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

Autor: Constantino CS; Orthopedic and Rehabilitation Engineering Center, Marquette University & Medical College of Wisconsin Milwaukee Wisconsin USA., Krzak JJ; Motion Analysis Laboratory, Shriners Hospital for Children Chicago Illinois USA.; Midwestern University, Physical Therapy Program, College of Health Sciences Downers Grove Illinois USA., Fial AV; Research and Instructional Services, Raynor Memorial Libraries, Marquette University Milwaukee Wisconsin USA., Kruger KM; Orthopedic and Rehabilitation Engineering Center, Marquette University & Medical College of Wisconsin Milwaukee Wisconsin USA.; Motion Analysis Laboratory, Shriners Hospital for Children Chicago Illinois USA., Rammer JR; Orthopedic and Rehabilitation Engineering Center, Marquette University & Medical College of Wisconsin Milwaukee Wisconsin USA., Radmanovic K; Orthopedic and Rehabilitation Engineering Center, Marquette University & Medical College of Wisconsin Milwaukee Wisconsin USA., Smith PA; Motion Analysis Laboratory, Shriners Hospital for Children Chicago Illinois USA., Harris GF; Orthopedic and Rehabilitation Engineering Center, Marquette University & Medical College of Wisconsin Milwaukee Wisconsin USA.; Motion Analysis Laboratory, Shriners Hospital for Children Chicago Illinois USA.
Jazyk: angličtina
Zdroj: JBMR plus [JBMR Plus] 2019 Oct 18; Vol. 3 (10), pp. e10216. Date of Electronic Publication: 2019 Oct 18 (Print Publication: 2019).
DOI: 10.1002/jbm4.10216
Abstrakt: 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.
(© 2019 American Society for Bone and Mineral Research.)
Databáze: MEDLINE
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