Identification of a Functional Risk Variant for Pemphigus Vulgaris in the ST18 Gene

PLoS Genet. 2016 May 5;12(5):e1006008. doi: 10.1371/journal.pgen.1006008. eCollection 2016 May.

Abstract

Pemphigus vulgaris (PV) is a life-threatening autoimmune mucocutaneous blistering disease caused by disruption of intercellular adhesion due to auto-antibodies directed against epithelial components. Treatment is limited to immunosuppressive agents, which are associated with serious adverse effects. The propensity to develop the disease is in part genetically determined. We therefore reasoned that the delineation of PV genetic basis may point to novel therapeutic strategies. Using a genome-wide association approach, we recently found that genetic variants in the vicinity of the ST18 gene confer a significant risk for the disease. Here, using targeted deep sequencing, we identified a PV-associated variant residing within the ST18 promoter region (p<0.0002; odds ratio = 2.03). This variant was found to drive increased gene transcription in a p53/p63-dependent manner, which may explain the fact that ST18 is up-regulated in the skin of PV patients. We then discovered that when overexpressed, ST18 stimulates PV serum-induced secretion of key inflammatory molecules and contributes to PV serum-induced disruption of keratinocyte cell-cell adhesion, two processes previously implicated in the pathogenesis of PV. Thus, the present findings indicate that ST18 may play a direct role in PV and consequently represents a potential target for the treatment of this disease.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Autoantibodies / genetics
  • Autoantibodies / immunology
  • Cytokines / genetics
  • Cytokines / metabolism
  • Female
  • Genetic Variation
  • Genome-Wide Association Study
  • High-Throughput Nucleotide Sequencing
  • Humans
  • Immunosuppressive Agents / adverse effects
  • Keratinocytes / metabolism
  • Keratinocytes / pathology
  • Male
  • Pedigree
  • Pemphigus / blood
  • Pemphigus / genetics*
  • Pemphigus / immunology
  • Pemphigus / therapy
  • Polymorphism, Single Nucleotide
  • Promoter Regions, Genetic / genetics*
  • Repressor Proteins / blood
  • Repressor Proteins / genetics*
  • Risk Factors
  • Skin / metabolism
  • Skin / pathology

Substances

  • Autoantibodies
  • Cytokines
  • Immunosuppressive Agents
  • Repressor Proteins
  • ST18 protein, human

Grants and funding

This work was supported in part by the Israel Science Foundation (ISF)-Natural Science Foundation of China (NSFC) Joint Scientific Research Program grant (ES), a Tel-Aviv Medical Center-Weizmann Institute of Sciences joint grant (ES and DL), the Crown Human Genome Center at the Weizmann Institute of Science (DL) and the German Science Foundation (EXC 306/2) (SI, DZ). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.