Loading…

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 people...

Full description

Saved in:
Bibliographic Details
Published in:Haemophilia : the official journal of the World Federation of Hemophilia 2021-03, Vol.27 (2), p.245-252
Main Authors: 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.
Format: Article
Language:English
Subjects:
Citations: Items that this one cites
Items that cite this one
Online Access:Get full text
Tags: Add Tag
No Tags, Be the first to tag this record!
cited_by cdi_FETCH-LOGICAL-c4438-30e931b33afeecbaeb23eb4f3a5bfbda1a7d049c92f4c3a08c3a9a3c753273a73
cites cdi_FETCH-LOGICAL-c4438-30e931b33afeecbaeb23eb4f3a5bfbda1a7d049c92f4c3a08c3a9a3c753273a73
container_end_page 252
container_issue 2
container_start_page 245
container_title Haemophilia : the official journal of the World Federation of Hemophilia
container_volume 27
creator 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.
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.
doi_str_mv 10.1111/hae.14278
format article
fullrecord <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_8048516</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2490606261</sourcerecordid><originalsourceid>FETCH-LOGICAL-c4438-30e931b33afeecbaeb23eb4f3a5bfbda1a7d049c92f4c3a08c3a9a3c753273a73</originalsourceid><addsrcrecordid>eNp1kctO3TAQhq0KVC7tghdAkdi0i4CvuWyQEDoFJASbdm1NnAnHKImDnQBn10fgGXkSfDiAAAkv7JHm8-_f_xCyw-g-i-tgDrjPJM-Lb2STiUylXLFsbVkrlhacZRtkK4RrSpngNPtONoRQpWKy2CQXs1tbY28wcU0CSW0DVLa14yIZwEPt7hPbx3K02I-P_x88Ds6PWCduGo3rMCzb8fnODfN4DX6Q9QbagD9fzm3y78_s7_Fpen55cnZ8dJ4aKUWRCoqlYJUQ0CCaCrDiAivZCFBVU9XAIK-pLE3JG2kE0CJuJQiTK8FzAbnYJocr3WGqOqxNdOeh1YO3HfiFdmD1x05v5_rK3eqCyiKGEwV-vQh4dzNhGHVng8G2hR7dFDSXJc1oxjMW0b1P6LWbfB-_p7miJY9ZMhWp3yvKeBeCx-bNDKN6OSUdY9LPU4rs7nv3b-TrWCJwsALubIuLr5X06dFsJfkEAQaeUQ</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2509235915</pqid></control><display><type>article</type><title>Evidence of a disability paradox in patient‐reported outcomes in haemophilia</title><source>Wiley-Blackwell Read &amp; Publish Collection</source><creator>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.</creator><creatorcontrib>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.</creatorcontrib><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.</description><identifier>ISSN: 1351-8216</identifier><identifier>EISSN: 1365-2516</identifier><identifier>DOI: 10.1111/hae.14278</identifier><identifier>PMID: 33595148</identifier><language>eng</language><publisher>England: Wiley Subscription Services, Inc</publisher><subject>cost‐effectiveness ; haemophilia ; health equity ; Hemophilia ; Original ; Patients ; patient‐reported outcome measurement ; Population ; Preferences ; Quality of life ; Statistical sampling</subject><ispartof>Haemophilia : the official journal of the World Federation of Hemophilia, 2021-03, Vol.27 (2), p.245-252</ispartof><rights>2021 The Authors. published by John Wiley &amp; Sons Ltd.</rights><rights>2021 The Authors. Haemophilia published by John Wiley &amp; Sons Ltd.</rights><rights>2021. This article is published under http://creativecommons.org/licenses/by-nc/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4438-30e931b33afeecbaeb23eb4f3a5bfbda1a7d049c92f4c3a08c3a9a3c753273a73</citedby><cites>FETCH-LOGICAL-c4438-30e931b33afeecbaeb23eb4f3a5bfbda1a7d049c92f4c3a08c3a9a3c753273a73</cites><orcidid>0000-0003-2014-3415 ; 0000-0002-0934-0680 ; 0000-0003-0758-286X</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>230,314,780,784,885,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/33595148$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>O’Hara, Jamie</creatorcontrib><creatorcontrib>Martin, Antony P.