Research Progress in Pseudoxanthoma Elasticum and Related Ectopic Mineralization Disorders

J Invest Dermatol. 2016 Mar;136(3):550-556. doi: 10.1016/j.jid.2015.10.065.

Abstract

Heritable ectopic mineralization disorders represent a phenotypically diverse group of conditions characterized by deposition of calcium phosphate complexes in soft connective tissues. The prototype of such conditions is pseudoxanthoma elasticum, and related conditions with overlapping clinical features include generalized arterial calcification of infancy and arterial calcification due to CD73 deficiency. Molecular genetic investigations have revealed mutations in the genes physiologically involved in generation of inorganic pyrophosphate and inorganic phosphate, and the findings suggest a unifying pathomechanism relating to reduced inorganic pyrophosphate/inorganic phosphate ratio. This hypothesis is based on the notion that inorganic pyrophosphate serves as a powerful inhibitor of mineralization, whereas inorganic phosphate is a promineralization factor, and an appropriate inorganic pyrophosphate/inorganic phosphate ratio is critical for prevention of ectopic mineralization under homeostatic conditions.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Review

MeSH terms

  • 5'-Nucleotidase / genetics
  • Animals
  • Biomedical Research / trends
  • Diphosphates / metabolism*
  • Forecasting
  • GPI-Linked Proteins / genetics
  • Genetic Predisposition to Disease / epidemiology*
  • Humans
  • Mice
  • Mice, Knockout
  • Molecular Biology / methods
  • Mutation
  • Phenotype
  • Pseudoxanthoma Elasticum / genetics*
  • Rare Diseases
  • Vascular Calcification / genetics*
  • Vascular Calcification / physiopathology

Substances

  • Diphosphates
  • GPI-Linked Proteins
  • diphosphoric acid
  • 5'-Nucleotidase
  • NT5E protein, human