Risedronate in adults with osteogenesis imperfecta type I: Increased bone mineral density and decreased bone turnover, but high fracture rate persists

Bradbury, L.A., Barlow, S., Geoghegan, F., Hannon, R.A., Stuckey, S.L., Wass, J.A.H., Russell, R.G.G., Brown, M.A., & Duncan, E.L. (2012) Risedronate in adults with osteogenesis imperfecta type I: Increased bone mineral density and decreased bone turnover, but high fracture rate persists. Osteoporosis International, 23(1), pp. 285-294.

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Abstract

Summary

Bisphosphonates can increase bone mineral density (BMD) in children with osteogenesis imperfecta (OI). In this study of adults with OI type I, risedronate increased BMD at lumbar spine (but not total hip) and decreased bone turnover. However, the fracture rate in these patients remained high.

Introduction

Intravenous bisphosphonates given to children with OI can increase BMD and reduce fracture incidence. Oral and/or intravenous bisphosphonates may have similar effects in adults with OI. We completed an observational study of the effect of risedronate in adults with OI type I.

Methods

Thirty-two adults (mean age, 39 years) with OI type I were treated with risedronate (total dose, 35 mg weekly) for 24 months. Primary outcome measures were BMD changes at lumbar spine (LS) and total hip (TH). Secondary outcome measures were fracture incidence, bone pain, and change in bone turnover markers (serum procollagen type I aminopropeptide (P1NP) and bone ALP). A meta-analysis of published studies of oral bisphosphonates in adults and children with OI was performed.

Results

Twenty-seven participants (ten males and seventeen females) completed the study. BMD increased at LS by 3.9% (0.815 vs. 0.846 g/cm 2, p=0.007; mean Z-score, -1.93 vs. -1.58, p=0.002), with no significant change at TH. P1NP fell by 37% (p=0.00041), with no significant change in bone ALP (p=0.15). Bone pain did not change significantly (p=0.6). Fracture incidence remained high, with 25 clinical fractures and 10 major fractures in fourteen participants (0.18 major fractures per person per year), with historical data of 0.12 fractures per person per year. The meta-analysis did not demonstrate a significant difference in fracture incidence in patients with OI treated with oral bisphosphonates.

Conclusions

Risedronate in adults with OI type I results in modest but significant increases in BMD at LS, and decreased bone turnover. However, this may be insufficient to make a clinically significant difference to fracture incidence.

Impact and interest:

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19 citations in Web of Science®

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ID Code: 89347
Item Type: Journal Article
Refereed: Yes
Keywords: Bisphosphonates, BMD, Bone turnover, Fracture, Osteogenesis imperfecta, bisphosphonic acid derivative, risedronic acid, adult, aged, article, bone density, bone mineral, bone pain, clinical article, constipation, diarrhea, female, gastrointestinal symptom, hip, human, incidence, lumbar spine, male, meta analysis (topic), observational study, outcome assessment, patient compliance, priority journal, prospective study, treatment outcome, visual analog scale, vitamin D deficiency, Adolescent, Biological Markers, Bone Density Conservation Agents, Bone Remodeling, Etidronic Acid, Follow-Up Studies, Fractures, Bone, Hip Joint, Humans, Lumbar Vertebrae, Middle Aged, Young Adult
DOI: 10.1007/s00198-011-1658-2
ISSN: 0937-941X
Divisions: Current > QUT Faculties and Divisions > Faculty of Health
Current > Institutes > Institute of Health and Biomedical Innovation
Copyright Owner: Copyright 2011 International Osteoporosis Foundation and National Osteoporosis Foundation
Deposited On: 20 Oct 2015 23:10
Last Modified: 28 Mar 2016 23:25

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