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.

Nystagmus in patients with congenital stationary night blindness (CSNB) originates from synchronously firing retinal ganglion cells.

Congenital nystagmus, involuntary oscillating small eye movements, is commonly thought to originate from aberrant interactions between brainstem nuclei and foveal cortical pathways. Here, we investigated whether nystagmus associated with congenital stationary night blindness (CSNB) results from primary deficits in the retina. We found that CSNB patients as well as an animal model (nob mice), both of which lacked functional nyctalopin protein (NYX, nyx) in ON bipolar cells (BCs) at their synapse with photoreceptors, showed oscillating eye movements at a frequency of 4-7 Hz. nob ON direction-selective ganglion cells (DSGCs), which detect global motion and project to the accessory optic system (AOS), oscillated with the same frequency as their eyes. In the dark, individual ganglion cells (GCs) oscillated asynchronously, but their oscillations became synchronized by light stimulation. Likewise, both patient and nob mice oscillating eye movements were only present in the light when contrast was present. Retinal pharmacological and genetic manipulations that blocked nob GC oscillations also eliminated their oscillating eye movements, and retinal pharmacological manipulations that reduced the oscillation frequency of nob GCs also reduced the oscillation frequency of their eye movements. We conclude that, in nob mice, synchronized oscillations of retinal GCs, most likely the ON-DCGCs, cause nystagmus with properties similar to those associated with CSNB in humans. These results show that the nob mouse is the first animal model for a form of congenital nystagmus, paving the way for development of therapeutic strategies.

Quality of life during occlusion therapy for amblyopia from the perspective of the children and from that of their parents, as proxy.

BACKGROUND: Parents pity their amblyopic child when they think that they suffer from occlusion therapy. We measured health-related quality of life during occlusion therapy. METHODS: We developed the Amblyopia Parents and Children Occlusion Questionnaire (APCOQ). It was designed by a focus group of patients, orthoptists and ophthalmologists and consisted of twelve items concerning skin contact of patch, activities, contact with other children, emotions and awareness of necessity to patch. Parents filled out the Proxy Version shortly before the Child Version was obtained from their child. Child Version item scores were compared with Proxy Version item scores and related to the child's age, visual acuity, refraction, angle of strabismus, and cause of amblyopia. RESULTS: 63 children were recruited by orthoptists, and their parents agreed to participate. Three children were excluded: one child with Down-syndrome, one child with cerebral palsy, and one child who had been treated by occlusion therapy. Included were 60 children (mean age 4.57 ± 1.34 SD) and 56 parents. Children had occluded 128 ± 45 SD days at interview. Prior to occlusion, 54 children had worn glasses. Cronbach's α was 0.74 for the Child Version and 0.76 for the Proxy Version. Children judged their quality of life better than their parents did, especially pertaining to skin contact and activities like games and watching TV. Notably, 13 children with initial visual acuity ≥ 0.6 logMAR in the amblyopic eye experienced little trouble with games during occlusion. Quality of life in eight children with strabismus of five years and older correlated negatively (Spearman rank mean rho = -0.43) with angle of strabismus. Children with amblyopia due to both refractive error and strabismus (n = 14) had, relatively, lowest quality of life, also according to their parents, as proxy. Several children did not know why they wore a patch, contrary to what their parents thought. CONCLUSIONS: Children's quality of life during occlusion therapy is affected less than their parents think, especially regarding skin contact, playing games and watching TV during occlusion. Quality of life correlates negatively with the angle of strabismus in children five years and older. Children do not know why they wear a patch, contrary to what their parents think. Notably, children with low visual acuity in the amblyopic eye, had little difficulty playing games.

Barriers to successful dichoptic treatment for amblyopia in young children.

