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12-03-2024

Measuring health-related quality of life in children with suspected genetic conditions: validation of the PedsQL proxy-report versions

Auteurs: Hadley Stevens Smith, Michael Leo, Katrina Goddard, Kristin Muessig, Frank Angelo, Sara Knight, Simon Outram, Nicole R. Kelly, Christine Rini

Gepubliceerd in: Quality of Life Research | Uitgave 6/2024

Abstract

Purpose

Measuring health-related quality of life (HRQoL) of children with suspected genetic conditions is important for understanding the effect of interventions such as genomic sequencing (GS). The Pediatric Quality of Life Inventory (PedsQL) is a widely used generic measure of HRQoL in pediatric patients, but its psychometric properties have not yet been evaluated in children undergoing diagnostic GS.

Methods

In this cross-sectional study, we surveyed caregivers at the time of their child’s enrollment into GS research studies as part of the Clinical Sequencing Evidence Generating Research (CSER) consortium. To evaluate structural validity of the PedsQL 4.0 Generic Core Scales and PedsQL Infant Scales parent proxy-report versions, we performed a confirmatory factor analysis of the hypothesized factor structure. To evaluate convergent validity, we examined correlations between caregivers’ reports of their child’s health, assessed using the EQ VAS, and PedsQL scores by child age. We conducted linear regression analyses to examine whether age moderated the association between caregiver-reported child health and PedsQL scores. We assessed reliability using Cronbach’s alpha.

Results

We analyzed data for 766 patients across all PedsQL age group versions (1–12 months through 13–18 years). Model fit failed to meet criteria for good fit, even after modification. Neither age group (categorical) nor age (continuous) significantly moderated associations between PedsQL scores and caregiver-reported child health. Cronbach’s alphas indicated satisfactory internal consistency for most PedsQL scales.

Conclusion

The PedsQL Generic Core Scales and Infant Scales may be appropriate to measure HRQoL in pediatric patients with suspected genetic conditions across a wide age range. While we found evidence of acceptable internal consistency and preliminary convergent validity in this sample, there were some potential problems with structural validity and reliability that require further attention.

vorige artikel Minimal clinically important differences in health-related quality of life after treatment with direct-acting antivirals for chronic hepatitis C: ANRS CO22 HEPATHER cohort (PROQOL-HCV)

volgende artikel Exploratory factor analysis and Rasch analysis to assess the structural validity of the Adult Social Care Outcomes Toolkit Proxy version (ASCOT-Proxy) completed by care home staff

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Titel: Measuring health-related quality of life in children with suspected genetic conditions: validation of the PedsQL proxy-report versions
Auteurs: Hadley Stevens Smith
Michael Leo
Katrina Goddard
Kristin Muessig
Frank Angelo
Sara Knight
Simon Outram
Nicole R. Kelly
Christine Rini
Publicatiedatum: 12-03-2024
Uitgeverij: Springer International Publishing
Gepubliceerd in: Quality of Life Research / Uitgave 6/2024
Print ISSN: 0962-9343
Elektronisch ISSN: 1573-2649
DOI: https://doi.org/10.1007/s11136-024-03623-1

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