The IMPACT Survey: the economic impact of osteogenesis imperfecta in adults

The IMPACT survey aimed to elucidate the humanistic, clinical and economic burden of osteogenesis imperfecta (OI) on individuals with OI, their families, caregivers and wider society. Research methodology, demographics and initial insights from the survey have been previously reported. The cost of illness (healthcare resource use, productivity loss, out-of-pocket spending) and drivers of the economic impact of OI are reported here. IMPACT was an international mixed-methods online survey in eight languages (fielded July–September 2021) targeting adults (aged ≥ 18 years) or adolescents (aged ≥ 12–17 years) with OI, caregivers with or without OI and other close relatives. Survey domains included demographics, socioeconomic factors, clinical characteristics, treatment patterns, quality of life and health economics. The health economic domain for adults, which included questions on healthcare resource use, productivity loss and out-of-pocket spending, was summarised. Regression and pairwise analyses were conducted to identify independent drivers and associations with respondent characteristics. Overall, 1,440 adults with OI responded to the survey. Respondents were mostly female (70