A further prospective study of denosumab 1 mg/kg s/c 12 weekly in 10 children with mutations in COL1A1 or COL1A2 was reported in 2016 and found significant increases in LSaBMD Z-score – + 0.96SDS at 48 weeks—but no change in vertebral morphometry or functional outcomes, although there was a strong trend towards an improved six minute walk test in those who were mobile.
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Medical Management for Fracture Prevention in Children with Osteogenesis Imperfecta.
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