A polyclonal allelic expression assay for detecting regulatory effects of transcript variants

Genome Med. 2020 Sep 11;12(1):79. doi: 10.1186/s13073-020-00777-8.

Abstract

We present an assay to experimentally test the regulatory effects of genetic variants within transcripts using CRISPR/Cas9 followed by targeted sequencing. We applied the assay to 32 premature stop-gained variants across the genome and in two Mendelian disease genes, 33 putative causal variants of eQTLs, and 62 control variants in HEK293T cells, replicating a subset of variants in HeLa cells. We detected significant effects in the expected direction (in 60% of variants), demonstrating the ability of the assay to capture regulatory effects of eQTL variants and nonsense-mediated decay triggered by premature stop-gained variants. The results suggest a utility for validating transcript-level effects of genetic variants.

Keywords: CRISPR/Cas9 genome editing; Nonsense-mediated decay; Regulatory variation; Variant validation; eQTL.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Alleles*
  • CRISPR-Cas Systems
  • Gene Editing
  • Gene Expression Regulation*
  • Genetic Variation*
  • HEK293 Cells
  • Humans
  • Nonsense Mediated mRNA Decay
  • Quantitative Trait Loci
  • Transcription, Genetic*

Associated data

  • figshare/10.6084/m9.figshare.9883232