6 randomized controlled trials (RCTs) with a total of 424 subjects and 751 patient-years of follow-up were included in this meta-analysis. The purpose of this study was to determine the effectiveness of bisphosphonates in the prevention of fractures in patients with osteogenesis imperfecta. Pooled results demonstrated that bisphosphonates did not significantly reduce the proportion of patients with fracture, but may reduce the risk of fractures in osteogenesis imperfecta patients. These result should be interpreted with caution as there were few studies identified comparing bisphosphonates to placebo in this patient population, and the majority of these studies were powered to detect differences in bone mineral density and not incidence of fracture. Further, appropriately powered studies are need on this topic to provide clarity.