Evaluating the Quantity and Quality of Health Economic Literature in Blinding Childhood Disorders: A Systematic Literature Review.

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.

Quality assessment of anterior segment OCT images: Development and validation of quality criteria.

BACKGROUND: The utility of medical imaging is dependant on image quality. We aimed to develop and validate quality criteria for ocular anterior segment optical coherence tomography (AS-OCT) images. METHODS: We undertook a cross-sectional study using AS-OCT images from patients aged 6-16. A novel three-level grading system (good, limited or poor) was developed based on the presence of image artefact (categorised as lid, eyelash, cropping, glare, or movement artefact). Three independent experts graded 2825 images, with agreement assessed using confusion matrices and intraclass correlation coefficients (ICC) for each parameter. RESULTS: There was very good inter-grader IQA agreement assessing image quality with ICC 0.85 (95 %CI: 0.84-0.87). The most commonly occurring artefact was eyelash artefact (1008/2825 images, 36 %). Graders labelled 621/2825 (22 %) images as good and 384 (14 %) as poor. There was complete agreement at either end of the confusion matrix with no 'good' images labelled as 'poor' by other graders, and vice versa. Similarly, there was very good agreement when assessing presence of lash (0.96,0.94-0.98), movement (0.97,0.96-0.99), glare (0.82,0.80-0.84) and cropping (0.90,0.88-0.92). CONCLUSIONS: The novel image quality assessment criteria (IQAC) described here have good interobserver agreement overall, and excellent agreement on the differentiation between 'good' and 'poor' quality images. The large proportion of images graded as 'limited' suggests the need for refine this classification, using the specific IQAC features, for which we also report high interobserver agreement. These findings support the future potential for wider clinical and community care implementation of AS-OCT for the diagnosis and monitoring of ocular disease.

Quantitative and qualitative assessment of anterior segment optical coherence tomography capture of disease state in childhood anterior uveitis.

BACKGROUND/AIMS: Anterior segment optical coherence tomography (AS-OCT) assessment of anterior chamber inflammation is an emerging tool. We describe the performance of AS-OCT in a paediatric population. METHODS: A mixed-methods prospective study, using routine clinical assessment as reference standard, and AS-OCT, with Tomey CASIA2 or Heidelberg Spectralis HS1, as index test, with data collected on patient perceptions of imaging. Repeatability, diagnostic indices, responsiveness to clinical change and clinical correlations of imaging-based metrics (image cell count, size, density and brightness) were assessed, with construction of receiver operated characteristic curves. Exploratory thematic analysis of responses from families was undertaken. RESULTS: A total of 90 children (180 eyes) underwent imaging. Bland Altman limits of agreement for CASIA2 repeatability ranged from +17 cells (95% CI 13.6 to 21.1) to -19 cells (95% CI -15.6 to -23.2) and HS1 from +1 (95% CI 0.9 to 1.2) to -1.0 (-1.2 to -0.8) cells. CASIA2 imaging had higher sensitivity of 0.92 (95% CI 0.78 to 0.97) vs HS1 imaging 0.17 (95% CI 0.07 to 0.34), with positive correlation between clinical grade and CASIA2 cell count (coefficient 12.8, p=0.02, 95% CI 2.2 to 23.4). Change in clinical grade at follow-up examinations correlated with change in image based 'cell' count (r2=0.79, p<0.001). Patients reported a potential positive impact of seeing their disease activity. CONCLUSION: Our findings suggest that OCT-based imaging holds the promise of deeper understanding of disease, improved patient experience and more granular monitoring of activity with resultant improved outcomes, but further work is needed to refine acquisition and analysis protocols.

Impact of Persisting Amblyopia on Socioeconomic, Health, and Well-Being Outcomes in Adult Life: Findings From the UK Biobank.

