OBJECTIVES: To evaluate the cost of lost productivity for adults with Osteogenesis imperfecta (OI) using the valuation of lost productivity (VOLP) questionnaire.

METHODS: We extracted data from RUDY, an online rare disease cohort study in the UK, from 2020 to 2021. Participants completed the VOLP questionnaire which measures absenteeism, presenteeism, and unemployment due to ill health, lost unpaid work, and the monetary cost of lost productivity to society over the three months prior. Participants were invited to complete a baseline, and then follow-up questionnaires every three months. Inclusion criteria were adults self-reporting with OI with a completed VOLP questionnaire at baseline and follow-up. Lost productivity due to ill-health was estimated as unpaid plus paid productivity loss, the latter made up of unemployment caused by ill-health and absenteeism.

RESULTS: The sample comprised of 12 participants, of which 5 were employed and, of the 7 unemployed, 3 reported being so due to ill-health. Of the recorded responses, many did not report any absenteeism or presenteeism at follow-up. Over the three-month follow-up period, mean unpaid productivity loss was £632 per participant, with an average loss of 6 unpaid work hours per week. Mean paid work productivity loss was £2,641, of which £2,304 were due to unemployment caused by ill-health and £337 by absenteeism. Mean total cost of lost productivity was £3,273 per participant diagnosed with OI over three months.

CONCLUSIONS: From these preliminary data, productivity loss for adults with osteogenesis imperfecta is significant and mainly driven by unemployment caused by ill-health. These costs should be included in economic evaluations of future treatments for the disease. Further work with larger samples is required to confirm these findings.