Comparing the Humanistic and Economic Impact of Osteogenesis Imperfecta Affected and Non-Affected Caregivers, When Caring for an Individual With Osteogenesis Imperfecta

Speaker(s)

Prince S1, van Welzenis T2, Wekre LL3, Westerheim I2, Raggio C4, Semler O5, Hart T6, Mulhall H1, Rauch F7
1Wickenstones Ltd, Oxford, UK, 2Osteogenesis Imperfecta Federation Europe, Heffen, Belgium, 3Sunnaas Rehabilitation Hospital, Nesoddtangen, Bjørnemyr, Norway, 4Hospital for Special Surgery, New York, NY, USA, 5University Hospital Cologne, Cologne, Germany, 6Osteogenesis Imperfecta Foundation, Gaithersburg, MD, USA, 7McGill University Hospital, Montreal, QC, Canada

Presentation Documents

OBJECTIVES: The humanistic and economic impact of osteogenesis imperfecta (OI) — a rare hereditary connective tissue disorder — on caregivers (CGs) of individuals with OI, who have OI themselves, is under-researched. This analysis seeks to quantify these impacts and compare to CGs of individuals with OI, who do not have OI themselves.

METHODS: The IMPACT Survey (fielded online July–September 2021 in eight languages) was developed with the OI Federation Europe and the OI Foundation (USA). It explored the self-reported clinical, humanistic, and economic impact of OI. The survey was open to adults (≥18 years) and adolescents (12–17 years) with OI, CGs (with or without OI) of individuals with OI, and other close relatives. Data were cleaned, coded, and analyzed using Microsoft Excel.

RESULTS: A total of 150 CGs with OI completed the survey (mean age 42 years; 72% female), with mild (50%), moderate (43%), or severe (5%) self-reported OI severity. Across 10 quality of life (QoL) areas, 39–85% of CGs with OI were impacted, with 3–25% experiencing severe impact. The most commonly impacted areas were leisure activity type (85%), happiness (71%), and job type (71%). Due to OI, 40–97% of CGs with OI worried across areas of their lives or those of their care recipients. Areas where the highest proportions of CGs with OI worried were their care recipients’ futures (97%), fractures (97%), and getting older (95%). CGs with OI also experienced impacted productivity and finances, missing 1.5 workdays (mean) and spending €95 out-of-pocket (mean) over 4 weeks due to OI. Compared with CGs with OI, CGs without OI (n=560) experienced similar levels of worry and QoL impact, missed more workdays (mean 1.9), and spent more out-of-pocket (mean €208) in 4 weeks.

CONCLUSIONS: CGs with OI experience impacted QoL, productivity, and finances due to their OI and their caregiving responsibilities.

Code

PCR184

Topic

Patient-Centered Research, Study Approaches

Topic Subcategory

Patient-reported Outcomes & Quality of Life Outcomes, Surveys & Expert Panels

Disease

Musculoskeletal Disorders (Arthritis, Bone Disorders, Osteoporosis, Other Musculoskeletal), Rare & Orphan Diseases