Abstract
Background
Evidence on the socioeconomic burden associated with childhood visual impairment, severe visual impairment and blindness (VI/SVI/BL) is needed to inform economic evaluations of existing and emerging interventions aimed at protecting or improving vision. This study aimed to evaluate the quantity and quality of literature on resource use and/or costs associated with childhood VI/SVI/BL disorders.
Methods
PubMed, Web of Science (Ovid), the National Health Service (NHS) Economic Evaluation Database and grey literature were searched in November 2020. The PubMed search was rerun in February 2022. Original articles reporting unique estimates of resource use or cost data on conditions resulting in bilateral VI/SVI/BL were eligible for data extraction. Quality assessment (QA) was undertaken using the Drummond checklist adapted for cost-of-illness (COI) studies.
Results
We identified 31 eligible articles, 27 from the peer-reviewed literature and four from the grey literature. Two reported on resource use, and 29 reported on costs. Cerebral visual impairment and optic nerve disorders were not examined in any included studies, whereas retinopathy of prematurity was the most frequently examined condition. The quality of studies varied, with economic evaluations having higher mean QA scores (82%) compared to COI studies (77%). Deficiencies in reporting were seen, particularly in the clinical definitions of conditions in economic evaluations and a lack of discounting and sensitivity analyses in COI studies.
Conclusions
There is sparse literature on resource use or costs associated with childhood visual impairment disorders. The first step in addressing this important evidence gap is to ensure core visual impairment outcomes are measured in future randomised control trials of interventions as well as cohort studies and are reported as a discrete health outcome.
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Data availability statement
All articles and grey literature can be found using the respective search strategies in supplementary file 1. All data supporting the findings of this study are available within the paper. Quality assessment scores for individual studies are available from the corresponding author on request.
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LJT is funded by the Medical Research Council University College London Doctoral Training Programme, the grant that supported this work: MR/N013867/1. ALS is supported by a National Institute for Health Research (NIHR) Clinician Scientist grant (grant number CS-2018-18-ST2-005). ALS and JSR are supported by the NIHR Biomedical Research Centres at Moorfields Eye Hospital/University College London Institute of Ophthalmology. JSR is an NIHR Senior Investigator. DEP is funded by an NIHR Advanced Fellowship (NIHR300054). All research at Great Ormond Street Hospital NHS Foundation Trust and University College London Great Ormond Street Institute of Child Health is made possible by the NIHR Great Ormond Street Hospital Biomedical Research Centre.
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LJT conceptualised the article, performed the literature search, screened all articles, extracted data and drafted the original manuscript. DEP and SK screened articles, extracted data and critically revised the manuscript. MCB supervised data visualisation and critically appraised the manuscript. ALS and JSR provided supervision on all aspects and critically appraised the manuscript. All authors approved the final manuscript.
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Teoh, L.J., Kellett, S., Patel, D.E. et al. Evaluating the Quantity and Quality of Health Economic Literature in Blinding Childhood Disorders: A Systematic Literature Review. PharmacoEconomics 42, 275–299 (2024). https://doi.org/10.1007/s40273-023-01311-5
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DOI: https://doi.org/10.1007/s40273-023-01311-5