Patient-Reported Health-Related Quality of Life in Individuals with Inherited Retinal Diseases

Deborah Schofield, PhD; Joshua Kraindler, MSc; Owen Tan, MComm; Rupendra Shrestha, PhD; Diana Jelovic, BAppSci; Sarah West, MBMSc; Alan Ma, PhD, FRACP; John Grigg, MD, FRANZCO; Robyn V. Jamieson, PhD, FRACP

Ophthalmology Science (Mar 2022)

Patient-Reported Health-Related Quality of Life in Individuals with Inherited Retinal Diseases

Deborah Schofield, PhD,
Joshua Kraindler, MSc,
Owen Tan, MComm,
Rupendra Shrestha, PhD,
Diana Jelovic, BAppSci,
Sarah West, MBMSc,
Alan Ma, PhD, FRACP,
John Grigg, MD, FRANZCO,
Robyn V. Jamieson, PhD, FRACP

Affiliations

Deborah Schofield, PhD: GenIMPACT: Centre for Economic Impacts of Genomic Medicine, Macquarie Business School, Macquarie University, North Ryde, Australia
Joshua Kraindler, MSc: GenIMPACT: Centre for Economic Impacts of Genomic Medicine, Macquarie Business School, Macquarie University, North Ryde, Australia; Correspondence: Joshua Kraindler, GenIMPACT: Centre for Economic Impacts of Genomic Medicine, Macquarie Business School, Macquarie University, Level 4, 4 Easten Road, North Ryde, 2109, Australia.
Owen Tan, MComm: GenIMPACT: Centre for Economic Impacts of Genomic Medicine, Macquarie Business School, Macquarie University, North Ryde, Australia
Rupendra Shrestha, PhD: GenIMPACT: Centre for Economic Impacts of Genomic Medicine, Macquarie Business School, Macquarie University, North Ryde, Australia
Diana Jelovic, BAppSci: Eye Genetics Research Unit, Children’s Medical Research Institute, University of Sydney, Sydney, Australia
Sarah West, MBMSc: GenIMPACT: Centre for Economic Impacts of Genomic Medicine, Macquarie Business School, Macquarie University, North Ryde, Australia
Alan Ma, PhD, FRACP: Eye Genetics Research Unit, Children’s Medical Research Institute, University of Sydney, Sydney, Australia; Sydney Children’s Hospitals Network, Westmead, Australia
John Grigg, MD, FRANZCO: Sydney Children’s Hospitals Network, Westmead, Australia; Save Sight Institute, University of Sydney, Sydney, Australia; Sydney Eye Hospital, Sydney, Australia
Robyn V. Jamieson, PhD, FRACP: Eye Genetics Research Unit, Children’s Medical Research Institute, University of Sydney, Sydney, Australia; Sydney Children’s Hospitals Network, Westmead, Australia; Save Sight Institute, University of Sydney, Sydney, Australia

Journal volume & issue: Vol. 2, no. 1
p. 100106

Abstract

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Purpose: To evaluate the impact of inherited retinal diseases (IRDs) on quality of life (QoL) using multiattributable health utilities derived from primary patient data. Design: Cross-sectional observational study. Participants: Seventy adult patients (mean age, 42.7 years) with IRD recruited from state-wide services in Australia. Methods: Health utility values were calculated from the Assessment of Quality of Life 8-Dimension (AQoL-8D). Linear regressions were used to analyze the relationship between the 25-item and 39-item National Eye Institute Visual Function Questionnaires (NEI-VFQ-25 and NEI-VFQ-39, respectively) and health utilities from the AQoL-8D. Main Outcome Measures: The AQoL-8D utility values were compared between the IRD cohort and population norms. Regressions were used to determine explanatory power of the NEI-VFQ-25 and NEI-VFQ-39 for health utilities from the AQoL-8D. Results: Average health-related utility for patients with IRD was 0.58, significantly lower than population norms of 0.80. The IRD patient scores were significantly lower than population norms for all 8 domains of the AQoL-8D. Regressions showed a statistically significant relationship between the NEI-VFQ-39 and AQoL-8D, with the NEI-VFQ-39 and other clinical data explaining up to 73% of the variation in AQoL-8D values and 69% of the variation in the NEI-VFQ-25 values. Conclusions: Patients with IRD have significantly lower utility values across all dimensions of QoL, with the largest differences in independent living, senses, and relationships. The NEI-VFQ-25 and NEI-VFQ-39 are highly correlated with overall AQoL-8D utilities and, combined with other data, can reasonably estimate QoL utilities required for cost-effectiveness studies.

Published in Ophthalmology Science

ISSN: 2666-9145 (Online)
Publisher: Elsevier
Country of publisher: United States
LCC subjects: Medicine: Ophthalmology
Website: https://www.journals.elsevier.com/ophthalmology-science/

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