A Ioannidis,et al.  Genetic associations: false or true? , 2003, Trends in molecular medicine.

[20]  N Risch,et al.  The Future of Genetic Studies of Complex Human Diseases , 1996, Science.

[21]  T. Beaty,et al.  Detection of genotype-environment interaction in case-control studies of birth defects: how big a sample size? , 1995, Teratology.

[22]  J. Danesh,et al.  Four paraoxonase gene polymorphisms in 11 212 cases of coronary heart disease and 12 786 controls: meta-analysis of 43 studies , 2004, The Lancet.

[23]  N. Holtzman,et al.  Will genetics revolutionize medicine? , 2000, The New England journal of medicine.

[24]  M. Khoury,et al.  An epidemiologic approach to ecogenetics. , 1988, American journal of human genetics.

[25]  Margaret A. Pericak-Vance,et al.  Effects of age, sex, and ethnicity on the association between apolipoprotein E genotype and Alzheimer disease , 1997 .

[26]  J. Ioannidis,et al.  Local Literature Bias in Genetic Epidemiology: An Empirical Evaluation of the Chinese Literature , 2005, PLoS medicine.

[27]  Muin J Khoury,et al.  Improving the prediction of complex diseases by testing for multiple disease-susceptibility genes. , 2003, American journal of human genetics.

[28]  Sarah Parish,et al.  Large-scale test of hypothesised associations between the angiotensin-converting-enzyme insertion/deletion polymorphism and myocardial infarction in about 5000 cases and 6000 controls , 1999, The Lancet.

[29]  J. Brooks Why most published research findings are false: Ioannidis JP, Department of Hygiene and Epidemiology, University of Ioannina School of Medicine, Ioannina, Greece , 2008 .

[30]  M. Bamshad,et al.  Genetic influences on health: does race matter? , 2005, JAMA.

[31]  J. Ioannidis,et al.  Replication validity of genetic association studies , 2001, Nature Genetics.

[32]  P. McKeigue,et al.  Problems of reporting genetic associations with complex outcomes , 2003, The Lancet.

[33]  M. García-Closas,et al.  Misclassification in case-control studies of gene-environment interactions: assessment of bias and sample size. , 1999, Cancer epidemiology, biomarkers & prevention : a publication of the American Association for Cancer Research, cosponsored by the American Society of Preventive Oncology.