Validation of the pedHALshort and HALshort in Dutch children and adults with haemophilia

Isolde A R Kuijlaars; Janjaap van der Net; Lize F D van Vulpen; Mariette H E Driessens; Saskia E M Schols; Melanie Tan; Samantha C Gouw; Kathelijn Fischer

doi:10.1111/hae.14628

Validation of the pedHALshort and HALshort in Dutch children and adults with haemophilia

Isolde A R Kuijlaars, Janjaap van der Net, Lize F D van Vulpen, Mariette H E Driessens, Saskia E M Schols, Melanie Tan, Samantha C Gouw, Kathelijn Fischer

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Abstract

INTRODUCTION: The Haemophilia Activities List (HAL) and paediatric HAL assess self-reported limitations in various daily activities. To reduce patient burden, shorter versions of the pedHAL (22 items) and HAL (18 items) have been developed.

AIM: This study aimed to determine the agreement between the pedHAL/HAL full and pedHAL/HAL short and construct validity and internal consistency of the pedHAL / HAL short in persons with haemophilia (PWH).

METHODS: A cross-sectional secondary analysis of the Hemophilia in the Netherlands-6 national survey was performed. Adult and paediatric PWH completed the original pedHAL/HAL full , from which pedHAL / HAL short were derived. Score differences between the original and short versions were calculated. Construct validity was studied by testing hypotheses regarding the relationship of the pedHAL/HAL short with the pedHAL/HAL full , Haemophilia & Exercise Project Test-Questionnaire (HEP-Test-Q), Canadian Haemophilia Outcomes-Kids' Life Assessment Tool (CHO-KLAT) and RAND 36-item Health Survey (RAND-36) (convergent/discriminant validity) as well as its ability to discriminate between subgroups (known-group validity). Internal consistency was assessed with Cronbach's α.

RESULTS: We included 113 children (median 10y [range 4-17], 53% severe haemophilia) and 691 adults (median 51y [range 18-88], 35% severe). Scores of the pedHAL/HAL full and pedHAL/HAL short were similar with high correlations (>0.9). Construct validity was confirmed for the pedHAL/HAL short . The HAL short was able to discriminate between different disease severities and ages. Cronbach's α of the pedHAL/HAL short was 0.95-0.97.

CONCLUSION: This study confirmed the agreement between the pedHAL/HAL full and the pedHAL/HAL short and the construct validity of the pedHAL/HAL short . The next step is to study construct validity of the pedHAL/HAL short when administered as short forms.

Original language	English
Pages (from-to)	1007-1015
Number of pages	9
Journal	Haemophilia
Volume	28
Issue number	6
Early online date	29 Jul 2022
DOIs	https://doi.org/10.1111/hae.14628
Publication status	Published - Nov 2022

Keywords

activities
haemophilia
participation
patient-reported outcome
validity

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Haemophilia - 2022 - Kuijlaars - Validation of the pedHALshort and HALshort in Dutch children and adults with haemophiliaFinal published version, 545 KBLicence: CC BY-NC

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@article{348731fd49264d7eb5ab101bec237afc,

title = "Validation of the pedHALshort and HALshort in Dutch children and adults with haemophilia",

abstract = "INTRODUCTION: The Haemophilia Activities List (HAL) and paediatric HAL assess self-reported limitations in various daily activities. To reduce patient burden, shorter versions of the pedHAL (22 items) and HAL (18 items) have been developed.AIM: This study aimed to determine the agreement between the pedHAL/HAL full and pedHAL/HAL short and construct validity and internal consistency of the pedHAL / HAL short in persons with haemophilia (PWH). METHODS: A cross-sectional secondary analysis of the Hemophilia in the Netherlands-6 national survey was performed. Adult and paediatric PWH completed the original pedHAL/HAL full , from which pedHAL / HAL short were derived. Score differences between the original and short versions were calculated. Construct validity was studied by testing hypotheses regarding the relationship of the pedHAL/HAL short with the pedHAL/HAL full , Haemophilia & Exercise Project Test-Questionnaire (HEP-Test-Q), Canadian Haemophilia Outcomes-Kids' Life Assessment Tool (CHO-KLAT) and RAND 36-item Health Survey (RAND-36) (convergent/discriminant validity) as well as its ability to discriminate between subgroups (known-group validity). Internal consistency was assessed with Cronbach's α. RESULTS: We included 113 children (median 10y [range 4-17], 53% severe haemophilia) and 691 adults (median 51y [range 18-88], 35% severe). Scores of the pedHAL/HAL full and pedHAL/HAL short were similar with high correlations (>0.9). Construct validity was confirmed for the pedHAL/HAL short . The HAL short was able to discriminate between different disease severities and ages. Cronbach's α of the pedHAL/HAL short was 0.95-0.97. CONCLUSION: This study confirmed the agreement between the pedHAL/HAL full and the pedHAL/HAL short and the construct validity of the pedHAL/HAL short . The next step is to study construct validity of the pedHAL/HAL short when administered as short forms. ",