</creatorcontrib><creatorcontrib>Nugent, Diane</creatorcontrib><creatorcontrib>Witkop, Michelle</creatorcontrib><creatorcontrib>Buckner, Tyler W.</creatorcontrib><creatorcontrib>Skinner, Mark W.</creatorcontrib><creatorcontrib>O’Mahony, Brian</creatorcontrib><creatorcontrib>Mulhern, Brendan</creatorcontrib><creatorcontrib>Morgan, George</creatorcontrib><creatorcontrib>Li, Nanxin</creatorcontrib><creatorcontrib>Sawyer, Eileen K.</creatorcontrib><title>Evidence of a disability paradox in patient‐reported outcomes in haemophilia</title><title>Haemophilia : the official journal of the World Federation of Hemophilia</title><addtitle>Haemophilia</addtitle><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.</description><subject>cost‐effectiveness</subject><subject>haemophilia</subject><subject>health equity</subject><subject>Hemophilia</subject><subject>Original</subject><subject>Patients</subject><subject>patient‐reported outcome measurement</subject><subject>Population</subject><subject>Preferences</subject><subject>Quality of life</subject><subject>Statistical sampling</subject><issn>1351-8216</issn><issn>1365-2516</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><sourceid>24P</sourceid><recordid>eNp1kctO3TAQhq0KVC7tghdAkdi0i4CvuWyQEDoFJASbdm1NnAnHKImDnQBn10fgGXkSfDiAAAkv7JHm8-_f_xCyw-g-i-tgDrjPJM-Lb2STiUylXLFsbVkrlhacZRtkK4RrSpngNPtONoRQpWKy2CQXs1tbY28wcU0CSW0DVLa14yIZwEPt7hPbx3K02I-P_x88Ds6PWCduGo3rMCzb8fnODfN4DX6Q9QbagD9fzm3y78_s7_Fpen55cnZ8dJ4aKUWRCoqlYJUQ0CCaCrDiAivZCFBVU9XAIK-pLE3JG2kE0CJuJQiTK8FzAbnYJocr3WGqOqxNdOeh1YO3HfiFdmD1x05v5_rK3eqCyiKGEwV-vQh4dzNhGHVng8G2hR7dFDSXJc1oxjMW0b1P6LWbfB-_p7miJY9ZMhWp3yvKeBeCx-bNDKN6OSUdY9LPU4rs7nv3b-TrWCJwsALubIuLr5X06dFsJfkEAQaeUQ</recordid><startdate>202103</startdate><enddate>202103</enddate><creator>O’Hara, Jamie</creator><creator>Martin, Antony P.</creator><creator>Nugent, Diane</creator><creator>Witkop, Michelle</creator><creator>Buckner, Tyler W.</creator><creator>Skinner, Mark W.</creator><creator>O’Mahony, Brian</creator><creator>Mulhern, Brendan</creator><creator>Morgan, George</creator><creator>Li, Nanxin</creator><creator>Sawyer, Eileen K.</creator><general>Wiley Subscription Services, Inc</general><general>John Wiley and Sons Inc</general><scope>24P</scope><scope>WIN</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7T5</scope><scope>8FD</scope><scope>FR3</scope><scope>H94</scope><scope>P64</scope><scope>RC3</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0003-2014-3415</orcidid><orcidid>https://orcid.org/0000-0002-0934-0680</orcidid><orcidid>https://orcid.org/0000-0003-0758-286X</orcidid></search><sort><creationdate>202103</creationdate><title>Evidence of a disability paradox in patient‐reported outcomes in haemophilia</title><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></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4438-30e931b33afeecbaeb23eb4f3a5bfbda1a7d049c92f4c3a08c3a9a3c753273a73</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>cost‐effectiveness</topic><topic>haemophilia</topic><topic>health equity</topic><topic>Hemophilia</topic><topic>Original</topic><topic>Patients</topic><topic>patient‐reported outcome measurement</topic><topic>Population</topic><topic>Preferences</topic><topic>Quality of life</topic><topic>Statistical sampling</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>O’Hara, Jamie</creatorcontrib><creatorcontrib>Martin, Antony P.