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

Deborah Schofield; Joshua Kraindler; Owen Tan; Rupendra Shrestha; Diana Jelovic; Sarah West; Alan Ma; John Grigg; Robyn V Jamieson

doi:10.1016/j.xops.2021.100106

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

Ophthalmol Sci. 2021 Dec 23;2(1):100106. doi: 10.1016/j.xops.2021.100106. eCollection 2022 Mar.

Authors

Deborah Schofield¹, Joshua Kraindler¹, Owen Tan¹, Rupendra Shrestha¹, Diana Jelovic², Sarah West¹, Alan Ma^{2

3}, John Grigg^{3

4

5}, Robyn V Jamieson^{2

3

4}

Affiliations

¹ GenIMPACT: Centre for Economic Impacts of Genomic Medicine, Macquarie Business School, Macquarie University, North Ryde, Australia.
² Eye Genetics Research Unit, Children's Medical Research Institute, University of Sydney, Sydney, Australia.
³ Sydney Children's Hospitals Network, Westmead, Australia.
⁴ Save Sight Institute, University of Sydney, Sydney, Australia.
⁵ Sydney Eye Hospital, Sydney, Australia.

Abstract

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.

Keywords: AQoL-8D, Assessment of Quality of Life 8-Dimension; Cost-effectiveness analysis; IRD, inherited retinal disease; Inherited retinal diseases; NEI-VFQ-25, 25-item National Eye Institute Visual Function Questionnaire; NEI-VFQ-39, 39-item National Eye Institute Visual Function Questionnaire; QALY, quality-adjusted life-year; QoL, quality of life; Quality of life; Utilities.