A new study published in the journal Haemophilia showed evidence of a disability paradox in patients with hemophilia. A disability paradox is a tendency for some patients to report superior quality of life (QoL) relative to the general population after adapting to a form of disability, the study investigators explained in their report.
The study employed an approach using a discrete choice experiment to assess health state valuations in patients with hemophilia, in which patients completed 15 out of 120 sets of choices. Valuations were based on attitudes around whether or not certain hypothetical scenarios were better than their alternatives.
Sets of choices were developed using the 5 dimensions of the EQ-5D-5L questionnaire, while also incorporating levels of severity and duration. The researchers analyzed questionnaire results for patients in comparison with those of members of the general population.
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A total of 1327 participants were evaluable after removing those with inconsistent responses. This total included 177 patients with hemophilia and 1150 from the general population.
Overall, participants with hemophilia responded with higher values than participants from the general population did for nearly all (98.9%) of the EQ-5D-5L states. There was a mean health state valuation difference of 0.17 between the 2 populations across all EQ-5D-5L states, which was considered clinically meaningful based on a minimal clinically important difference of 0.07.
Results were similar for patients regardless of type of hemophilia or severity of the condition, and severe hemophilia was associated with higher valuations in 95.4% of EQ-5D-5L states than seen in the general population. Responses indicated that mobility and self-care were areas that showed more substantial QOL impacts in participants with hemophilia.
“Our study revealed evidence of the disability paradox in haemophilia patient-reported
outcomes, where people with haemophilia reported significantly higher health state valuations than otherwise healthy peers from the general US population,” the study investigators wrote in their report.
The researchers suggested that clinical and health technology assessments of patients with hemophilia should consider the disability paradox in this population. They also concluded that the burden of hemophilia may be underestimated, and standard QoL tools may be inaccurate in these patients without consideration of this paradox, which may have implications concerning health policy.
Disclosures: Some authors have declared affiliations with or received grant support from the pharmaceutical industry. Please refer to the original study for a full list of authors’ disclosures.
Reference
O’Hara J, Martin AP, Nugent D, et al. Evidence of a disability paradox in patient-reported outcomes in haemophilia. Haemophilia. Published online February 17, 2021. doi:10.1111/hae.14278
