Deficient mineralization of intramembranous bone in vitamin D-24-hydroxylase-ablated mice is due to elevated 1,25-dihydroxyvitamin D and not to the absence of 24,25-dihydroxyvitamin D

Endocrinology. 2000 Jul;141(7):2658-66. doi: 10.1210/endo.141.7.7579.

Abstract

The 25-hydroxyvitamin D-24-hydroxylase enzyme (24-OHase) is responsible for the catabolic breakdown of 1,25-dihydroxyvitamin D [1,25(OH)2D], the active form of vitamin D. The 24-OHase enzyme can also act on the 25-hydroxyvitamin D substrate to generate 24,25-dihydroxyvitamin D, a metabolite whose physiological importance remains unclear. We report that mice with a targeted inactivating mutation of the 24-OHase gene had impaired 1,25(OH)2D catabolism. Surprisingly, complete absence of 24-OHase activity during development leads to impaired intramembranous bone mineralization. This phenotype was rescued by crossing the 24-OHase mutant mice to mice harboring a targeted mutation in the vitamin D receptor gene, confirming that the elevated 1,25(OH)2D levels, acting through the vitamin D receptor, were responsible for the observed accumulation of osteoid. Our results confirm the physiological importance of the 24-OHase enzyme for maintaining vitamin D homeostasis, and they reveal that 24,25-dihydroxyvitamin D is a dispensable metabolite during bone development.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • 24,25-Dihydroxyvitamin D 3 / deficiency*
  • Alleles
  • Animals
  • Bone Density*
  • Calcitriol / blood
  • Calcitriol / metabolism*
  • Calcitriol / pharmacology
  • Cytochrome P-450 Enzyme System / deficiency*
  • Cytochrome P-450 Enzyme System / genetics
  • Female
  • Hybridization, Genetic
  • Kidney / drug effects
  • Kidney / pathology
  • Mice
  • Mice, Knockout / genetics
  • Mutation / physiology
  • Phenotype
  • Rats
  • Receptors, Calcitriol / deficiency*
  • Receptors, Calcitriol / genetics
  • Steroid Hydroxylases / deficiency*
  • Steroid Hydroxylases / genetics
  • Vitamin D3 24-Hydroxylase

Substances

  • Receptors, Calcitriol
  • 24,25-Dihydroxyvitamin D 3
  • Cytochrome P-450 Enzyme System
  • Steroid Hydroxylases
  • Vitamin D3 24-Hydroxylase
  • Calcitriol