Treatment of children with Osteogenesis imperfecta in Estonia

Acta Paediatr. 2003 Apr;92(4):452-5. doi: 10.1111/j.1651-2227.2003.tb00577.x.

Abstract

Aim: To analyse the changes in fracture rate, bone density and histology in children with Osteogenesis imperfecta receiving treatment with alendronate (oral bisphosphonate) and calcitriol.

Methods: Children treated at Tartu University Hospital from 1995 to 2001 were examined for Osteogenesis imperfecta. Radiographs and bone density measurements were obtained for all patients at the beginning of the study. Four patients also had bone biopsies prior to and one year after beginning treatment. The children were then given alendronate in weight-dependent dosages and also calcitriol. The number of fractures during the treatment period was recorded and follow-up bone density measurements were made.

Results: Fifteen patients were treated during the 6-y period; mean follow-up approximately 3 y. It was found that the number of bone fractures had decreased significantly (p < 0.0001). Bone density improved in all 15 patients. Histologic studies revealed an increased number of osteoblasts and thickness of bone trabeculae as well as a more regular bone lamellar structure at the time of the second operation.

Conclusion: The complex treatment of Osteogenesis imperfecta should include alendronate and calcitriol to decrease fractures and improve bone mineral density.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Administration, Oral
  • Adolescent
  • Alendronate / administration & dosage*
  • Alendronate / therapeutic use*
  • Bone Density / drug effects*
  • Calcitriol / administration & dosage*
  • Calcitriol / therapeutic use*
  • Calcium Channel Agonists / administration & dosage*
  • Calcium Channel Agonists / therapeutic use*
  • Child
  • Child, Preschool
  • Drug Therapy, Combination
  • Estonia
  • Female
  • Follow-Up Studies
  • Fractures, Bone / etiology*
  • Fractures, Bone / pathology
  • Fractures, Bone / prevention & control*
  • Humans
  • Infant
  • Male
  • Osteogenesis Imperfecta / complications*
  • Osteogenesis Imperfecta / drug therapy*
  • Osteogenesis Imperfecta / pathology
  • Outcome Assessment, Health Care
  • Retrospective Studies
  • Time Factors

Substances

  • Calcium Channel Agonists
  • Calcitriol
  • Alendronate