PURPOSE: In an ongoing randomised clinical trial comparing dichoptic VR video games with patching for amblyopia, we evaluated any potential barriers to successful use of this novel amblyopia treatment method. METHODS: From December 2017, all newly diagnosed amblyopic children were recruited. Excluded were children under age 4 and patients with strabismus exceeding 30PD. The video game was played for 1 h per week at the outpatient clinic under direct supervision. Records were kept of difficulties encountered during treatment and categorised into domains. Factors influencing the successful completion of this treatment were identified and related to patient characteristics. RESULTS: Ninety-one children were recruited for the trial, 20 parents refused participation before randomisation, because of the logistical challenges the outpatient dichoptic treatment would cause them. Of the 17 children who commenced dichoptic treatment (median age 6.2 years; IQR 4.9-8.4 years), 10 did not complete treatment. Children under age 5.5 years were unable to comprehend the game settings or the game itself. Older children (N = 7; 41%) were less willing to comply with the video game. Loss of interest in the game (N = 8; 47%) was found to be a limiting factor at all ages. CONCLUSION: Half of the children failed to complete VR dichoptic treatment, mainly due to young age. In countries with nationwide screening where amblyopia is detected before age 6, the applicability of such dichoptic treatment is limited.

Assessment of hearing screening programmes across 47 countries or regions III: provision of childhood hearing screening after the newborn period.

OBJECTIVE: To inventory provision and features of childhood hearing screening after the newborn period (CHS), primarily in Europe. DESIGN: From each participating country or region, experts provided information through an extensive questionnaire: implementation year, age at screening, test method, pass criteria, screening location, screener profession, and quality indicators: coverage, referral, follow-up and detection rates, supplemented by literature sources. STUDY SAMPLE: Forty-two European countries or regions, plus Russia, Malawi, Rwanda, India, and China. RESULTS: CHS was performed universally with pure-tone audiometry screening (PTS) in 17 countries or regions, whereas non-universal CHS was performed in eight with PTS or whisper tests. All participating countries with universal PTS had newborn hearing screening. Coverage rate was provided from three countries, detection rate from one, and referral and follow-up rate from two. In four countries, universal PTS was performed at two ages. Earliest universal PTS was performed in a (pre)school setting by nurses (n = 9, median age: 5 years, range: 3-7), in a healthcare setting by doctors and nurses (n = 7, median age: 4.5 years, range: 4-7), or in both (n = 1). CONCLUSIONS: Within universal CHS, PTS was mostly performed at 4-6 years by nurses. Insufficient collection of data and monitoring with quality indicators impedes evaluation of screening.

Assessment of hearing screening programmes across 47 countries or regions II: coverage, referral, follow-up and detection rates from newborn hearing screening.

OBJECTIVES: To assess the performance of newborn hearing screening (NHS) programmes, through selected quality measures and their relationship to protocol design. DESIGN: NHS coverage, referral, follow-up and detection rates were aggregated. Referral rates were compared to age at screening step 1, number of steps, and test method: OAE or aABR. STUDY SAMPLE: A questionnaire on existing hearing screening was completed by experts from countries in Europe, plus Russia, Malawi, Rwanda, India and China. RESULTS: Out of 47 countries or regions, NHS coverage rates were reported from 26, referral rates from 23, follow up from 12 and detection rates from 13. Median coverage rate for step 1 was 96%. Referral rate from step 1 was 6-22% where screening may be performed <24 h from birth, 2-15% for >24 h, and 4% for >72 h. Referral rates to diagnostic assessment averaged 2.1% after one to two steps using OAE only, 1.7% after two steps including aABR, and 0.8% after three to four steps including aABR. Median detection rate for bilateral permanent hearing impairment ≥40dB was 1 per 1000 infants. CONCLUSION: Referral rates were related to age, test method and number of screening steps. Quality measures were not available for many NHS programmes.

Assessment of hearing screening programmes across 47 countries or regions I: provision of newborn hearing screening.

