Genome-wide discovery of multiple sclerosis genetic risk variant allelic regulatory activity
Multiple sclerosis is an immune-mediated demyelinating disease of the central nervous system with a complex etiology involving environmental and genetic factors. Numerous genetic risk loci for multiple sclerosis have been nominated through genome-wide association studies, with most associated variants residing in noncoding regions. However, further work is needed to understand how genetic variation contributes to disease-related alterations to gene expression. Here, we use Massively Parallel Reporter Assays to identify genetic risk variants with genotype-dependent enhancing or silencing activity within a set of 14,275 variants distributed among multiple sclerosis risk loci that have reached genome-wide or suggestive significance. We applied our Massively Parallel Reporter Assay library to Epstein-Barr-virus-transformed B cell lines derived from two patients with multiple sclerosis, as well as the ENCODE Tier 1 cell line GM12878. In total, our approach discovered 150 allelic enhancing variants and 286 allelic silencing variants, collectively representing 83 independent multiple sclerosis risk loci. Our systematic, genome-scale approach implicates potentially causal genotype-dependent gene regulatory mechanisms for over a third of the known multiple sclerosis risk loci, providing a unique resource for the discovery of the genetic mechanisms underlying this chronic inflammatory disease.
Authors
- Lee Edsall (ORCID: https://orcid.org/0000-0002-0326-2829)
- Leah C. Kottyan (ORCID: https://orcid.org/0000-0003-3979-2220)
- Molly Shook (ORCID: https://orcid.org/0009-0001-9419-9879)
- Kristen S. Fisher (ORCID: https://orcid.org/0000-0002-1598-8571)
- Aram Zabeti (ORCID: https://orcid.org/0000-0002-0424-7460)
- Lucinda P. Lawson (ORCID: https://orcid.org/0000-0003-3939-7829)
- Marissa Granitto (ORCID: https://orcid.org/0000-0002-1758-9881)
- Lois Parks (ORCID: https://orcid.org/0000-0001-5689-7492)
- Sreeja Parameswaran (ORCID: https://orcid.org/0009-0002-3631-3669)
- Matthew T. Weirauch (ORCID: https://orcid.org/0000-0001-7977-9122)
- Xiaoting Chen (ORCID: https://orcid.org/0000-0002-3782-3962)
- Carmy Forney (ORCID: https://orcid.org/0000-0001-6418-5794)
Institutions
- Cincinnati Children's Hospital Medical Center (US)
- Baylor College of Medicine (US)
- University of Cincinnati (US)
- University of Cincinnati Medical Center (US)
Publication Details
- Journal
- G3 Genes Genomes Genetics
- Published
- 2025-08-21
- DOI
- https://doi.org/10.1093/g3journal/jkaf192
- Primary Topic
- RNA Research and Splicing
- Type
- article
- Field-Weighted Citation Impact
- 0.00
Funders
- Cincinnati Children's Hospital Medical Center
- National Institutes of Health