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Beneficial effect of long term intravenous bisphosphonate treatment of osteogenesis imperfecta.

AIM: To find an effective symptomatic treatment for osteogenesis imperfecta (OI). METHODS: In a prospective observational study disodium pamidronate (APD) was given as monthly intravenous infusions to 28 children and adolescents (aged 0.6-18 years) with severe OI or a milder form of the disease, but with spinal compression fractures. RESULTS: During treatment for 2-9 years, dual energy x ray absorptiometry measurements of the total body and of the lumbar spine showed a gradual increase in bone density. All bone metabolism variables in serum (alkaline phosphatase, osteocalcin, procollagen 1 C-terminal peptide, collagen 1 teleopeptide) and urine (deoxypyridinoline) indicated that there was a decrease in bone turnover. All patients experienced beneficial effects and the younger patients reported a major improvement in wellbeing, pain, and mobility without significant side effects. Vertebral remodelling was also seen. CONCLUSIONS: APD seems to be an efficient symptomatic treatment for children and adolescents with OI.

Pubmed ID: 11970931


  • Aström E
  • Söderhäll S


Archives of disease in childhood

Publication Data

May 23, 2002

Associated Grants


Mesh Terms

  • Absorptiometry, Photon
  • Adolescent
  • Biological Markers
  • Bone Density
  • Bone Remodeling
  • Child
  • Child, Preschool
  • Diphosphonates
  • Fractures, Spontaneous
  • Health Status
  • Humans
  • Infant
  • Infusions, Intravenous
  • Long-Term Care
  • Osteogenesis Imperfecta
  • Pain
  • Prospective Studies
  • Recurrence
  • Spinal Cord Compression
  • Treatment Outcome