OBJECTIVES: Newborn hearing screening (NHS) varies regarding number and type of tests, location, age, professionals and funding. We compared the provision of existing screening programmes. DESIGN: A questionnaire containing nine domains: demography, administration, existing screening, coverage, tests, diagnosis, treatment, cost and adverse effects, was presented to hearing screening experts. Responses were verified. Clusters were identified based on number of screening steps and use of OAE or aABR, either for all infants or for well and high-risk infants (dual-protocol). STUDY SAMPLE: Fifty-two experts completed the questionnaire sufficiently: 40 European countries, Russia, Malawi, Rwanda, India and China. RESULTS: It took considerable effort to find experts for all countries with sufficient time and knowledge. Data essential for evaluation are often not collected. Infants are first screened in maternity wards in most countries. Human development index and health expenditure were high among countries with dual protocols, three screening steps, including aABR, and low among countries without NHS and countries using OAE for all infants. Nationwide implementation of NHS took 6 years, on average. CONCLUSION: The extent and complexity of NHS programmes are primarily related to health expenditure and HDI. Data collection should be improved to facilitate comparison of NHS programmes across borders.

Prevalence of permanent neonatal hearing impairment: systematic review and Bayesian meta-analysis.

Objective: To investigate the variance in reported prevalence rates of permanent neonatal hearing impairment (HI) worldwide.Design: A systematic review and meta-analysis was performed on reported prevalence rates of sensorineural and permanent conductive or mixed HI worse than 40 dB in neonates, detected as a result of a screening programme or audiometric study.Study sample: For meta-analysis, 35 articles were selected, 25 from high-income countries and 10 from middle-income countries according to the world bank classification system.Results: The prevalence rate of permanent uni- and bilateral HI worse than 40 dB in neonates varied from 1 to 6 per 1000, the overall prevalence was 2.21 per 1000 [1.71, 2.8]. In NICU populations the prevalence rate was higher with a larger fraction of bilateral cases. Although not significant, prevalence rates were slightly higher in Asia compared to Europe and the number of infants lost to follow-up appeared higher in countries with lower gross national income.Conclusion: Substantial variations exist in prevalence rates of neonatal permanent HI across countries and regions. There is a strong need for more data from low-income countries to identify demographic factors that account for this variability in reported prevalence rates. Reporting these data in a uniform way is advocated.

Differences in quality-of-life dimensions of Adult Strabismus Quality of Life and Amblyopia & Strabismus Questionnaires.

PURPOSE: The Adult Strabismus Quality of Life Questionnaire (AS-20) and the Amblyopia & Strabismus Questionnaire (A&SQ) both measure health-related quality of life in strabismus patients. We evaluated to what extent these instruments cover similar domains by identifying the underlying quality-of-life factors of the combined questionnaires. METHODS: Participants were adults from a historic cohort with available orthoptic childhood data documenting strabismus and/or amblyopia. They had previously completed the A&SQ and were now asked to complete the AS-20. Factor analysis was performed on the correlation-matrix of the combined AS-20 and A&SQ data to identify common underlying factors. The identified factors were correlated with the clinical variables of angle of strabismus, degree of binocular vision, and visual acuity of the worse eye. RESULTS: One hundred ten patients completed both questionnaires (mean age, 44 years; range, 38-51 years). Six factors were found that together explained 78% of the total variance. The factor structure was dominated by the first four factors. One factor contained psychosocial and social-contact items, and another factor depth-perception items from both questionnaires. A third factor contained seven items-only from the AS-20-on eye strain, stress, and difficulties with reading and with concentrating. A fourth factor contained seven items-only from the A&SQ-on fear of losing the better eye and visual disorientation, specific for amblyopia. Current visual acuity of the worse eye correlated with depth-perception items and vision-related items, whereas current binocular vision correlated with psychosocial and social-contact items, in 93 patients. CONCLUSIONS: Factor analysis suggests that the AS-20 and A&SQ measure a similar psychosocial quality-of-life domain. However, functional problems like avoidance of reading, difficulty in concentrating, eye stress, reading problems, inability to enjoy hobbies, and need for frequent breaks when reading are represented only in the AS-20. During the development of the A&SQ, asthenopia items were considered insufficiently specific for strabismus and were excluded a priori. The patients who generated the items for the AS-20 had, in majority, adulthood-onset strabismus and diplopia and were, hence, more likely to develop such complaints than our adult patients with childhood-onset strabismus and/or amblyopia.

