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Improved Methodology for Studying Postnatal Osteogenesis via Intramembranous Ossification in a Murine Bone Marrow Injury Model
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Cyclic pamidronate infusion for neonatal-onset osteogenesis imperfecta

Chia-Hsuan Lin1, Yin-Hsiu Chien2, Shinn-Forng Peng3

  • 1Department of Pediatrics, National Taiwan University Hospital and National Taiwan University College of Medicine, Taiwan.

Pediatrics and Neonatology
|February 4, 2014
PubMed

Insights

Early cyclic pamidronate treatment in severe osteogenesis imperfecta (OI) improves bone density and function. This intervention in infants with neonatal OI leads to favorable outcomes, including normal ambulatory function.

Area of Science:

  • Pediatric Orthopedics
  • Medical Genetics
  • Pharmacology

Background:

  • Severe osteogenesis imperfecta (OI) presents with low bone mass, frequent fractures, and pain from birth, leading to a poor prognosis.
  • Neonatal OI significantly impacts infant health and development, necessitating early intervention strategies.

Purpose of the Study:

  • To evaluate the efficacy of early cyclic pamidronate treatment in infants with severe osteogenesis imperfecta.
  • To assess the long-term outcomes, including bone mineral density, fracture rates, and functional status, in patients treated before one year of age.

Main Methods:

  • Six infants with severe OI, experiencing fractures in utero or within the first month of life, received cyclic pamidronate infusions starting at a mean age of 2.8 months.
  • Treatment involved regular pamidronate infusions, with outcomes monitored for bone mineral density, fracture incidence, and functional development.

Main Results:

  • Patients tolerated treatment, with transient hypocalcemia as the primary side effect during the first infusion.
  • Improvements in irritability and feeding were noted within 2-3 months post-infusion.
  • Significant increases in bone mineral density were observed over the first two years, with a fracture rate of 0.6 per year. At a mean age of 6.4 years, five patients achieved normal ambulatory function, though they remained shorter than average.

Conclusions:

  • Early initiation of cyclic pamidronate therapy in neonatal osteogenesis imperfecta can lead to favorable clinical outcomes.
  • Timely treatment is crucial for improving bone health and functional abilities in infants with severe OI.
Abstract

Keywords:
bisphosphonatebone mineral densityneonateosteogenesis imperfecta

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