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The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study

BACKGROUND: The Hemophilia Joint Health Score (HJHS) was developed and validated to detect arthropathy in children. Additional evidence is required to show validity in adults. We studied the convergent and discriminant construct validity of the HJHS version 2.1(HJHSv2.1) in adults with hemophilia. A...

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Autores principales: St‐Louis, Jean, Abad, Audrey, Funk, Sharon, Tilak, Merlyn, Classey, Stephen, Zourikian, Nichan, McLaughlin, Paul, Lobet, Sébastien, Hernandez, Grace, Akins, Stacie, Wells, Anna J., Manco‐Johnson, Marilyn, John, Judy, Austin, Steve, Chowdhary, Pratima, Hermans, Cedric, Nugent, Diane, Bakeer, Nihal, Mangles, Sarah, Hilliard, Pamela, Blanchette, Victor S., Feldman, Brian M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8956786/
https://www.ncbi.nlm.nih.gov/pubmed/35356667
http://dx.doi.org/10.1002/rth2.12690
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author St‐Louis, Jean
Abad, Audrey
Funk, Sharon
Tilak, Merlyn
Classey, Stephen
Zourikian, Nichan
McLaughlin, Paul
Lobet, Sébastien
Hernandez, Grace
Akins, Stacie
Wells, Anna J.
Manco‐Johnson, Marilyn
John, Judy
Austin, Steve
Chowdhary, Pratima
Hermans, Cedric
Nugent, Diane
Bakeer, Nihal
Mangles, Sarah
Hilliard, Pamela
Blanchette, Victor S.
Feldman, Brian M.
author_facet St‐Louis, Jean
Abad, Audrey
Funk, Sharon
Tilak, Merlyn
Classey, Stephen
Zourikian, Nichan
McLaughlin, Paul
Lobet, Sébastien
Hernandez, Grace
Akins, Stacie
Wells, Anna J.
Manco‐Johnson, Marilyn
John, Judy
Austin, Steve
Chowdhary, Pratima
Hermans, Cedric
Nugent, Diane
Bakeer, Nihal
Mangles, Sarah
Hilliard, Pamela
Blanchette, Victor S.
Feldman, Brian M.
author_sort St‐Louis, Jean
collection PubMed
description BACKGROUND: The Hemophilia Joint Health Score (HJHS) was developed and validated to detect arthropathy in children. Additional evidence is required to show validity in adults. We studied the convergent and discriminant construct validity of the HJHS version 2.1(HJHSv2.1) in adults with hemophilia. A secondary aim was to define age‐related normative adult HJHSv2.1 reference values. METHODS: We studied 192 adults with hemophilia, and 120 healthy adults in four age‐matched groups—18 to 29, 30 to 40, 41 to 50, and >50 years—at nine centers. Trained physiotherapists scored the HJHS and World Federation of Hemophilia (WFH) joint score. Health history, the Functional Independence Scale of Hemophilia (FISH), Hemophilia Activities List (HAL), and Short‐Form McGill Pain Questionnaire (SF‐MPQ) were also collected. RESULTS: The median age was 35.0 years. Of participants with hemophilia, 68% had severe, 14% moderate, and 18% mild disease. The HJHS correlated strongly with WFH score (Spearman’s rho [r(s) ] = .95, P < .001). Moderate correlations were seen between the FISH (r(s)  = .50, P < .001) and SF‐MPQ Present Pain Intensity (r(s)  = .50, P < .001), while a modest correlation was found with the HAL (r(s)  = −.37, P < .001). The HJHS significantly differentiated between age groups (Kruskal‐Wallis T = 35.02, P < .001) and disease severity in participants with hemophilia. The HJHS had high internal reliability (Cronbach’s α = .88). We identified duration of swelling as a redundant item in the HJHS. CONCLUSIONS: The HJHS shows evidence of strong convergent and discriminant construct validity to detect arthropathy in adults with hemophilia and is well suited for use in this population.
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spelling pubmed-89567862022-03-29 The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study St‐Louis, Jean Abad, Audrey Funk, Sharon Tilak, Merlyn Classey, Stephen Zourikian, Nichan McLaughlin, Paul Lobet, Sébastien Hernandez, Grace Akins, Stacie Wells, Anna J. Manco‐Johnson, Marilyn John, Judy Austin, Steve Chowdhary, Pratima Hermans, Cedric Nugent, Diane Bakeer, Nihal Mangles, Sarah Hilliard, Pamela Blanchette, Victor S. Feldman, Brian M. Res Pract Thromb Haemost Original Articles BACKGROUND: The Hemophilia Joint Health Score (HJHS) was developed and validated to detect arthropathy in children. Additional evidence is required to show validity in adults. We studied the convergent and discriminant construct validity of the HJHS version 2.1(HJHSv2.1) in adults with hemophilia. A secondary aim was to define age‐related normative adult HJHSv2.1 reference values. METHODS: We studied 192 adults with hemophilia, and 120 healthy adults in four age‐matched groups—18 to 29, 30 to 40, 41 to 50, and >50 years—at nine centers. Trained physiotherapists scored the HJHS and World Federation of Hemophilia (WFH) joint score. Health history, the Functional Independence Scale of Hemophilia (FISH), Hemophilia Activities List (HAL), and Short‐Form McGill Pain Questionnaire (SF‐MPQ) were also collected. RESULTS: The median age was 35.0 years. Of participants with hemophilia, 68% had severe, 14% moderate, and 18% mild disease. The HJHS correlated strongly with WFH score (Spearman’s rho [r(s) ] = .95, P < .001). Moderate correlations were seen between the FISH (r(s)  = .50, P < .001) and SF‐MPQ Present Pain Intensity (r(s)  = .50, P < .001), while a modest correlation was found with the HAL (r(s)  = −.37, P < .001). The HJHS significantly differentiated between age groups (Kruskal‐Wallis T = 35.02, P < .001) and disease severity in participants with hemophilia. The HJHS had high internal reliability (Cronbach’s α = .88). We identified duration of swelling as a redundant item in the HJHS. CONCLUSIONS: The HJHS shows evidence of strong convergent and discriminant construct validity to detect arthropathy in adults with hemophilia and is well suited for use in this population. John Wiley and Sons Inc. 2022-03-25 /pmc/articles/PMC8956786/ /pubmed/35356667 http://dx.doi.org/10.1002/rth2.12690 Text en © 2022 The Authors. Research and Practice in Thrombosis and Haemostasis published by Wiley Periodicals LLC on behalf of International Society on Thrombosis and Haemostasis (ISTH). https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.
spellingShingle Original Articles
St‐Louis, Jean
Abad, Audrey
Funk, Sharon
Tilak, Merlyn
Classey, Stephen
Zourikian, Nichan
McLaughlin, Paul
Lobet, Sébastien
Hernandez, Grace
Akins, Stacie
Wells, Anna J.
Manco‐Johnson, Marilyn
John, Judy
Austin, Steve
Chowdhary, Pratima
Hermans, Cedric
Nugent, Diane
Bakeer, Nihal
Mangles, Sarah
Hilliard, Pamela
Blanchette, Victor S.
Feldman, Brian M.
The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study
title The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study
title_full The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study
title_fullStr The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study
title_full_unstemmed The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study
title_short The Hemophilia Joint Health Score version 2.1 Validation in Adult Patients Study: A multicenter international study
title_sort hemophilia joint health score version 2.1 validation in adult patients study: a multicenter international study
topic Original Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8956786/
https://www.ncbi.nlm.nih.gov/pubmed/35356667
http://dx.doi.org/10.1002/rth2.12690
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