Shortening the Haemophilia Activities List (HAL) from 42 items to 18 items

Abstract

Introduction:The Haemophilia Activities List (HAL) was developed to measure activities and participation in persons with haemophilia (PWH). Shortening the questionnaire may facilitate use of the HAL. Aim:The aim of this study was to determine which items of the HAL are redundant, to construct a shorter version of the HAL, and to determine the construct validity of the HAL short .Methods: A secondary analysis was performed on pooled data of two published studies using the HAL (seven domains, 42 items, optimum score: 100) in adults with haemophilia A/B. Data were divided into a derivation (62%) and a validation set (38%).Redundant items were identified by evaluation of: floor and ceiling effects, proportions of missing and 'not applicable' responses, inter-item correlations, component loadings in an exploratory factor analysis, internal consistency, and item-total correlations. Correlations with the SF-36 and EQ-5D-5L were used to determine construct validity of the HAL short .Results: Data on 680 PWH were evaluated. In the derivation dataset (n = 420), median age was 30 years (range 18-80), 43% had severe haemophilia and 61% received prophylaxis. Median (IQR) HAL sum score was 65.0 (55.7-88.8). The stepwise procedure resulted in a HAL short of 18 items with a median sum score of 63.3 (54.4-86.7). Construct validity was similar for the HAL and HAL short in the validation dataset (n = 260). Conclusion:This clinimetric study resulted in a >50% shortening of the HAL. The 18item HAL short reduces patient burden and is expected to capture the information on activities and participation. The HAL short needs further validation.

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

“…In addition, the proportions of ‘positive’ scores (pedHAL ≤95, HAL ≤90) and positive predictive value of the pedHAL/HAL short were calculated and shown in a cross table. The thresholds of ≤95 for the pedHAL and ≤90 for the HAL are based on reported limits of agreement (LoA) of test‐retest data 4,21 and in accordance with previous studies 9,10,22 …”

“…Internal consistency of the HAL short (Cronbach's α 0.97) was comparable to other outcomes on physical functioning like the RAND‐36 physical functioning and HEP‐test‐Q (Cronbach's α : 0.95–0.96) 12,19 . Although reduction of one or more items can be considered, 24 the stepwise approach including item deletion based on internal consistency did not result in less items 10 …”

Key considerations to reduce or address respondent burden in patient-reported outcome (PRO) data collection

“…Historically, PROs have relied on long questionnaires which may be more precise but more time-consuming for patients, which are potentially burdensome and may affect compliance 11 , 23 , 36 , 37 . For instance, the original Kidney Disease Quality of Life (KDQOL) questionnaire has 134 items.…”

“…Patient's health status and functional disability should be considered during the PROM selection process to ensure that length and content are tailored to limit response burden 44,45 . Several studies focused on the development of short forms of existing measures and found that this enhanced the feasibility of collecting PROs on a regular basis and potentially could reduce respondent burden 23,36,41,[46][47][48][49][50][51][52][53][54][55] . The use of statistical methods such as factor analysis and Rasch can assist with item reduction during the validation of PROMs.…”

Rasch Measurement and Patient Reported Value Claims: A Primer for Hemophilia

“…Likert-based polytomous instruments in hemophilia for which we must assume only ordinal properties include: (i) the Haem-A-QoL (adult) and Haemo-QoL (pediatric) instruments which rely on the summation of integer scores from Likert response scales (some reordered) with subscales and an overall score to create composite total raw and transformed ordinal scores 35 ; (ii) the Canadian Hemophilia Outcomes-Kids Life Assessment Tool (CHO-KLAT) version 2.0 (youth), with a recent revision to version 3.0 (youth) with 40 items and seven domains with Likert integer summed sub-scales and total score standardized to 100 36 ; and (iii) the Hemophilia Activities List (HAL) designed to measure the impact of hemophilia on self-perceived functional abilities in adults with hemophilia has 42 Likert items in seven domains where the most important outcomes are overall integer sum score and three component scores for upper extremity activities, basic lower extremity activities and complex lower extremity activities, with each domain also scored and, more recently, a shortened version of 18 items 37 . None of these instruments has the required interval scale measurement property for a single attribute which is the unique contribution from applying Rasch rules to instrument development.…”