Leveraging base-pair mammalian constraint to understand genetic variation and human disease
Sullivan, Patrick F ; Meadows, Jennifer R S ; Gazal, Steven ; Phan, BaDoi N ; Li, Xue ; Genereux, Diane P ; Dong, Michael X ; Bianchi, Matteo ; Andrews, Gregory ; Sakthikumar, Sharadha ... show 10 more
Citations
Authors
Meadows, Jennifer R S
Gazal, Steven
Phan, BaDoi N
Li, Xue
Genereux, Diane P
Dong, Michael X
Bianchi, Matteo
Andrews, Gregory
Sakthikumar, Sharadha
Nordin, Jessika
Roy, Ananya
Christmas, Matthew J
Marinescu, Voichita D
Wang, Chao
Wallerman, Ola
Xue, James
Yao, Shuyang
Sun, Quan
Szatkiewicz, Jin
Wen, Jia
Huckins, Laura M
Lawler, Alyssa
Keough, Kathleen C
Zheng, Zhili
Zeng, Jian
Wray, Naomi R
Li, Yun
Johnson, Jessica
Chen, Jiawen
Paten, Benedict
Reilly, Steven K
Hughes, Graham M
Weng, Zhiping
Pollard, Katherine S
Pfenning, Andreas R
Forsberg-Nilsson, Karin
Karlsson, Elinor K
Lindblad-Toh, Kerstin
Student Authors
Gregory Andrews
Faculty Advisor
Academic Program
Document Type
Publication Date
Keywords
Subject Area
Embargo Expiration Date
Link to Full Text
Abstract
Thousands of genomic regions have been associated with heritable human diseases, but attempts to elucidate biological mechanisms are impeded by an inability to discern which genomic positions are functionally important. Evolutionary constraint is a powerful predictor of function, agnostic to cell type or disease mechanism. Single-base phyloP scores from 240 mammals identified 3.3% of the human genome as significantly constrained and likely functional. We compared phyloP scores to genome annotation, association studies, copy-number variation, clinical genetics findings, and cancer data. Constrained positions are enriched for variants that explain common disease heritability more than other functional annotations. Our results improve variant annotation but also highlight that the regulatory landscape of the human genome still needs to be further explored and linked to disease.
Source
Sullivan PF, Meadows JRS, Gazal S, Phan BN, Li X, Genereux DP, Dong MX, Bianchi M, Andrews G, Sakthikumar S, Nordin J, Roy A, Christmas MJ, Marinescu VD, Wang C, Wallerman O, Xue J, Yao S, Sun Q, Szatkiewicz J, Wen J, Huckins LM, Lawler A, Keough KC, Zheng Z, Zeng J, Wray NR, Li Y, Johnson J, Chen J; Zoonomia Consortium§; Paten B, Reilly SK, Hughes GM, Weng Z, Pollard KS, Pfenning AR, Forsberg-Nilsson K, Karlsson EK, Lindblad-Toh K. Leveraging base-pair mammalian constraint to understand genetic variation and human disease. Science. 2023 Apr 28;380(6643):eabn2937. doi: 10.1126/science.abn2937. Epub 2023 Apr 28. PMID: 37104612; PMCID: PMC10259825.
Year of Medical School at Time of Visit
Sponsors
Dates of Travel
DOI
Permanent Link to this Item
PubMed ID
Other Identifiers
Notes
Funding and Acknowledgements
Corresponding Author
Related Resources
This article is based on a previously available preprint in bioRxiv, https://doi.org/10.1101/2023.03.10.531987