Affordable Access

deepdyve-link
Publisher Website

Effect of Bisphosphonates on Function and Mobility Among Children With Osteogenesis Imperfecta: A Systematic Review

Authors
  • Constantino, Christopher S.1
  • Krzak, Joseph J.2, 3
  • Fial, Alissa V.4
  • Kruger, Karen M.1, 2
  • Rammer, Jacob R.1
  • Radmanovic, Katarina1
  • Smith, Peter A.2
  • Harris, Gerald F.1, 2
  • 1 Orthopedic and Rehabilitation Engineering Center, Marquette University & Medical College of Wisconsin, USA , (United States)
  • 2 Motion Analysis Laboratory, Shriners Hospital for Children, USA , (United States)
  • 3 Midwestern University, Physical Therapy Program, College of Health Sciences, USA , (United States)
  • 4 Research and Instructional Services, Raynor Memorial Libraries, Marquette University, USA , (United States)
Type
Published Article
Journal
JBMR Plus
Publisher
John Wiley and Sons Inc.
Publication Date
Oct 18, 2019
Volume
3
Issue
10
Identifiers
DOI: 10.1002/jbm4.10216
PMID: 31687649
PMCID: PMC6820458
Source
PubMed Central
Keywords
License
Unknown
External links

Abstract

Osteogenesis imperfecta (OI) is a rare genetic connective tissue disorder that results in bone fragility and deformity. Management is multi‐disciplinary. Although pharmacologic intervention with bisphosphonates (BP) is a standard of care for individuals with severe OI, no consensus or reviews were found that focus on the effects of bisphosphonates on function and mobility. PubMed, CINAHL, Cochrane Library, Web of Science, and PEDro databases were searched for eligible articles for this review. Methodological quality was assessed using the Cochrane Collaboration's tool for risk of bias. Twenty‐six studies (801 children) were reviewed and five showed a low risk of bias. Included studies showed significant variability among clinical protocols for administering BP. Randomized controlled trials did not demonstrate a significant improvement in function and mobility with oral BP administration, while non‐randomized open‐label uncontrolled studies demonstrated that oral and intravenous BP administration objectively improved function and mobility. The most common outcome measure used by the studies included in this review was the Bleck score. Effect sizes (d = 0.28 ‐ 4.5) varied among studies. This systematic review also summarized the apparent confounding variables affecting results of previous studies and provided suggestions to improve the quality of future studies.

Report this publication

Statistics

Seen <100 times