</creatorcontrib><creatorcontrib>Nugent, Diane</creatorcontrib><creatorcontrib>Witkop, Michelle</creatorcontrib><creatorcontrib>Buckner, Tyler W.</creatorcontrib><creatorcontrib>Skinner, Mark W.</creatorcontrib><creatorcontrib>O’Mahony, Brian</creatorcontrib><creatorcontrib>Mulhern, Brendan</creatorcontrib><creatorcontrib>Morgan, George</creatorcontrib><creatorcontrib>Li, Nanxin</creatorcontrib><creatorcontrib>Sawyer, Eileen K.</creatorcontrib><collection>Wiley Open Access</collection><collection>Wiley Open Access</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Technology Research Database</collection><collection>Engineering Research Database</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>Biotechnology and BioEngineering Abstracts</collection><collection>Genetics Abstracts</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Haemophilia : the official journal of the World Federation of Hemophilia</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>O’Hara, Jamie</au><au>Martin, Antony P.</au><au>Nugent, Diane</au><au>Witkop, Michelle</au><au>Buckner, Tyler W.</au><au>Skinner, Mark W.</au><au>O’Mahony, Brian</au><au>Mulhern, Brendan</au><au>Morgan, George</au><au>Li, Nanxin</au><au>Sawyer, Eileen K.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Evidence of a disability paradox in patient‐reported outcomes in haemophilia</atitle><jtitle>Haemophilia : the official journal of the World Federation of Hemophilia</jtitle><addtitle>Haemophilia</addtitle><date>2021-03</date><risdate>2021</risdate><volume>27</volume><issue>2</issue><spage>245</spage><epage>252</epage><pages>245-252</pages><issn>1351-8216</issn><eissn>1365-2516</eissn><abstract>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.</abstract><cop>England</cop><pub>Wiley Subscription Services, Inc</pub><pmid>33595148</pmid><doi>10.1111/hae.14278</doi><tpages>8</tpages><orcidid>https://orcid.org/0000-0003-2014-3415</orcidid><orcidid>https://orcid.org/0000-0002-0934-0680</orcidid><orcidid>https://orcid.org/0000-0003-0758-286X</orcidid><oa>free_for_read</oa></addata></record>
fulltext fulltext
identifier ISSN: 1351-8216
ispartof Haemophilia : the official journal of the World Federation of Hemophilia, 2021-03, Vol.27 (2), p.245-252
issn 1351-8216
1365-2516
language eng
recordid cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_8048516
source Wiley-Blackwell Read & Publish Collection
subjects cost‐effectiveness
haemophilia
health equity
Hemophilia
Original
Patients
patient‐reported outcome measurement
Population
Preferences
Quality of life
Statistical sampling
title Evidence of a disability paradox in patient‐reported outcomes in haemophilia
url http://sfxeu10.hosted.exlibrisgroup.com/loughborough?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-12-24T19%3A58%3A23IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_pubme&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Evidence%20of%20a%20disability%20paradox%20in%20patient%E2%80%90reported%20outcomes%20in%20haemophilia&rft.jtitle=Haemophilia%20:%20the%20official%20journal%20of%20the%20World%20Federation%20of%20Hemophilia&rft.au=O%E2%80%99Hara,%20Jamie&rft.date=2021-03&rft.volume=27&rft.issue=2&rft.spage=245&rft.epage=252&rft.pages=245-252&rft.issn=1351-8216&rft.eissn=1365-2516&rft_id=info:doi/10.1111/hae.14278&rft_dat=%3Cproquest_pubme%3E2490606261%3C/proquest_pubme%3E%3Cgrp_id%3Ecdi_FETCH-LOGICAL-c4438-30e931b33afeecbaeb23eb4f3a5bfbda1a7d049c92f4c3a08c3a9a3c753273a73%3C/grp_id%3E%3Coa%3E%3C/oa%3E%3Curl%3E%3C/url%3E&rft_id=info:oai/&rft_pqid=2509235915&rft_id=info:pmid/33595148&rfr_iscdi=true