Decreased bone density in carriers and patients of an Israeli family with the osteoporosis-pseudoglioma syndrome

Isr Med Assoc J. 2003 Jun;5(6):419-21.

Abstract

Background: The osteoporosis-pseudoglioma syndrome is a rare autosomal recessive disorder characterized by severe juvenile-onset osteoporosis and congenital or early-onset blindness. Other manifestations include muscular hypotonia, ligamentous laxity, mild mental retardation and seizures. The gene responsible was recently identified to be the low density lipoprotein receptor-related family member LRP5 on chromosome 11q11-12.

Objective: To measure bone density in two siblings with the OPPG syndrome as well as in their family members (parents and siblings).

Methods: Bone mineral density was determined in the lumbar spine (antero-posterior), femoral neck, two-thirds distal forearm (> 95% cortical bone) and ultradistal forearm (predominantly trabecular bone) by dual-energy X-ray absorptiometry.

Results: The studies revealed osteoporotic changes both in the patients and the carriers.

Conclusion: The findings demonstrate that OPPG carriers have reduced bone mass, which is a risk factor for development of early osteoporotic changes.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / pathology*
  • Absorptiometry, Photon
  • Adolescent
  • Adult
  • Blindness / genetics*
  • Bone Density*
  • Case-Control Studies
  • Chromosomes, Human, Pair 11 / genetics*
  • Consanguinity
  • Forearm / pathology
  • Genes, Recessive / genetics
  • Heterozygote
  • Humans
  • Israel
  • Jews / genetics
  • LDL-Receptor Related Proteins
  • Low Density Lipoprotein Receptor-Related Protein-5
  • Lumbar Vertebrae / pathology
  • Male
  • Mutation / genetics
  • Osteoporosis / genetics*
  • Osteoporosis / pathology*
  • Pedigree
  • Receptors, LDL / genetics*
  • Risk Factors
  • Seizures / genetics*
  • Syndrome
  • Tunisia

Substances

  • LDL-Receptor Related Proteins
  • LRP5 protein, human
  • Low Density Lipoprotein Receptor-Related Protein-5
  • Receptors, LDL