Cited by 857Genetic Associations and EpidemiologyGenetic Mapping and Diversity in Plants and AnimalsForensic and Genetic Research

Rare Variants Create Synthetic Genome-Wide Associations

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Dickson, Samuel P., Wang, Kai, Krantz, Ian, Hakonarson, Hakon, Goldstein, David B.·2010·PLoS Biology

Abstract

Genome-wide association studies (GWAS) have now identified at least 2,000 common variants that appear associated with common diseases or related traits (http://www.genome.gov/gwastudies), hundreds of which have been convincingly replicated. It is generally thought that the associated markers reflect the effect of a nearby common (minor allele frequency >0.05) causal site, which is associated with the marker, leading to extensive resequencing efforts to find causal sites. We propose as an alternative explanation that variants much less common than the associated one may create "synthetic associations" by occurring, stochastically, more often in association with one of the alleles at the common site versus the other allele. Although synthetic associations are an obvious theoretical possibility, they have never been systematically explored as a possible explanation for GWAS findings. Here, we use simple computer simulations to show the conditions under which such synthetic associations will arise and how they may be recognized. We show that they are not only possible, but inevitable, and that under simple but reasonable genetic models, they are likely to account for or contribute to many of the recently identified signals reported in genome-wide association studies. We also illustrate the behavior of synthetic associations in real datasets by showing that rare causal mutations responsible for both hearing loss and sickle cell anemia create genome-wide significant synthetic associations, in the latter case extending over a 2.5-Mb interval encompassing scores of "blocks" of associated variants. In conclusion, uncommon or rare genetic variants can easily create synthetic associations that are credited to common variants, and this possibility requires careful consideration in the interpretation and follow up of GWAS signals.

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  • The Procrustean theory that common variant signals inevitably reflect "synthetic associations" to rare, high penetrance mutations is not credible.

    “The Procrustean theory that common variant signals inevitably reflect “synthetic associations” 124 to rare, high penetrance mutations is not credible. 77,125–127”

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References 40 · Cited by 857

Authors
Dickson, S. P., Wang, K., Krantz, I., Hakonarson, H., & Goldstein, D. B.
Affiliations
  • North Carolina State University (US)
  • Children's Hospital of Philadelphia (US)
  • Duke University (US)
Corresponding author
Goldstein, David B.
Venue
PLoS Biology
Published
2010-01-26
Volume
8
Issue
1
Pages
e1000294
ISSN
  • 1544-9173 (ISSN-L)
  • 1545-7885
OpenAlex
W2059867286
Cited by
857
References
40
Citations by year
2026: 10 · 2025: 10 · 2024: 11 · 2023: 11 · 2022: 14 · 2021: 21 · 2020: 17 · 2019: 36 · 2018: 27 · 2017: 25 · 2016: 48 · 2015: 55 · 2014: 70 · 2013: 109 · 2012: 133
FWCI
90.03
Type
journal article
Language
en
Open access
Locations
  • PLoS Biology (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion)
  • DukeSpace (Duke University) (submittedVersion, cc-by-nc-nd)
Funders
Children's Hospital of Philadelphia
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic Associations and Epidemiology
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic Mapping and Diversity in Plants and Animals
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Forensic and Genetic Research
Keywords
rare variants, common genetic variants, genome-wide association study, causal variants, linkage disequilibrium, computer simulation, sickle cell disease, hearing loss
MeSH terms
Anemia, Sickle Cell, Genotype, Humans, Probability, Risk Factors, Genetic Predisposition to Disease, Polymorphism, Single Nucleotide, Hearing Loss, Genome-Wide Association Study

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