Coronary pathology of inherited generalized arterial calcification of infancy: a case report

Cardiovasc Pathol. 2018 Sep-Oct:36:15-19. doi: 10.1016/j.carpath.2018.05.001. Epub 2018 May 18.

Abstract

Generalized arterial calcification of infancy (GACI), or idiopathic infantile arterial calcification, is a rare autosomal-recessive disease recognized aAs an inherited disorder characterized by severe pathologic calcification of large- and medium-sized arteries accompanied by smooth muscle cell (SMC) hyperplasia leading to vascular obstruction [1]. The prognosis is extremely poor, with 85% of affected infants dying within the first 6 months of life. Loss-of-function mutations in the ectonucleotide pyrophosphatase phosphodiesterase 1 (ENPP1) gene is recognized as the main defect associated with GACI [1]. The underlying pathogenesis of osteogenic transition leading to calcification and severe stenosis in GACI, however, is poorly understood. Herein, we present a case of a GACI patient with cardiac complications who exhibited extensive vascular disease at autopsy.

Keywords: Generalized arterial calcification of infancy.

Publication types

  • Case Reports

MeSH terms

  • Autopsy
  • Biopsy
  • Coronary Angiography
  • Coronary Vessels / diagnostic imaging
  • Coronary Vessels / pathology*
  • Fatal Outcome
  • Genetic Predisposition to Disease
  • Humans
  • Infant
  • Male
  • Muscle, Smooth, Vascular / pathology
  • Mutation
  • Myocardium / pathology
  • Neointima
  • Phenotype
  • Phosphoric Diester Hydrolases / genetics
  • Pyrophosphatases / genetics
  • Vascular Calcification / diagnostic imaging
  • Vascular Calcification / enzymology
  • Vascular Calcification / genetics
  • Vascular Calcification / pathology*

Substances

  • Phosphoric Diester Hydrolases
  • ectonucleotide pyrophosphatase phosphodiesterase 1
  • Pyrophosphatases

Supplementary concepts

  • Arterial calcification of infancy