Investigating the psychometric properties of the EQ-5D-Y-3L, EQ-5D-Y-5L, CHU-9D, and PedsQL in children and adolescents with osteogenesis imperfecta

The objective of this study was to evaluate and compare the psychometric properties of the EQ-5D-Y-3L, EQ-5D-Y-5L, CHU-9D, and PedsQL, in a sample of children and adolescents with osteogenesis imperfecta (OI). A web-based cross-sectional survey was conducted among Chinese children and adolescents with OI in 2021. The EQ-5D-Y-3L, EQ-5D-Y-5L, CHU-9D, and PedsQL were used to assess the health-related quality of life for the participants. Construct validity, including convergent and divergent validity, known-group validity, and test–retest reliability, was examined to assess the psychometric properties of the measures. A total of 157 pediatric OI patients self-completed the questionnaire. Few of them reported the full health status. A strong ceiling effect was observed for all dimensions on the EQ-5D-Y and most on CHU-9D. Most dimensions of the EQ-5D-Y and CHU-9D showed statistically significant correlations with the hypothesized PedsQL subscales. The test–retest reliability for the EQ-5D-Y-3L, EQ-5D-Y-5L, and CHU-9D was acceptable. The EQ-5D-Y-5L showed a better known-group validity than EQ-5D-Y-3L, CHU-9D, and PedsQL in differentiating patients in risk groups. Conclusion : The results confirmed that the EQ-5D-Y and CHU-9D are reliable and valid in pediatric OI patients. The EQ-5D-Y-5L performed better than EQ-5D-Y-3L regarding acceptability, convergent validity, and discriminatory power. What is Known: • Performance of the preference-based measures has never been reported in patients with Osteogenesis imperfecta. What is New: • The EQ-5D-Y demonstrated higher sensitivity and discriminatory power than the CHU-9D in patients with osteogenesis Imperfecta • The EQ-5D-Y-3L performed slightly better than EQ-5D-Y-5L regarding convergent validity and discriminant ability