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Evidence of a disability paradox in patient‐reported outcomes in haemophilia
- Source :
- Haemophilia
- Publication Year :
- 2021
- Publisher :
- Wiley, 2021.
-
Abstract
- IntroductionPeople with inherited and long-term conditions such as haemophilia have been shown to adapt to their levels of disability, often reporting better quality of life (QoL) than expected from the general population (the disability paradox).AimTo investigate the disability paradox in people with haemophilia in the United States by examining preference differences in health state valuations versus the general population.MethodsWe conducted a discrete choice experiment including duration to capture valuations of health states based on patient-reported preferences. Participants indicated their preferences for hypothetical health states using the EQ-5D-5L, where each participant completed 15 of the 120 choice tasks. Response inconsistencies were evaluated with dominated and repeated scenarios. Conditional-logit regressions with random sampling of the general population responses were used to match the sample of patients with haemophilia. We compared model estimates and derived preferences associated with EQ-5D-5L health states.ResultsAfter removing respondents with response inconsistencies, 1327/2138 (62%) participants remained (177/283 haemophilia; 1150/1900 general population). Patients with haemophilia indicated higher preference value for 99% of EQ-5D-5L health states compared to the general population (when matched on age and gender). The mean health state valuation difference of 0.17 indicated a meaningful difference compared to a minimal clinically important difference threshold of 0.07. Results were consistent by haemophilia type and severity.ConclusionOur findings indicated the presence of a disability paradox among patients with haemophilia, who reported higher health states than the general population, suggesting the impact of haemophilia may be underestimated if general population value sets are used.
- Subjects :
- Cost effectiveness
Health Status
Population
haemophilia
Sample (statistics)
030204 cardiovascular system & hematology
Hemophilia A
Haemophilia
03 medical and health sciences
0302 clinical medicine
Quality of life
Surveys and Questionnaires
medicine
Humans
Patient Reported Outcome Measures
Clinical Haemophilia
education
Genetics (clinical)
health equity
education.field_of_study
business.industry
Minimal clinically important difference
1103 Clinical Sciences
Original Articles
cost‐effectiveness
Hematology
General Medicine
medicine.disease
Preference
Health equity
quality of life
Cardiovascular System & Hematology
Original Article
business
patient‐reported outcome measurement
030215 immunology
Demography
Subjects
Details
- Language :
- English
- Database :
- OpenAIRE
- Journal :
- Haemophilia
- Accession number :
- edsair.doi.dedup.....908db512a5fae403b79fd4bd3ddadcfd