Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled Trials

Objective To assess the effectiveness and safety of oral bisphosphonates in increasing bone mineral density (BMD), reducing fractures, and improving clinical function in patients with osteogenesis imperfecta (OI). Methods Studies were eligible for inclusion if they were randomized controlled trials...

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Main Authors: Zhi‐min Ying, Bin Hu, Shi‐gui Yan
Format: Article
Language:English
Published: Wiley 2020-08-01
Series:Orthopaedic Surgery
Subjects:
Online Access:https://doi.org/10.1111/os.12611
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author Zhi‐min Ying
Bin Hu
Shi‐gui Yan
author_facet Zhi‐min Ying
Bin Hu
Shi‐gui Yan
author_sort Zhi‐min Ying
collection DOAJ
description Objective To assess the effectiveness and safety of oral bisphosphonates in increasing bone mineral density (BMD), reducing fractures, and improving clinical function in patients with osteogenesis imperfecta (OI). Methods Studies were eligible for inclusion if they were randomized controlled trials of directly comparing oral bisphosphonate therapy with placebo‐group in OI patients. Data synthesis regarding to bone mineral density as measured by dual‐energy X‐ray absorptiometry (DEXA), decreased fracture incidence, change in biochemical markers of bone and mineral metabolism, bone histology, growth, bone pain, quality of life, and others were assessed, and meta‐analysis done when possible. Results From 98 potential references and six randomized controlled studies a total of 263 participants receiving oral bisphosphonates and 143 placebo treatments contributed data to meta‐analysis. Pooled meta‐analysis of three studies suggested that there was significant difference between bisphosphonate treated group and placebo in number of patients with at least one fracture (mean difference 0.53, 95% confidence interval 0.32–0.89, P = 0.02). Pooled meta‐analysis of two studies suggested that significant difference was noted between bisphosphonate treated group and placebo in mean percentage change in spine BMD (T‐score) (mean difference 28.43, 95% confidence interval 7.09‐49.77, P = 0.009). The similar effect was shown in the term of mean change (Z‐score) in spine BMD. Conclusions Significant improvement in lumbar areal BMD in patients affected with OI has been shown when treated with oral bisphosphonates, even though only a small population was enrolled. We cannot draw a definite conclusion that the increase in BMD can be translated into fracture reduction and clinical functional improvement. The optimal method, dose, type, initiation, and duration of oral bisphosphonates therapy still remains unclear. Well‐designed, adequately‐powered, placebo‐controlled RCTs investigating the effects of oral bisphosphonates on fractures reduction and improvement in quality of life in both children and adults are studied here.
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spelling doaj.art-53ad489739fa4f909c5e821673b035c82022-12-22T00:23:36ZengWileyOrthopaedic Surgery1757-78531757-78612020-08-011241293130310.1111/os.12611Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled TrialsZhi‐min Ying0Bin Hu1Shi‐gui Yan2Department of Orthopaedic Surgery, the Second Affiliated Hospital School of Medicine, Zhejiang University Zhejiang ChinaDepartment of Orthopaedic Surgery, the Second Affiliated Hospital School of Medicine, Zhejiang University Zhejiang ChinaDepartment of Orthopaedic Surgery, the Second Affiliated Hospital School of Medicine, Zhejiang University Zhejiang ChinaObjective To assess the effectiveness and safety of oral bisphosphonates in increasing bone mineral density (BMD), reducing fractures, and improving clinical function in patients with osteogenesis imperfecta (OI). Methods Studies were eligible for inclusion if they were randomized controlled trials of directly comparing oral bisphosphonate therapy with placebo‐group in OI patients. Data synthesis regarding to bone mineral density as measured by dual‐energy X‐ray absorptiometry (DEXA), decreased fracture incidence, change in biochemical markers of bone and mineral metabolism, bone histology, growth, bone pain, quality of life, and others were assessed, and meta‐analysis done when possible. Results From 98 potential references and six randomized controlled studies a total of 263 participants receiving oral bisphosphonates and 143 placebo treatments contributed data to meta‐analysis. Pooled meta‐analysis of three studies suggested that there was significant difference between bisphosphonate treated group and placebo in number of patients with at least one fracture (mean difference 0.53, 95% confidence interval 0.32–0.89, P = 0.02). Pooled meta‐analysis of two studies suggested that significant difference was noted between bisphosphonate treated group and placebo in mean percentage change in spine BMD (T‐score) (mean difference 28.43, 95% confidence interval 7.09‐49.77, P = 0.009). The similar effect was shown in the term of mean change (Z‐score) in spine BMD. Conclusions Significant improvement in lumbar areal BMD in patients affected with OI has been shown when treated with oral bisphosphonates, even though only a small population was enrolled. We cannot draw a definite conclusion that the increase in BMD can be translated into fracture reduction and clinical functional improvement. The optimal method, dose, type, initiation, and duration of oral bisphosphonates therapy still remains unclear. Well‐designed, adequately‐powered, placebo‐controlled RCTs investigating the effects of oral bisphosphonates on fractures reduction and improvement in quality of life in both children and adults are studied here.https://doi.org/10.1111/os.12611AlendronateClodronateEtidronateOral bisphosphonatesEsteogenesis imperfecta
spellingShingle Zhi‐min Ying
Bin Hu
Shi‐gui Yan
Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled Trials
Orthopaedic Surgery
Alendronate
Clodronate
Etidronate
Oral bisphosphonates
Esteogenesis imperfecta
title Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled Trials
title_full Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled Trials
title_fullStr Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled Trials
title_full_unstemmed Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled Trials
title_short Oral Bisphosphonate Therapy for Osteogenesis Imperfecta: A Systematic Review and Meta‐Analysis of Six Randomized Placebo‐Controlled Trials
title_sort oral bisphosphonate therapy for osteogenesis imperfecta a systematic review and meta analysis of six randomized placebo controlled trials
topic Alendronate
Clodronate
Etidronate
Oral bisphosphonates
Esteogenesis imperfecta
url https://doi.org/10.1111/os.12611
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AT binhu oralbisphosphonatetherapyforosteogenesisimperfectaasystematicreviewandmetaanalysisofsixrandomizedplacebocontrolledtrials
AT shiguiyan oralbisphosphonatetherapyforosteogenesisimperfectaasystematicreviewandmetaanalysisofsixrandomizedplacebocontrolledtrials