Inflamed Ulcerative Colitis Regions Associated With MRGPRX2-Mediated Mast Cell Degranulation and Cell Activation Modules, Defining a New Therapeutic Target

Gastroenterology. 2021 Apr;160(5):1709-1724. doi: 10.1053/j.gastro.2020.12.076. Epub 2021 Jan 6.

Abstract

Background & aims: Recent literature has implicated a key role for mast cells in murine models of colonic inflammation, but their role in human ulcerative colitis (UC) is not well established. A major advance has been the identification of mrgprb2 (human orthologue, MRGPX2) as mediating IgE-independent mast cell activation. We sought to define mechanisms of mast cell activation and MRGPRX2 in human UC.

Methods: Colon tissues were collected from patients with UC for bulk RNA sequencing and lamina propria cells were isolated for MRGPRX2 activation studies and single-cell RNA sequencing. Genetic association of all protein-altering G-protein coupled receptor single-nucleotide polymorphism was performed in an Ashkenazi Jewish UC case-control cohort. Variants of MRGPRX2 were transfected into Chinese hamster ovary (CHO) and human mast cell (HMC) 1.1 cells to detect genotype-dependent effects on β-arrestin recruitment, IP-1 accumulation, and phosphorylated extracellular signal-regulated kinase.

Results: Mast cell-specific mediators and adrenomedullin (proteolytic precursor of PAMP-12, an MRGPRX2 agonist) are up-regulated in inflamed compared to uninflamed UC. MRGPRX2 stimulation induces carboxypeptidase secretion from inflamed UC. Of all protein-altering GPCR alleles, a unique variant of MRGPRX2, Asn62Ser, was most associated with and was bioinformatically predicted to alter arrestin recruitment. We validated that the UC protective serine allele enhances β-arrestin recruitment, decreases IP-1, and increases phosphorylated extracellular signal-regulated kinase with MRGPRX2 agonists. Single-cell RNA sequencing defines that adrenomedullin is expressed by activated fibroblasts and epithelial cells and that interferon gamma is a key upstream regulator of mast cell gene expression.

Conclusion: Inflamed UC regions are distinguished by MRGPRX2-mediated activation of mast cells, with decreased activation observed with a UC-protective genetic variant. These results define cell modules of UC activation and a new therapeutic target.

Keywords: G-Protein Coupled Receptors; Mast Cells; Single Cell Sequencing; Ulcerative Colitis.

Publication types

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

MeSH terms

  • Adrenomedullin / genetics
  • Adrenomedullin / metabolism
  • Animals
  • CHO Cells
  • Case-Control Studies
  • Cell Degranulation*
  • Colitis, Ulcerative / genetics
  • Colitis, Ulcerative / immunology
  • Colitis, Ulcerative / metabolism*
  • Colon / immunology
  • Colon / metabolism*
  • Cricetulus
  • Extracellular Signal-Regulated MAP Kinases / metabolism
  • Genetic Variation
  • Humans
  • Inositol Phosphates / metabolism
  • Ligands
  • Mast Cells / immunology
  • Mast Cells / metabolism*
  • Nerve Tissue Proteins / genetics
  • Nerve Tissue Proteins / metabolism*
  • Phosphorylation
  • Receptors, G-Protein-Coupled / genetics
  • Receptors, G-Protein-Coupled / metabolism*
  • Receptors, Neuropeptide / genetics
  • Receptors, Neuropeptide / metabolism*
  • beta-Arrestin 2 / genetics
  • beta-Arrestin 2 / metabolism

Substances

  • ADM protein, human
  • ARRB2 protein, human
  • Inositol Phosphates
  • Ligands
  • MRGPRX2 protein, human
  • Nerve Tissue Proteins
  • Receptors, G-Protein-Coupled
  • Receptors, Neuropeptide
  • beta-Arrestin 2
  • Adrenomedullin
  • Extracellular Signal-Regulated MAP Kinases