Rare Variants Create Synthetic Genome-Wide Associations
Open accessAbstract
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.
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
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”
From Genetic architectures of psychiatric disorders: the emerging picture and its implications (2012) · cites this paper as [125]
Self-check
Percentages, sums and reported differences re-computed from the paper’s own numbers.
The self-check needs the paper’s full text.
The citation lists haven’t been fetched yet
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
- Publisher
- Public Library of Science (PLoS)
- Published
- 2010-01-26
- Volume
- 8
- Issue
- 1
- Pages
- e1000294
- ISSN
- 1544-9173 (ISSN-L)
- 1545-7885
- PMC
- PMC2811148
- 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
- gold
- doi.org
- 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
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review