Validation of the pedHAL_short and HAL_short 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/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.

Validity and reliability test of the Indonesian version of the PedHALshort questionnaire

“…The internal consistency of the PedHALshort questionnaire in the total group was high, with Cronbach's α value of 0.85 (0.74–0.92). These results are in accordance with research in the Netherlands which obtained Cronbach's α value of 0.95–0.97 12 . The lowest ICC in the age group of 4−7 years may be attributed to the smaller group size and the fact that parents completed the form.…”

“…These findings align with the construct validity tested in the Netherlands where it was linked to the Haemophilia & Exercise Project–Test–Questionnaire (HEP‐test‐Q) with the results of PedHALshort ( r = .42) and CHO‐KLAT (PedHALshort r = .44) showing a moderate correlation 12 . Research of physical function in adolescents with haemophilia in the UK also found a moderate correlation between PedHAL and Haemo‐QoL SF r = −.561 ( p < .0001) 13 .…”

“…These results are in accordance with research in the Netherlands which obtained Cronbach's α value of 0.95-0.97. 12 The lowest ICC in the age group of 4−7 years may be attributed to the smaller group size and the fact that parents completed the form. PedHALshort Indonesia language version can be used to assess the limitation in activity and participation in Indonesia population.…”

The value‐based healthcare approach to haemophilia: Development of outcome measures for the evaluation of care of people with haemophilia

“…The normalised HAL/PedHAL score was categorised as ≥90 (good mobility and physical activities), 65−90 (fair), < 65 (poor). The cut‐offs for both tools were defined on clinical experience and published study on BPI 35–36 and HAL/PedHAL 37–39 performance, respectively. A categorisation scheme is reported in Table S3 and combination of three levels of these three items to form possible health states in Figure S2.…”