Protein misfolding diseases: Prospects of pharmacological treatment

Clin Genet. 2018 Mar;93(3):450-458. doi: 10.1111/cge.13088. Epub 2017 Dec 4.

Abstract

Protein misfolding has been linked to numerous inherited diseases. Loss- and gain-of-function mutations (common features of genetic diseases) may cause the destabilization of proteins, leading to alterations in their properties and/or cellular location, resulting in their incorrect functioning. Misfolded proteins can, however, be rescued via the use of proteostasis regulators and/or pharmacological chaperones, suggesting that treatments with small molecules might be developed for a range of genetic diseases. This work describes the potential of these small molecules in this respect, including for the treatment of congenital disorder of glycosylation (CDG) due to phosphomannomutase 2 deficiency (PMM2-CDG).

Keywords: conformational diseases; congenital disorders of glycosylation; inborn errors of metabolism; misfolding diseases; pharmacological chaperones; protein folding; proteostasis regulators.

Publication types

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

MeSH terms

  • Animals
  • Clinical Trials as Topic
  • Drug Discovery
  • Genetic Predisposition to Disease
  • Glycosylation / drug effects
  • Humans
  • Mitochondria
  • Mutation
  • Proteostasis Deficiencies / diagnosis
  • Proteostasis Deficiencies / drug therapy*
  • Proteostasis Deficiencies / etiology*
  • Proteostasis Deficiencies / metabolism