Modelling the cost-effectiveness of a newborn hearing screening programme; usability and pitfalls.

OBJECTIVE: The EUSCREEN project concerns the study of European vision and hearing screening programmes. Part of the project was the development of a cost-effectiveness model to analyse such programmes. We describe the development and usability of an online tool to enable stakeholders to design, analyse or modify a newborn hearing screening (NHS) programme. DESIGN: Data from literature, from existing NHS programmes, and observations by users were used to develop and refine the tool. Required inputs include prevalence of the hearing impairment, test sequence and its timing, attendance, sensitivity, and specificity of each screening step. Outputs include the number of cases detected and the costs of screening and diagnostics. STUDY SAMPLE: Eleven NHS programmes with reliable data. RESULTS: Three analyses are presented, exploring the effect of low attendance, number of screening steps, testing in the maternity ward, or screening at a later age, on the benefits and costs of the programme. Knowledge of the epidemiology of a staged screening programme is crucial when using the tool. CONCLUSIONS: This study presents a tool intended to aid stakeholders to design a new or analyse an existing hearing screening programme in terms of benefits and costs.

A population-level post-screening treatment cost framework to help inform vision screening choices for children under the age of seven.

PURPOSE/BACKGROUND: Visual acuity (VA) screening in children primarily detects low VA and amblyopia between 3 and 6 years of age. Photoscreening is a low-cost, lower-expertise alternative which can be carried out on younger children and looks instead for refractive amblyopia risk factors so that early glasses may prevent or mitigate the conditions. The long-term benefits and costs of providing many children with glasses in an attempt to avoid development of amblyopia for some of them needs clarification. This paper presents a framework for modeling potential post-referral costs of different screening models once referred children reach specialist services. METHODS: The EUSCREEN Screening Cost-Effectiveness Model was used together with published literature to estimate referral rates and case mix of referrals from different screening modalities (photoscreening and VA screening at 2, 3-4 years and 4-5 years). UK 2019-20 published National Health Service (NHS) costings were used across all scenarios to model the comparative post-referral costs to the point of discharge from specialist services. Potential costs were compared between a) orthoptist, b) state funded ophthalmologist and c) private ophthalmologist care. RESULTS: Earlier VA screening and photoscreening yield higher numbers of referrals because of lower sensitivity and specificity for disease, and a different case mix, compared to later VA screening. Photoscreening referrals are a mixture of reduced VA caused by amblyopia and refractive error, and children with amblyopia risk factors, most of which are treated with glasses. Costs relate mainly to the secondary care providers and the number of visits per child. Treatment by an ophthalmologist of a referral at 2 years of age can be more than x10 more expensive than an orthoptist service receiving referrals at 5 years, but outcomes can still be good from referrals aged 5. CONCLUSIONS: All children should be screened for amblyopia and low vision before the age of 6. Very early detection of amblyopia refractive risk factors may prevent or mitigate amblyopia for some affected children, but population-level outcomes from a single high-quality VA screening at 4-5 years can also be very good. Total patient-journey costs incurred by earlier detection and treatment are much higher than if screening is carried out later because younger children need more professional input before discharge, so early screening is less cost-effective in the long term. Population coverage, local healthcare models, local case-mix, public health awareness, training, data monitoring and audit are critical factors to consider when planning, evaluating, or changing any screening programme.

Implementation of Newborn Hearing Screening in Albania.

Newborn hearing screening (NHS) was implemented in Albania in four maternity hospitals in 2018 and 2019. Implementation outcome, screening outcome, and screening quality measures were evaluated. Infants were first screened by midwives and nurses before discharge from the maternity hospital and returned for follow-up screening. Acceptability, appropriateness, feasibility, adoption, fidelity, coverage, attendance, and stepwise and final-referral rates were assessed by onsite observations, interviews, questionnaires, and a screening database. A post hoc analysis was performed to identify reasons for loss to follow up (LTFU) in a multivariate logistic regression. In total, 22,818 infants were born, of which 96.6% were screened. For the second screening step, 33.6% of infants were LTFU, 40.4% for the third, and 35.8% for diagnostic assessment. Twenty-two (0.1%) were diagnosed with hearing loss of ≥40 dB, six unilateral. NHS was appropriate and feasible: most infants are born in maternity hospitals, hence nurses and midwives could perform screening, and screening rooms and logistic support were supplied. Adoption among screeners was good. Referral rates decreased steadily, reflecting increasing skill. Occasionally, screening was repeated during a screening step, contrary to the protocol. NHS in Albania was implemented successfully, though LTFU was high. It is important to have effective data tracking and supervision throughout the screening.

Availability of data for cost-effectiveness comparison of child vision and hearing screening programmes.