Patient-reported utilities in bilateral visual impairment from amblyopia and age-related macular degeneration.

BACKGROUND: Utility of visual impairment caused by amblyopia is important for the cost-effectiveness of screening for amblyopia (lazy eye, prevalence 3-3.5 %). We previously measured decrease of utility in 35-year-old persons with unilateral persistent amblyopia. The current observational case-control study aimed to measure loss of utility in patients with amblyopia with recent decrease of vision in their better eye. As these patients are rare, the sample was supplemented by patients with bilateral age-related macular degeneration with similar decrease of vision. METHODS: From our out-patient department, two groups of patients with recent deterioration to bilateral visual acuity less than Snellen 0.5 (bilateral visual impairment, BVI) were recruited, with either persistent amblyopia and age-related macular degeneration (AMB + AMD), or with bilateral age-related macular degeneration (BAMD). To measure utility, the time trade-off method and the standard gamble method were applied through interviews. Correlations were sought between utility values and visual acuity, age and Visual Function Questionnaire-25 scores. RESULTS: Seventeen AMB + AMD patients (mean age 72.9 years), and 63 BAMD patients (mean age 79.6 years) were included in the study. Among AMB + AMD, 80 % were willing to trade lifetime in exchange for cure. The overall mean time trade-off utility was 0.925. Among BAMD, 75 % were willing to trade, utility was 0.917. Among AMB + AMD, 38 % accepted risk of death in exchange for cure, overall mean standard gamble utility was 0.999. Among BAMD, 49 % accepted risk of death, utility was 0.998. Utility was not related to visual acuity but it was to age (p = 0.02). CONCLUSION: Elderly patients with BVI, caused by persistent amblyopia and age-related macular degeneration (AMD) or by bilateral AMD, had an approximately 8 % loss of TTO utility. Notably, the 8 % loss in elderly with BVI differs little from the 3.7 % loss we found previously in 35-year-old persons with unilateral amblyopia with good vision in the other eye. The moderate impact of BVI in senescence could be explained by adaptation, comorbidity, avoidance of risk and a changed percept of cure.

Nationwide inventory of risk factors for retinopathy of prematurity in the Netherlands.

OBJECTIVES: To study the incidence and risk factors for retinopathy of prematurity (ROP) in the Netherlands. STUDY DESIGN: Prospective, approximating population-based study that included infants with gestational age (GA) <32 weeks and/or birth weight (BW) <1500 g born in 2009. Pediatricians and ophthalmologists of all hospitals involved in care for premature infants reported data that were matched with the national perinatal database for risk factor analysis. RESULTS: Of 1380 infants, median GA 29.8 weeks (IQR 28.1-31.1) and median BW 1260 g (IQR 1020-1500), ROP developed in 21.9%. Logistic regression identified GA and BW as risk factors for ROP (P < .001). After adjustment for GA and BW, additional risk factors were inhaled nitric oxide (iNO; OR 2.6, 95% CI 1.1-6.2, P = .03), stay at a neonatal intensive care unit >28 days (OR 1.6, 95% CI 1.1-2.6, P = .03), and artificial ventilation >7 days (OR 1.6, 95% CI 1.1-2.5, P = .02). Prenatal glucocorticoids (OR 0.6, 95% CI 0.4-0.8, P < .001) and female sex (OR 0.7, 95% CI 0.5-0.99, P = .04) showed a lesser incidence of ROP. iNO remained significant after correction for all significant factors (OR 2.6, 95% CI 1.1-6.2, P = .03). CONCLUSION: In addition to established risk factors (GA, BW, stay at a neonatal intensive care unit >28 days, and artificial ventilation >7 days), treatment with iNO as risk factor for ROP is a novel finding.

Supervised dichoptic gaming versus monitored occlusion therapy for childhood amblyopia: Effectiveness and efficiency.

