Capturing the experiences of patients with inherited optic neuropathies: a systematic review of patient-reported outcome measures (PROMs) and qualitative studies.
Authors
Chen, Benson S
Galus, Tomasz
Tadić, Valerija
Horton, Mike
Pesudovs, Konrad
Braithwaite, Tasanee
Yu-Wai-Man, Patrick
Publication Date
2022-06Journal Title
Graefes Arch Clin Exp Ophthalmol
ISSN
0721-832X
Publisher
Springer Science and Business Media LLC
Volume
260
Issue
6
Pages
2045-2055
Language
en
Type
Article
This Version
VoR
Metadata
Show full item recordCitation
Chen, B. S., Galus, T., Archer, S., Tadić, V., Horton, M., Pesudovs, K., Braithwaite, T., & et al. (2022). Capturing the experiences of patients with inherited optic neuropathies: a systematic review of patient-reported outcome measures (PROMs) and qualitative studies.. Graefes Arch Clin Exp Ophthalmol, 260 (6), 2045-2055. https://doi.org/10.1007/s00417-021-05534-0
Abstract
PURPOSE: To identify and comprehensively evaluate studies capturing the experience of individuals affected by an inherited optic neuropathy (ION), focusing on patient-reported outcome measures (PROMs) and qualitative studies where the health status and quality of life (QoL) of these individuals have been explored. METHODS: Systematic review of five databases using a search strategy combining four concepts: (1) ION; (2) QoL and health status; (3) PROMs; and (4) qualitative research. Studies assessing the impact of ION on any QoL domain using a PROM or qualitative methodology were included and appraised, using criteria based on the COSMIN checklist (for PROM studies) and the CASP checklist (for qualitative studies). RESULTS: Of 1326 unique articles identified, six studies were included. Five PROMs were identified: Visual Function Index (VF-14); Hospital Anxiety and Depression Scale (HADS); a novel graphical online assessment tool (NGOAT) for reporting emotional response to vision loss; a new PROM informed by the DSM-V Criteria for Major Depressive Disorder; and an interpersonal and career 'impact rating' PROM. The psychometric performance of included PROMs were poorly described. Qualitative studies found that vision loss resulted in psychosocial losses including loss of social and communication skills and loss of independence and freedom. Factors that modified the response to vision loss were also identified. CONCLUSION: The current PROMs used by individuals with ION have poor content coverage, primarily measuring activity limitation and emotional well-being, and insufficient reporting of psychometric performance. There is a need to develop a PROM for individuals ION to report their experiences of living with their condition.
Keywords
Neurophthalmology, Leber hereditary optic neuropathy, Dominant optic atrophy, Quality of life, Patient-reported outcome measure
Identifiers
s00417-021-05534-0, 5534
External DOI: https://doi.org/10.1007/s00417-021-05534-0
This record's URL: https://www.repository.cam.ac.uk/handle/1810/336689
Rights
Licence:
http://creativecommons.org/licenses/by/4.0/
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