OBJECTIVES: This study aimed to investigate associations between persisting amblyopia into adulthood and its "real-life" impacts and inform the current debate about the value of childhood vision screening programs. METHODS: Associations between persisting amblyopia and diverse socioeconomic, health, and well-being outcomes were investigated in multivariable-adjusted (sex, age, ethnicity, deprivation) regression models, with 126 400 participants (aged 40-70 years) of the UK Biobank with complete ophthalmic data. Analysis by age group (cohort 1, 60-70 years; cohort 2, 50-59 years; cohort 3, 40-49 years) assessed temporal trends. RESULTS: Of 3395 (3%) participants with confirmed amblyopia, overall 77% (2627) had persisting amblyopia, declining from 78% in cohort 1 to 73% in cohort 3. The odds of persisting amblyopia were 5.91 (5.24-6.66) and 2.49 (2.21-2.81) times greater in cohort 1 and cohort 2, respectively, than cohort 3. The odds were also higher for more socioeconomically deprived groups and for white ethnicity. Reduced participation in sport, adverse general and mental health, and well-being were all independently associated with persisting amblyopia, with the strongest associations in the youngest cohorts. Associations with lower educational attainment and economic outcomes were only evident in the oldest cohort. CONCLUSIONS: There has been a decline in the overall frequency of persisting amblyopia since the introduction of universal child vision screening in the United Kingdom. Nevertheless, most adults treated for amblyopia in childhood have persisting vision deficits. There was no evidence that persisting amblyopia has vision-mediated effects on educational, employment-related, or economic outcomes. The observed adverse outcomes were largely those not directly mediated by vision. Patients undergoing treatment should be counseled about long-term outcomes.

Trends in Visual Health Inequalities in Childhood Through Associations of Visual Function With Sex and Social Position Across 3 UK Birth Cohorts.

Importance: Despite the existing country-specific strategies tackling social inequalities in visual health in adults, little is known about trends in visual function in childhood and its association with social position. Objective: To investigate the distribution of childhood visual function in the United Kingdom and associations with early-life social position between 1961 and 1986, a period of significant social change. Design, Setting, and Participants: Longitudinal cohort study using harmonized data sets from the British 1946, 1958, and 1970 national birth cohorts. In total, 14 283 cohort members with complete data on visual acuity at age 15 or 16 years, measured in 1961, 1974, and 1986, respectively, for each cohort, and social position were assessed. Main Outcomes and Measures: Using habitual distance visual acuity (with correction if prescribed), participants were assigned to a visual function category ranging from bilateral normal to visual impairment/severe visual impairment/blindness (International Statistical Classification of Diseases, Tenth Revision, Clinical Modification). Distribution of visual function over time and associations with social position (risk ratios [RRs] and 95% confidence intervals) were analyzed. Results: Complete data were available for 3152 participants (aged 15 years; 53% boys [n = 1660]) in the 1946 Medical Research Council National Survey of Health and Development, 6683 participants (aged 16 years; 51% boys [n = 3420]) in the 1958 National Child Development Study, and 4448 participants (aged 16 years; 48% boys [n = 2156]) in the 1970 British Birth Cohort Study. The proportion of children with bilateral normal vision decreased by 1.3% (95% CI, -5.1% to 2.7%) in 1974 and 1.7% (95% CI, -5.9% to 2.7%) in 1986. The risk of overall impaired vision increased by 1.20 times (95% CI, 1.01-1.43) and the risk of visual impairment/severe visual impairment/blindness by 1.75 times (95% CI, 1.03-2.98) during this period. Girls were consistently at increased risk of all vision impairment categories. Higher social position at birth and in childhood was associated with reduced risk of visual impairment/severe visual impairment/blindness (RR, 0.58; 95% CI, 0.20-1.68) and unilateral impairment (RR, 0.89; 95% CI, 0.72-1.11), respectively. Conclusions and Relevance: Our study provides evidence of temporal decline in childhood visual function between 1961 and 1986. Despite the limited power of the analysis owing to the small sample size of those with impaired vision, we found an emergence of a contribution of sociodemographic status to the cohort effect that may be the antecedent of the current picture of childhood blindness. Equally, early-life social position may also have contributed to the current social patterning in visual function in older adults in the United Kingdom. These findings highlight the potential value of targeting children in national ophthalmic public policies tackling inequalities.

Protocol for a scoping review to map evidence from randomised controlled trials on paediatric eye disease to disease burden.