OBJECTIVE: For cost-effectiveness comparison of child vision and hearing screening programmes, methods and data should be available. We assessed the current state of data collection and its availability in Europe. METHODS: The EUSCREEN Questionnaire, conducted in 2017-2018, assessed paediatric vision and hearing screening programmes in 45 countries in Europe. For the current study, its items on data collection, monitoring and evaluation, and six of eleven items essential for cost-effectiveness analysis: prevalence, sensitivity, specificity, coverage, attendance and loss to follow-up, were reappraised with an additional questionnaire. RESULTS: The practice of data collection in vision screening was reported in 36% (N = 42) of countries and in hearing screening in 81% (N = 43); collected data were published in 12% and 35%, respectively. Procedures for quality assurance in vision screening were reported in 19% and in hearing screening in 26%, research of screening effectiveness in 43% and 47%, whereas cost-effectiveness analysis was performed in 12% for both. Data on prevalence of amblyopia were reported in 40% and of hearing loss in 77%, on sensitivity of screening tests in 17% and 14%, on their specificity in 19% and 21%, on coverage of screening in 40% and 84%, on attendance in 21% and 37%, and on loss to follow-up in 12% and 40%, respectively. CONCLUSIONS: Data collection is insufficient in hearing screening and even more so in vision screening: data essential for cost-effectiveness comparison could not be reported from most countries. When collection takes place, this is mostly at a local level for quality assurance or accountability, and data are often not accessible. The resulting inability to compare cost-effectiveness among screening programmes perpetuates their diversity and inefficiency.

The cost-effectiveness of different visual acuity screening strategies in three European countries: A microsimulation study.

Childhood vision screening programmes in Europe differ by age, frequency and location at which the child is screened, and by the professional who performs the test. The aim of this study is to compare the cost-effectiveness for three countries with different health care structures. We developed a microsimulation model of amblyopia. The natural history parameters were calibrated to a Dutch observational study. Sensitivity, specificity, attendance, lost to follow-up and costs in the three countries were based on the EUSCREEN Survey. Quality adjusted life-years (QALYs) were calculated using assumed utility loss for unilateral persistent amblyopia (1%) and bilateral visual impairment (8%). We calculated the cost-effectiveness of screening (with 3.5% annual discount) by visual acuity measurement at age 5 years or 4 and 5 years in the Netherlands by nurses in child healthcare centres, in England and Wales by orthoptists in schools and in Romania by urban kindergarten nurses. We compared screening at various ages and with various frequencies. Assuming an amblyopia prevalence of 36 per 1,000 children, the model predicted that 7.2 cases of persistent amblyopia were prevented in the Netherlands, 6.6 in England and Wales and 4.5 in Romania. The cost-effectiveness was €24,159, €19,981 and €23,589, per QALY gained respectively, compared with no screening. Costs/QALY was influenced most by assumed utility loss of unilateral persistent amblyopia. For all three countries, screening at age 5, or age 4 and 5 years were optimal. Despite differences in health care structure, vision screening by visual acuity measurement seemed cost-effective in all three countries.

Implementation of paediatric vision screening in urban and rural areas in Cluj County, Romania.

BACKGROUND: In 2018 and 2019, paediatric vision screening was implemented in Cluj County, Romania, where universal paediatric vision screening does not yet exist. We report on the preparation and the first year of implementation. METHODS: Objectives, target population and screening protocol were defined. In cities, children were screened by kindergarten nurses. In rural areas, kindergartens have no nurses and children were screened by family doctors' nurses, initially at the doctors' offices, later also in rural kindergartens. CME-accredited training courses and treatment pathways were organised. Implementation was assessed through on-site observations, interviews, questionnaires and analysis of screening results of referred children. RESULTS: Out of 12,795 eligible four- and five-year-old children, 7,876 were screened in 2018. In the cities, kindergarten nurses screened most children without difficulties. In Cluj-Napoca 1.62x the average annual birth rate was screened and in the small cities 1.64x. In the rural areas, however, nurses of family doctors screened only 0.49x the birth rate. In 51 out of 75 rural communes, no screening took place in the first year. Of 118 rural family doctors' nurses, 51 had followed the course and 26 screened children. They screened only 41 children per nurse, on average, as compared to 80 in the small cities and 100 in Cluj-Napoca. Screening at rural kindergartens met with limited success. These are attended by few children because of low population density, parents working abroad or children being kept at home in case of bad weather and road conditions. CONCLUSIONS: Three times fewer children were screened in rural areas as compared to urban areas. Kindergartens in rural areas are too small to employ nurses and family doctors' nurses do not have easy access to many children and have competing healthcare priorities: there are 1.5x as many family doctors in urban areas as compared to rural areas. For nationwide scaling-up of vision screening, nurses should be enabled to screen a sufficient number of children in rural areas.