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Osteogenesis Imperfecta: Skeletal Outcomes After Bisphosphonate Discontinuation at Final Height.

著者 Robinson ME , Trejo P , Palomo T , Glorieux FH , Rauch F
J Bone Miner Res.2019 Jul 29 ; ():.
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Intravenous cyclical bisphosphonates are widely used to treat children with moderate to severe osteogenesis imperfecta (OI). Bisphosphonates are often discontinued when growth is completed, but subsequent skeletal changes have not been studied in detail. We assessed 31 patients (22 females) with OI who had started intravenous bisphosphonates (either pamidronate or zoledronic acid) before 13 years of age, were treated for at least two years (range 4.7-15.7 years) and discontinued treatment after completion of growth, when their age ranged between 13.4-20.0 years (mean: 16.4 years). At 4 years after treatment discontinuation, lumbar spine areal bone mineral density (BMD) had increased by 4% (P < 0.05). Peripheral quantitative computed tomography of the radius showed a decrease in trabecular volumetric BMD at the distal metaphysis of 19% but an increase in cortical volumetric BMD of 4% (P < 0.05 both). None of the patients sustained a new vertebral compression fracture during follow-up. The proportion of patients with new long-bone fractures was higher in the two years before treatment discontinuation than in the last two years of follow-up (42% and 16%, respectively; P < 0.05). This article is protected by copyright. All rights reserved.
PMID: 31356699 [PubMed - as supplied by publisher]
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