PURPOSE: To compare the effectiveness and efficiency of supervised dichoptic action-videogame play to occlusion therapy in children with amblyopia. METHODS: Newly diagnosed children with amblyopia aged 4-12 years were recruited, excluding strabismus >30PD. After 16 weeks of refractive adaptation children were randomized to gaming 1 h/week supervised by the researcher, or electronically monitored occlusion 2 h/day. The gaming group played a dichoptic action-videogame using virtual reality goggles, which included the task of catching a snowflake presented intermittently to the amblyopic eye. Contrast for the fellow eye was self-adjusted until 2 identical images were perceived. The primary outcome was visual acuity (VA) change from baseline to 24 weeks. RESULTS: We recruited 96 children, 29 declined and 2 were excluded for language or legal issues. After refractive adaptation, 24 of the remaining 65 no longer met the inclusion criteria for amblyopia, and 8 dropped out. Of 16 children treated with gaming, 7 (6.7 years) completed treatment, whereas 9 younger children (5.3 years) did not. Of 17 treated with occlusion, 14 (5.1 years) completed treatment and 3 (4.5 years) did not. Of 5 children with small-angle strabismus, 3 treated with occlusion completed treatment and 2 treated with gaming did not. Median VA improved by 0.30 logMAR (IQR 0.20-0.40) after gaming, 0.20 logMAR (0.00-0.30) after occlusion (p = 0.823). Treatment efficiency was 1.25 logMAR/100 h (range 0.42-2.08) with gaming, 0.08 (-0.19-0.68) with occlusion (p < 0.001). CONCLUSION: Dichoptic gaming seems a viable alternative for older children with refractive amblyopia after glasses adaptation. Treatment efficiency with gaming under continuous supervision was 15 times higher than with occlusion at home.

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.

An educational cartoon accelerates amblyopia therapy and improves compliance, especially among children of immigrants.

PURPOSE: We showed previously that an educational cartoon that explains without words why amblyopic children should wear their eye patch improves compliance, especially in children of immigrant parents who speak Dutch poorly. We now implemented this cartoon in clinics in low socioeconomic status (SES) areas with a large proportion of immigrants and clinics elsewhere in the Netherlands. DESIGN: Clinical, prospective, nonrandomized, preimplementation, and postimplementation study. PARTICIPANTS: Amblyopic children aged 3 to 6 years who started occlusion therapy. METHODS: Preimplementation, children received standard orthoptic care. Postimplementation, children starting occlusion therapy received the cartoon in addition. At implementation, treating orthoptists followed a course on compliance. In low SES areas, compliance was measured electronically during 1 week. MAIN OUTCOME MEASURES: The clinical effects of the cartoon-electronically measured compliance, outpatient attendance rate, and speed of reduction in interocular-acuity difference (SRIAD)-averaged over 15 months of observation. RESULTS: In low SES areas, 114 children were included preimplementation versus 65 children postimplementation; elsewhere in the Netherlands, 335 versus 249 children were included. In low SES areas, mean electronically measured compliance was 52.0% preimplementation versus 62.3% postimplementation (P=0.146); 41.8% versus 21.6% (P=0.043) of children occluded less than 30% of prescribed occlusion time. Attendance rates in low SES areas were 60.3% preimplementation versus 76.0% postimplementation (P=0.141), and 82.7% versus 84.5%, respectively, elsewhere in the Netherlands. In low SES areas, the SRIAD was 0.215 log/year preimplementation versus 0.316 log/year postimplementation (P=0.025), whereas elsewhere in the Netherlands, these were 0.244 versus 0.292 log/year, respectively (P=0.005; the SRIAD's improvement was significantly better in low SES areas than elsewhere, P=0.0203). This advantage remained after adjustment for confounding factors. Overall, 25.1% versus 30.1% (P=0.038) had completed occlusion therapy after 15 months. CONCLUSIONS: After implementation of the cartoon, electronically measured compliance improved, attendance improved, acuity increased more rapidly, and treatment was shorter. This may be due, in part, to additional measures such as the course on compliance. However, that these advantages were especially pronounced in children in low SES areas with a large proportion of immigrants who spoke Dutch poorly supports its use in such areas.

