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 - 2022/11/1
Y1 - 2022/11/1
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/HALfull and pedHAL/HALshort and construct validity and internal consistency of the pedHAL/HALshort 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/HALfull, from which pedHAL/HALshort 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/HALshort with the pedHAL/HALfull, 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/HALfull and pedHAL/HALshort were similar with high correlations (>0.9). Construct validity was confirmed for the pedHAL/HALshort. The HALshort was able to discriminate between different disease severities and ages. Cronbach's α of the pedHAL/HALshort was 0.95–0.97. Conclusion: This study confirmed the agreement between the pedHAL/HALfull and the pedHAL/HALshort and the construct validity of the pedHAL/HALshort. The next step is to study construct validity of the pedHAL/HALshort 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/HALfull and pedHAL/HALshort and construct validity and internal consistency of the pedHAL/HALshort 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/HALfull, from which pedHAL/HALshort 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/HALshort with the pedHAL/HALfull, 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/HALfull and pedHAL/HALshort were similar with high correlations (>0.9). Construct validity was confirmed for the pedHAL/HALshort. The HALshort was able to discriminate between different disease severities and ages. Cronbach's α of the pedHAL/HALshort was 0.95–0.97. Conclusion: This study confirmed the agreement between the pedHAL/HALfull and the pedHAL/HALshort and the construct validity of the pedHAL/HALshort. The next step is to study construct validity of the pedHAL/HALshort 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 - https://doi.org/10.1111/hae.14628
DO - https://doi.org/10.1111/hae.14628
M3 - Article
C2 - 35905309
SN - 1351-8216
VL - 28
SP - 1007
EP - 1015
JO - Haemophilia
JF - Haemophilia
IS - 6
ER -