Using yeast to model calcium-related diseases: example of the Hailey-Hailey disease

Biochim Biophys Acta. 2014 Oct;1843(10):2315-21. doi: 10.1016/j.bbamcr.2014.02.011. Epub 2014 Feb 25.

Abstract

Cross-complementation studies offer the possibility to overcome limitations imposed by the inherent complexity of multicellular organisms in the study of human diseases, by taking advantage of simpler model organisms like the budding yeast Saccharomyces cerevisiae. This review deals with, (1) the use of S. cerevisiae as a model organism to study human diseases, (2) yeast-based screening systems for the detection of disease modifiers, (3) Hailey-Hailey as an example of a calcium-related disease, and (4) the presentation of a yeast-based model to search for chemical modifiers of Hailey-Hailey disease. The preliminary experimental data presented and discussed here show that it is possible to use yeast as a model system for Hailey-Hailey disease and suggest that in all likelihood, yeast has the potential to reveal candidate drugs for the treatment of this disorder. This article is part of a Special Issue entitled: Calcium signaling in health and disease. Guest Editors: Geert Bultynck, Jacques Haiech, Claus W. Heizmann, Joachim Krebs, and Marc Moreau.

Keywords: ATP2C1/PMR1; Budding yeast Saccharomyces cerevisiae; Calcium; Hailey–Hailey disease; Yeast models for human diseases; Yeast-based drug screening assay.

Publication types

  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • Administration, Cutaneous
  • Calcium / metabolism*
  • Calcium Signaling
  • Calcium-Transporting ATPases / deficiency
  • Calcium-Transporting ATPases / genetics*
  • Dermatologic Agents / pharmacology
  • Dihydroxycholecalciferols / pharmacology
  • Gene Expression
  • Humans
  • Keratinocytes / drug effects
  • Keratinocytes / metabolism
  • Keratinocytes / pathology
  • Models, Biological*
  • Molecular Chaperones / genetics*
  • Mutation
  • Pemphigus, Benign Familial / drug therapy
  • Pemphigus, Benign Familial / genetics*
  • Pemphigus, Benign Familial / metabolism
  • Pemphigus, Benign Familial / pathology
  • Saccharomyces cerevisiae / genetics*
  • Saccharomyces cerevisiae / metabolism
  • Saccharomyces cerevisiae Proteins / genetics*
  • Skin / drug effects
  • Skin / metabolism
  • Skin / pathology

Substances

  • Dermatologic Agents
  • Dihydroxycholecalciferols
  • Molecular Chaperones
  • SSC1 protein, S cerevisiae
  • Saccharomyces cerevisiae Proteins
  • 1 alpha,24-dihydroxyvitamin D3
  • ATP2C1 protein, human
  • Calcium-Transporting ATPases
  • Calcium