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Evidence of a disability paradox in patient‐reported outcomes in haemophilia

INTRODUCTION: People 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). AIM: To investigate the disability paradox in peop...

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Autores principales: O’Hara, Jamie, Martin, Antony P., Nugent, Diane, Witkop, Michelle, Buckner, Tyler W., Skinner, Mark W., O’Mahony, Brian, Mulhern, Brendan, Morgan, George, Li, Nanxin, Sawyer, Eileen K.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8048516/
https://www.ncbi.nlm.nih.gov/pubmed/33595148
http://dx.doi.org/10.1111/hae.14278
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author O’Hara, Jamie
Martin, Antony P.
Nugent, Diane
Witkop, Michelle
Buckner, Tyler W.
Skinner, Mark W.
O’Mahony, Brian
Mulhern, Brendan
Morgan, George
Li, Nanxin
Sawyer, Eileen K.
author_facet O’Hara, Jamie
Martin, Antony P.
Nugent, Diane
Witkop, Michelle
Buckner, Tyler W.
Skinner, Mark W.
O’Mahony, Brian
Mulhern, Brendan
Morgan, George
Li, Nanxin
Sawyer, Eileen K.
author_sort O’Hara, Jamie
collection PubMed
description INTRODUCTION: People 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). AIM: To investigate the disability paradox in people with haemophilia in the United States by examining preference differences in health state valuations versus the general population. METHODS: We 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. RESULTS: After 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. CONCLUSION: Our 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.
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spelling pubmed-80485162021-04-16 Evidence of a disability paradox in patient‐reported outcomes in haemophilia O’Hara, Jamie Martin, Antony P. Nugent, Diane Witkop, Michelle Buckner, Tyler W. Skinner, Mark W. O’Mahony, Brian Mulhern, Brendan Morgan, George Li, Nanxin Sawyer, Eileen K. Haemophilia Original Articles INTRODUCTION: People 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). AIM: To investigate the disability paradox in people with haemophilia in the United States by examining preference differences in health state valuations versus the general population. METHODS: We 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. RESULTS: After 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. CONCLUSION: Our 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. John Wiley and Sons Inc. 2021-02-17 2021-03 /pmc/articles/PMC8048516/ /pubmed/33595148 http://dx.doi.org/10.1111/hae.14278 Text en © 2021 The Authors. Haemophilia published by John Wiley & Sons Ltd. https://creativecommons.org/licenses/by-nc/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by-nc/4.0/ (https://creativecommons.org/licenses/by-nc/4.0/) License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited and is not used for commercial purposes.
spellingShingle Original Articles
O’Hara, Jamie
Martin, Antony P.
Nugent, Diane
Witkop, Michelle
Buckner, Tyler W.
Skinner, Mark W.
O’Mahony, Brian
Mulhern, Brendan
Morgan, George
Li, Nanxin
Sawyer, Eileen K.
Evidence of a disability paradox in patient‐reported outcomes in haemophilia
title Evidence of a disability paradox in patient‐reported outcomes in haemophilia
title_full Evidence of a disability paradox in patient‐reported outcomes in haemophilia
title_fullStr Evidence of a disability paradox in patient‐reported outcomes in haemophilia
title_full_unstemmed Evidence of a disability paradox in patient‐reported outcomes in haemophilia
title_short Evidence of a disability paradox in patient‐reported outcomes in haemophilia
title_sort evidence of a disability paradox in patient‐reported outcomes in haemophilia
topic Original Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8048516/
https://www.ncbi.nlm.nih.gov/pubmed/33595148
http://dx.doi.org/10.1111/hae.14278
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