Rates and Pattern of Antifracture Drug Use in 6475 Adults and Children With Osteogenesis Imperfecta

Osteogenesis imperfecta (OI) is a genetic disorder characterized by increased bone fragility. Despite this, the use of antifracture drugs (AFD) among people with OI in the United States is not known.

This study examined the rates and types of AFD use in OI and whether use patterns varied with age, sex, and clinical severity.

We conducted a cohort study using medical and prescription claims data from January 1, 2011, to December 31, 2022, in MarketScan and from January 1, 2006, to December 31, 2021, in a 5% Medicare sample. Durable medical equipment claims for long-term wheelchair were used to define severe OI. Each individual with OI was matched to 5 reference patients within the same database by year of birth, sex, race, index year, and baseline enrollment duration.

We identified 6494 people with OI and 32,470 individuals. The median [interquartile range] age was 21 years [8-43]. Adults comprised 55.8% of cohort; 57.6% were female. AFD use was 25.9% in the overall OI cohort and was higher in the severe OI group than in the milder group (38.3% vs 22.9%, respectively; standard mean difference 0.34). Use was highest in children, was much lower in young adults, and rose gradually with increasing age. Bisphosphonates were the most prescribed AFD across all age groups and OI severity levels.

These findings suggest that AFDs drugs are underutilized in the general US population of adults and children with OI. Our observed rates are lower than those reported in other studies, likely because our cohort reflects individuals in the general community rather than patients treated in specialized bone clinics.