Cited by 186Human-Animal Interaction StudiesWilliams Syndrome ResearchGenetic and phenotypic traits in livestock

Structural variants in genes associated with human Williams-Beuren syndrome underlie stereotypical hypersociability in domestic dogs

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vonHoldt, Bridgett M., Shuldiner, Emily, Koch, Ilana Janowitz, Kartzinel, Rebecca Y., Hogan, Andrew, Brubaker, Lauren, Wanser, Shelby, Stahler, Daniel +4 more·2017·Science Advances

Abstract

, genes previously implicated in the behavioral phenotype of patients with WBS and contained within the WBS locus, contribute to extreme sociability in dogs. This finding suggests that there are commonalities in the genetic architecture of WBS and canine tameness and that directional selection may have targeted a unique set of linked behavioral genes of large phenotypic effect, allowing for rapid behavioral divergence of dogs and wolves, facilitating coexistence with humans.

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  • Among the genes under selection in both AMH and one or more domesticates, as well as in those under selection in multiple domesticates though not AMH, one finds multiple strong candidates for neurodevelopmental diseases and syndromes.

    “In a similar vein, among the genes under selection in both AMH and one or more domesticates, as well as in those under selection in multiple domesticates though not AMH, one finds multiple strong candidates for neurodevelopmental diseases and syndromes (see also [128]).”
  • Structural variants in WBS genes, for example in GTF2I and its paralogs, underlie stereotypical hypersociability in domestic dogs and foxes.

    “Structural variants in WBS genes, for example in the case of GTF2I and its paralogs, have been shown to underlie stereotypical hypersociability in domestic dogs and foxes (8, 9).”

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Research works printed but not linked: 1

  1. [40]American Kennel Club, The New Complete Dog Book: Official Breed Standards and All-New Profiles for 200 Breeds (21st Edition) (Lumina Media, 2014).

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Authors
vonHoldt, B. M., Shuldiner, E., Koch, I. J., Kartzinel, R. Y., Hogan, A., Brubaker, L., Wanser, S., Stahler, D., Wynne, C. D. L., Ostrander, E. A., Sinsheimer, J. S., & Udell, M. A. R.
Affiliations
  • Princeton University (US)
  • National Institutes of Health (US)
  • Oregon State University (US)
  • National Park Service (US)
  • Arizona State University (US)
  • University of California, Los Angeles (US)
Corresponding author
vonHoldt, Bridgett M.
Venue
Science Advances
Published
2017-07-07
Volume
3
Issue
7
Pages
e1700398-e1700398
ISSN
2375-2548 (ISSN-L)
OpenAlex
W2739139079
Cited by
186
References
97
Citations by year
2026: 11 · 2025: 18 · 2024: 17 · 2023: 15 · 2022: 28 · 2021: 27 · 2020: 21 · 2019: 18 · 2018: 27 · 2017: 4
FWCI
12.67
Type
journal article
Language
en
Open access
Locations
  • Science Advances (publishedVersion)
  • PubMed Central (publishedVersion, cc-by-nc)
  • PubMed (publishedVersion)
  • Arizona State University Library Digital Repository (Arizona State University) (submittedVersion, cc-by-nc-nd)
  • eScholarship (California Digital Library) (submittedVersion)
Funders
  • National Institutes of Health (award321887, GM086887)
  • National Science Foundation (award321886, DEB-1245373, award321888, DMS 1264153)
  • Princeton University (award321885, EEB, Dean of the College, Council on Science and Technology)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Human-Animal Interaction Studies
  • Life Sciences › Neuroscience › Developmental Neuroscience › Williams Syndrome Research
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic and phenotypic traits in livestock
Keywords
Williams syndrome, structural variants, dogs, hypersociability, sociability, directional selection, human-dog coexistence, comparative genomics
MeSH terms
Animals, Behavior, Animal, Dog Diseases, Dogs, Humans, Social Behavior, Stereotyped Behavior, Genetic Variation, Reproducibility of Results, Polymerase Chain Reaction, Williams Syndrome, Computational Biology, Genetic Predisposition to Disease, Polymorphism, Single Nucleotide, Genetic Association Studies, Molecular Sequence Annotation

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