BACKGROUND: Currently, about 2 per 1000 children in the industrialised world are severely visually impaired or blind (SVI/BL) due to diverse uncommon conditions that are usually present from early infancy. The impact of SVI/BL is lifelong and life-changing. Thus, children are a priority in the WHO-led global initiative against avoidable blindness. The aim of this scoping review is to assess the current evidence base on interventions to prevent or treat the major causes of childhood SVI/BL, specifically the degree of alignment between robust interventional research (RCTs) and the burden (relative frequency) of the key causative disorders, identifying gaps in the evidence base for tackling childhood blindness. METHODS/DESIGN: We will perform a scoping review of the published literature of randomised controlled trials (RCTs) for clinical interventions that prevent or treat eye and vision diseases in children (<18 years old). Major electronic databases MEDLINE (PUBMED), EMBASE and the Cochrane CENTRAL will be searched to identify published trials using a comprehensive paediatric specific strategy informed by previous searches. The outcome of our study, randomised clinical trial activity, will be measured by the total number of RCTs and total paediatric participants randomised. The quantity and distribution of activity across diseases will be classified in the broad categories of anatomical site affected (per WHO taxonomy). The degree of alignment between paediatric trial activity and burden of SVI/BL disease (relative proportion) will be measured using a test of association (Spearman's correlation coefficient). DISCUSSION: Despite the global public health importance of childhood blindness, there has been no assessment of the completeness of the evidence base regarding clinical interventions to prevent or treat the causative disorders. This scoping review will measure the degree of alignment between the published evidence and the burden of disease to identify gaps in current knowledge and consider the underlying reasons, informing clinicians, policy makers and funders about research priorities.

Laser refractive surgery in the UK Biobank study: Frequency, distribution by sociodemographic factors, and general health, happiness, and social participation outcomes.

PURPOSE: To evaluate the frequency and distribution of laser refractive surgery in the United Kingdom by sociodemographic factors and outcomes of social participation and well-being. SETTING: Six regional recruitment centers in England and Wales. DESIGN: Cross-sectional epidemiological study. METHODS: Data were collected on sociodemographic factors and medical history; self-report on eyes/vision included reason for wearing optical correction, eye diseases, and treatment received (including refractive laser surgery). Mean spherical equivalent was used to categorize individuals as myopic (<-1.0 diopter) or hypertrophic (>+1.0 diopter). RESULTS: Between 2009 and 2010, 117 281 subjects recruited by UK Biobank undertook an ophthalmic assessment, including autorefraction. Of those with refractive error within a range eligible for laser refractive surgery (n = 60 352), 1892 (3.1%) reported having bilateral refractive surgery and 549 (0.9%) unilateral surgery. Frequency of bilateral surgery decreased with increasing age and was higher in women. Frequency did not vary with educational attainment or accommodation status but increased with income among working age adults. Social participation, for example, regular visits to a pub or social club, was more common among those who underwent surgery. Other eye conditions were reported by 28% of those reporting refractive surgery compared with 11% of those eligible for treatment but not reporting surgery. CONCLUSION: This study provides information not available routinely on the frequency and distribution of laser refractive surgery in an adult UK population. A high frequency of ocular conditions conventionally considered contraindications to laser refractive surgery raises the possibility that extant guidance on patient selection may not be followed.

Incidence and patterns of detection and management of childhood-onset hereditary retinal disorders in the UK.

BACKGROUND: A prospective, national population-based cross-sectional study to enable understanding of the burden and management in the UK of hereditary retinal disorders presenting in childhood. METHODS: Children aged <16 years with a new diagnosis of an inherited retinal disorder made between September 2006 and February 2008 in the UK were identified through two national active surveillance schemes. Clinical and socio-demographic information was collected on each child at diagnosis and 9 months later using standardised questionnaires. RESULTS: 241 patients were reported with 24 distinct diagnoses. 14% had additional systemic disorders and 13% had dual sensory impairment. Annual incidence was 1.4/100,000 children (aged 0-15 years) and the cumulative incidence by age 16 years was 22.3/100,000 children. The most common mode of inheritance was autosomal recessive. A significantly higher rate was seen in males than females (relative rate (RR) 1.53), in children of Asian compared with White ethnicity (RR 7.12) and in those in the worst quintile of socio-economic deprivation compared with those in the best (RR 1.43). Parents most commonly detected a problem with their child's vision. Up to seven different health professionals were involved in a child's early management, and variations were noted in the proportion of eligible children having assessments for low vision aids, statement of educational needs and certification as sight-impaired. CONCLUSIONS: These findings illustrate the highly heterogeneous nature of childhood retinal dystrophies and provide previously unavailable data on disease incidence, distributions and management, which are important for service provision and for planning future treatment programmes, particularly as novel therapies become available.