Identifying severe COVID-19 risk variants modulating enhancer reporter activity in lung cells
Common genetic variants contribute to risk for complex human diseases. However, despite thousands of associations, variants modulating disease risk and their functional impact remain largely unknown. This includes SARS-CoV-2 infection, where outcomes range from asymptomatic to fatal. Most genetic risk variants associated with COVID-19 disease, identified through genome wide association studies, are located in the non-coding genome and may function by altering gene expression in disease-relevant cells and tissues. To address this at scale, we tested >4800 severe COVID-19-associated variants to determine the impact of individual variants and variant combinations on regulatory activity using Self-Transcribing Active Regulatory Region sequencing, a massively-parallel reporter assay. Focusing on variants that may have their impact in the lung, in a lung epithelial cell line (A549) we identify 166 variants within active sequences, of which 29 modulate activity allele-specifically. Evaluating variant combinations, we observe both additive and non-additive effects on regulatory activity. We employ state-of-the-art deep learning models to interpret allele-specific variant effects on regulatory activity and endogenous genomic features. Our work provides a set of prioritised severe COVID-19-associated variants that modulate regulatory activity in lung epithelial cells, candidate transcription factors, and candidate target genes with potential to be disease modifying.
Authors
- Elias T. Friman (ORCID: https://orcid.org/0000-0001-9944-6560)
- Wendy A. Bickmore (ORCID: https://orcid.org/0000-0001-6660-7735)
- Giovanna Weykopf (ORCID: https://orcid.org/0009-0006-2048-4796)
- Simon C. Biddie
Institutions
- Edinburgh Royal Infirmary (GB)
- Institute of Genetics and Cancer (GB)
- University of Edinburgh (GB)
Publication Details
- Journal
- PLoS Genetics
- Published
- 2026-07-17
- DOI
- https://doi.org/10.1371/journal.pgen.1012222
- Primary Topic
- Genomics and Rare Diseases
- Type
- article
- Field-Weighted Citation Impact
- 0.00
Funders
- UK Research and Innovation
- Academy of Medical Sciences
- Medical Research Council
- Institute of Genetics
- Medical Research Foundation