keywords = "activities, haemophilia, participation, patient-reported outcome, validity",

author = "Kuijlaars, {Isolde A R} and {van der Net}, Janjaap and {van Vulpen}, {Lize F D} and Driessens, {Mariette H E} and Schols, {Saskia E M} and Melanie Tan and Gouw, {Samantha C} and Kathelijn Fischer",

note = "Funding Information: I.A.R. Kuijlaars does not have any conflict of interest regarding this manuscript other than membership of the group that developed the HAL. L.F.D. van Vulpen has performed consultancy for Sobi, Tremeau, and C.S.L. Behring and received a research grant from CSL Behring and Grifols, all paid to the institution. S.E.M. Schols has received travel grants from Bayer and Takeda, consultancy grants from Takeda and Novo Nordisk and she has received a research grant from Bayer. S.C. Gouw has received an unrestricted research grant from Sobi. The Van Creveldkliniek has received speaker's fees from Bayer, Baxter/Shire, SOBI/Biogen, CSL Behring and NovoNordisk; consultancy fees from Bayer, Biogen, CSL‐Behring, Freeline, NovoNordisk, Roche and SOBI; and research support from Bayer, Baxter/Shire, Novo Nordisk, Pfizer and Biogen for work done by K. Fischer. The other authors have no competing interests. Publisher Copyright: {\textcopyright} 2022 The Authors. Haemophilia published by John Wiley & Sons Ltd.",

year = "2022",

month = nov,

doi = "10.1111/hae.14628",

language = "English",

volume = "28",

pages = "1007--1015",

journal = "Haemophilia",

issn = "1351-8216",

publisher = "Wiley-Blackwell",

number = "6",

}

TY - JOUR

T1 - Validation of the pedHALshort and HALshort in Dutch children and adults with haemophilia

AU - Kuijlaars, Isolde A R

AU - van der Net, Janjaap

AU - van Vulpen, Lize F D

AU - Driessens, Mariette H E

AU - Schols, Saskia E M

AU - Tan, Melanie

AU - Gouw, Samantha C

AU - Fischer, Kathelijn

N1 - Funding Information: I.A.R. Kuijlaars does not have any conflict of interest regarding this manuscript other than membership of the group that developed the HAL. L.F.D. van Vulpen has performed consultancy for Sobi, Tremeau, and C.S.L. Behring and received a research grant from CSL Behring and Grifols, all paid to the institution. S.E.M. Schols has received travel grants from Bayer and Takeda, consultancy grants from Takeda and Novo Nordisk and she has received a research grant from Bayer. S.C. Gouw has received an unrestricted research grant from Sobi. The Van Creveldkliniek has received speaker's fees from Bayer, Baxter/Shire, SOBI/Biogen, CSL Behring and NovoNordisk; consultancy fees from Bayer, Biogen, CSL‐Behring, Freeline, NovoNordisk, Roche and SOBI; and research support from Bayer, Baxter/Shire, Novo Nordisk, Pfizer and Biogen for work done by K. Fischer. The other authors have no competing interests. Publisher Copyright: © 2022 The Authors. Haemophilia published by John Wiley & Sons Ltd.

