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Two doses of pamidronate in infants with osteogenesis imperfecta

S Senthilnathan1, E Walker, N J Bishop

  • 1University of Sheffield, Western Bank, Sheffield S10 2TH, UK. n.j.bishop@sheffield.ac.uk

Archives of Disease in Childhood
|December 20, 2007
PubMed

Insights

Higher doses of pamidronate (12 mg/kg/year) improved bone density in infants with osteogenesis imperfecta (OI) compared to lower doses (6 mg/kg/year). This treatment also improved fractures and vertebral size without over-suppressing bone turnover.

Area of Science:

  • Pediatric Endocrinology
  • Skeletal Dysplasias
  • Pharmacology

Background:

  • Osteogenesis imperfecta (OI) is a genetic disorder characterized by fragile bones.
  • Current intravenous pamidronate regimens for OI range from 3-12 mg/kg/year.
  • The optimal dosage of pamidronate for infants with OI remains under investigation.

Purpose of the Study:

  • To evaluate the effect of two different annual doses of pamidronate (6 vs. 12 mg/kg/year) on skeletal health in infants with OI.
  • To assess bone mineral density, fracture incidence, and bone turnover markers.

Main Methods:

  • A cohort of 12 infants with OI was recruited over 4 years.
  • Infants received either 6 or 12 mg/kg/year of intravenous pamidronate.
  • Skeletal surveys and DXA bone density measurements were performed at baseline and 12 months.

Main Results:

  • Bone mass increased in both treatment groups.
  • Infants receiving 12 mg/kg/year showed significantly higher spine bone density compared to those receiving 6 mg/kg/year (p=0.04).
  • Fracture outcomes improved or stabilized in most infants, and metaphyseal remodeling was not impaired.

Conclusions:

  • Pamidronate dosage may influence lumbar spine bone acquisition in infants with OI.
  • Pamidronate treatment improved vertebral size in infants with prior crush fractures.
  • The drug effectively reduced bone turnover markers without excessive suppression.
Abstract

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