A review of preference-based measures for the assessment of quality of life in children and adolescents with cerebral palsy
To examine the psychometric properties and suitability for use within the context of cerebral palsy research in children and adolescents of generic preference-based outcome measures (PROMs).
Nine electronic databases were searched in this systematic review. The consensus-based standards for the selection of health measurement instruments (COSMIN) checklist were used to measure the psychometric properties of the PROMs. A meta-analysis was used to pool correlation coefficients for convergent validity using the Schmidt–Hunter method. Heterogeneity was assessed using the I-squared statistic (I2).
Four preference-based PROMs were identified from eight studies: Health Utilities Index—Mark 2 and 3 (HUI-2 and HUI-3, respectively), the Assessment Quality of Life-4 dimension (AQoL-4D) and the EuroQol-5 dimension 3 level (EQ-5D-3L). Only the HUI system was primarily developed for application with children/adolescents though health-state values for scoring the PROM were elicited from adults. The HUI-3 covered the most relevant constructs though it excludes important modules of health-related quality of life (HRQOL) such as activity limitations and participation restrictions. In terms of psychometric properties, evidence was presented for only five of COSMIN measurement properties: reliability (HUI3), measurement error (HUI-3), content validity (HUI-2 and HUI-3), Hypotheses testing (HUI-3 and AQoL-4D) and criterion validity (HUI-3). No papers reported on internal consistency, structural validity, cross-cultural validity or responsiveness of the preference-based measures in children and adolescents with cerebral palsy.
This review highlights the dearth in studies using preference-based PROMs to measure HRQOL associated with cerebral palsy in children and adolescents. The HUI-3 demonstrated the strongest psychometric properties, though it does not cover all dimensions relevant to this population.
KeywordsQuality of life Utility Instruments Child Adolescent Cerebral palsy
The Sixteen-dimensional measure of HRQOL
The adolescent health utility measure
Assessment Quality of Life
Child health utility 9-dimensions
Cerebral palsy quality of life
EuroQol-5 dimensions 3 levels
EuroQol-5 dimensions youth version
Gross motor function classification system
Health-related quality of life
Health Utilities Index
National Institute for Health and Care Excellence
Pharmaceutical Benefits Advisory Committee
Pediatric Quality of Life Inventory
Preferred reporting items for systematic reviews and meta-analysis
Patient-reported outcome measures
International Prospective Register of Systematic Reviews
Quality of life
Quality of life instrument for people with developmental disability
Quality of well-being scale
Visual analogue scale
CM is supported by the Australian Government Research Training Program Scholarship.
CM, GC and JR formulated the idea for the study. CM wrote the first draft and the co-authors (EH, GC, RR, JR) revised the study for important intellectual content. CM will act as a guarantor for the work.
Compliance with ethical standards
Conflict of interest
All authors declared no conflict of interest.
Research involving human and animal rights
This manuscript is a systematic review which only contains data from previously published studies. No clinical trials were conducted nor were patient data collected for this research.
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