PY - 2022/11

Y1 - 2022/11

N2 - INTRODUCTION: The Haemophilia Activities List (HAL) and paediatric HAL assess self-reported limitations in various daily activities. To reduce patient burden, shorter versions of the pedHAL (22 items) and HAL (18 items) have been developed.AIM: This study aimed to determine the agreement between the pedHAL/HAL full and pedHAL/HAL short and construct validity and internal consistency of the pedHAL / HAL short in persons with haemophilia (PWH). METHODS: A cross-sectional secondary analysis of the Hemophilia in the Netherlands-6 national survey was performed. Adult and paediatric PWH completed the original pedHAL/HAL full , from which pedHAL / HAL short were derived. Score differences between the original and short versions were calculated. Construct validity was studied by testing hypotheses regarding the relationship of the pedHAL/HAL short with the pedHAL/HAL full , Haemophilia & Exercise Project Test-Questionnaire (HEP-Test-Q), Canadian Haemophilia Outcomes-Kids' Life Assessment Tool (CHO-KLAT) and RAND 36-item Health Survey (RAND-36) (convergent/discriminant validity) as well as its ability to discriminate between subgroups (known-group validity). Internal consistency was assessed with Cronbach's α. RESULTS: We included 113 children (median 10y [range 4-17], 53% severe haemophilia) and 691 adults (median 51y [range 18-88], 35% severe). Scores of the pedHAL/HAL full and pedHAL/HAL short were similar with high correlations (>0.9). Construct validity was confirmed for the pedHAL/HAL short . The HAL short was able to discriminate between different disease severities and ages. Cronbach's α of the pedHAL/HAL short was 0.95-0.97. CONCLUSION: This study confirmed the agreement between the pedHAL/HAL full and the pedHAL/HAL short and the construct validity of the pedHAL/HAL short . The next step is to study construct validity of the pedHAL/HAL short when administered as short forms.

AB - INTRODUCTION: The Haemophilia Activities List (HAL) and paediatric HAL assess self-reported limitations in various daily activities. To reduce patient burden, shorter versions of the pedHAL (22 items) and HAL (18 items) have been developed.AIM: This study aimed to determine the agreement between the pedHAL/HAL full and pedHAL/HAL short and construct validity and internal consistency of the pedHAL / HAL short in persons with haemophilia (PWH). METHODS: A cross-sectional secondary analysis of the Hemophilia in the Netherlands-6 national survey was performed. Adult and paediatric PWH completed the original pedHAL/HAL full , from which pedHAL / HAL short were derived. Score differences between the original and short versions were calculated. Construct validity was studied by testing hypotheses regarding the relationship of the pedHAL/HAL short with the pedHAL/HAL full , Haemophilia & Exercise Project Test-Questionnaire (HEP-Test-Q), Canadian Haemophilia Outcomes-Kids' Life Assessment Tool (CHO-KLAT) and RAND 36-item Health Survey (RAND-36) (convergent/discriminant validity) as well as its ability to discriminate between subgroups (known-group validity). Internal consistency was assessed with Cronbach's α. RESULTS: We included 113 children (median 10y [range 4-17], 53% severe haemophilia) and 691 adults (median 51y [range 18-88], 35% severe). Scores of the pedHAL/HAL full and pedHAL/HAL short were similar with high correlations (>0.9). Construct validity was confirmed for the pedHAL/HAL short . The HAL short was able to discriminate between different disease severities and ages. Cronbach's α of the pedHAL/HAL short was 0.95-0.97. CONCLUSION: This study confirmed the agreement between the pedHAL/HAL full and the pedHAL/HAL short and the construct validity of the pedHAL/HAL short . The next step is to study construct validity of the pedHAL/HAL short when administered as short forms.

KW - activities

KW - haemophilia

KW - participation

KW - patient-reported outcome

KW - validity

UR - http://www.scopus.com/inward/record.url?scp=85135128721&partnerID=8YFLogxK

U2 - 10.1111/hae.14628

DO - 10.1111/hae.14628

M3 - Article

C2 - 35905309

SN - 1351-8216

VL - 28

SP - 1007

EP - 1015

JO - Haemophilia

JF - Haemophilia

IS - 6

ER -

Validation of the pedHALshort and HALshort in Dutch children and adults with haemophilia

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