This subproject is one of many research subprojects utilizing theresources provided by a Center grant funded by NIH/NCRR. The subproject andinvestigator (PI) may have received primary funding from another NIH source,and thus could be represented in other CRISP entries. The institution listed isfor the Center, which is not necessarily the institution for the investigator.This clinical research proposal describes a study to evaluate the efficacy and safety of Bisphosphonate Therapy for Osteogenesis Imperfecta (OI). OI is a heritable disorder of collagen synthesis. Affected persons have low bone mineral density and experience multiple fractures and progressive bony deformity. In its most severe form, the disorder is lethal in infancy. To date, no effective therapy exists for this debilitating condition. Improvements in bone mineral density and in fracture rates in a small number of children treated with intravenous bisphosphonates have been reported. The efficacy of oral bisphosphonates has not been established. We will characterize the changed effected by oral bisphosphonate therapy and compare them to a regimen of intravenous bisphosphonate therapy in a group of children with OI.
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