Are We Agreed? Self- Versus Proxy-Reporting of Paediatric Health-Related Quality of Life (HRQoL) Using Generic Preference-Based Measures: A Systematic Review and Meta-Analysis

Abstract

Objective The aim of this study was to examine the level of agreement between self-and proxy-reporting of health-related quality of life (HRQoL) in children (under 18 years of age) using generic preference-based measures. Methods A systematic review of primary studies that reported agreement statistics for self and proxy assessments of overall and/or dimension-level paediatric HRQoL using generic preference-based measures was conducted. Where available, data on intraclass correlation coefficients (ICCs) were extracted to summarise overall agreement levels, and Cohen's kappa was used to describe agreement across domains. A meta-analysis was also performed to synthesise studies and estimate the level of agreement between self-and proxy-reported paediatric overall and domain-level HRQoL. Results Of the 30 studies included, 25 reported inter-rater agreement for overall utilities, while 17 reported domain-specific agreement. Seven generic preference-based measures were identified as having been applied: Health Utilities Index (HUI) Mark 2 and 3, EQ-5D measures, Child Health Utility 9 Dimensions (CHU9D), and the Quality of Well-Being (QWB) scale. A total of 45 dyad samples were included, with a total pooled sample of 3084 children and 3300 proxies. Most of the identified studies reported a poor inter-rater agreement for the overall HRQoL using ICCs. In contrast to more observable HRQoL domains relating to physical health and functioning, the inter-rater agreement was low for psychosocial-related domains, e.g., 'emotion' and 'cognition' attributes of both HUI2 and HUI3, and 'feeling worried, sad, or unhappy' and 'having pain or discomfort' domains of the EQ-5D. Parents demonstrated a higher level of agreement with children relative to health professionals. Child self-and proxy-reports of HRQoL showed lower agreement in cancer-related studies than in non-cancerrelated studies. The overall ICC from the meta-analysis was estimated to be 0.49 (95% confidence interval 0.34-0.61) with poor inter-rater agreement. Conclusion This study provides evidence from a systematic review of studies reporting dyad assessments to demonstrate the discrepancies in inter-rater agreement between child and proxy reporting of overall and domain-level paediatric HRQoL using generic preference-based measures. Further research to drive the inclusion of children in self-reporting their own HRQoL wherever possible and limiting the reliance on proxy reporting of children's HRQoL is warranted.

Development and validation of TreatHSP-QoL: a patient-reported outcome measure for health-related quality of life in hereditary spastic paraplegia

“…The lowest correlation of 0.64 was reached for the subdomain (I) general quality of life and attitude to the disease since this subdomain includes the most personal items like emotional wellbeing or concerns about the future. A meta-analysis of self- and proxy-reports of HRQoL in children also showed that the inter-rater agreement was lower for psychosocial-related domains such as emotion and cognition than for more observable domains relating to physical health and functioning [ 47 ].…”

A cross-sectional study evaluating health-related quality of life of Chinese pediatric patients with hematological malignancies using EQ-5D-Y

“…Both ECOG and HRQoL are independent health evaluations performed by clinicians and patients themselves. It has been suggested that the health status rated by the pediatric patients alone is not comprehensive ( 24 ) where bigger differences existed between proxy-reported and self-reported health outcomes in measuring pediatric patients than adults ( 25 ). Therefore, the agreement between the two health indicators can serve as a validation of our results.…”

Development and validation of TreatHSP-QoL – a patient-reported outcome measure for health-related quality of life in hereditary spastic paraplegia

“…The lowest correlation of 0.64 was reached for the subdomain (I) general quality of life and attitude to the disease since this subdomain includes the most personal items like emotional wellbeing or concerns about the future. A meta-analysis of self-and proxy-reports of HRQoL in children also showed that the inter-rater agreement was lower for psychosocial-related domains such as emotion and cognition than for more observable domains relating to physical health and functioning [29].…”