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.

Sociocultural and psychological determinants in migrants for noncompliance with occlusion therapy for amblyopia.

BACKGROUND: Compliance with occlusion therapy for amblyopia in children is low when their parents have a low level of education, speak Dutch poorly, or originate from another country. We determined how sociocultural and psychological determinants affect compliance. METHODS: Included were amblyopic children between the ages of 3 and 6, living in low socio-economic status (SES) areas. Compliance with occlusion therapy was measured electronically. Their parents completed an oral questionnaire, based on the "Social Position & Use of Social Services by Migrants and Natives" questionnaire that included demographics and questions on issues like education, employment, religion and social contacts. Parental fluency in Dutch was rated on a five-point scale. Regression analysis was used to describe the relationship between the level of compliance and sociocultural and psychological determinants. RESULTS: Data from 45 children and their parents were analyzed. Mean electronically measured compliance was 56 ± 44 percent. Children whose parents had close contact with their neighbors or who were highly dependent on their family demonstrated low levels of compliance. Children of parents who were members of a club and who had positive conceptualizations of Dutch society showed high levels of compliance. Poor compliance was also associated with low income, depression, and when patching interfered with the child's outdoor activity. Religion was not associated with compliance. CONCLUSIONS: Poor compliance with occlusion therapy seems correlated with indicators of social cohesion. High social cohesion at micro level, i.e., family, neighbors and friends, and low social cohesion on macro level, i.e., Dutch society, are associated with noncompliance. However, such parents tend to speak Dutch poorly, so it is difficult to determine its actual cause.

Vertical position of the orbits in nonsyndromic plagiocephaly in childhood and its relation to vertical strabismus.

PURPOSE: To determine the existence of a correlation between the vertical angle of strabismus and the vertical angle between the orbital axes in nonsyndromic plagiocephaly in childhood. METHODS: Patients were included when diagnosed with plagiocephaly. Orthoptic measurements showed a vertical strabismus and three-dimensional computed tomographic (CT) imaging of the skull was available. Patients were excluded if plagiocephaly was part of a syndrome or if any surgical intervention had taken place before our measurements. Three-dimensional CT imaging was used to calculate the vertical angle between the orbital axes in 3 reference planes (VAO) perpendicular to a line of reference through the lower borders of the maxilla (VAOmax), both auditory canals (VAOaud), and the lower points of the external occipital protuberances (VAOocc). RESULTS: Fourteen patients were included (mean age, 14 mo). Three-dimensional CT measurements showed a mean (SD) VAOmax of 1.70 (2.31) degrees, VAOaud of -1.54 (1.46) degrees, and VAOocc of -2.06 (4.29) degrees (a negative value indicates that the eye on the affected side was situated lower in the head). The mean vertical angle of strabismus was -2.39 (4.69) degrees in gaze toward the affected side, 3.66 (3.77) degrees in gaze ahead, and 8.14 (5.63) degrees in gaze toward the nonaffected side. The Pearson test showed no significant correlations. CONCLUSIONS: The clinical observation that vertical strabismus in adult plagiocephaly is correlated with the vertical angle of the orbital axes could not be confirmed in young children.

Scope and costs of autorefraction and photoscreening for childhood amblyopia-a systematic narrative review in relation to the EUSCREEN project data.

BACKGROUND: Amblyopia screening can target reduced visual acuity (VA), its refractive risk factors, or both. VA testing is imprecise under 4 years of age, so automated risk-factor photoscreening appears an attractive option. This review considers photoscreening used in community services, focusing on costs, cost-effectiveness and scope of use, compared with EUSCREEN project Country Reports describing how photo- and automated screening is used internationally. METHODS: A systematic narrative review was carried out of all English language photoscreening literature to September 10th 2018, using publicly available search terms. Where costs were considered, a CASP economic evaluation checklist was used to assess data quality. RESULTS: Of 370 abstracts reviewed, 55 reported large-scale community photoscreening projects. Five addressed cost-effectiveness specifically, without original data. Photoscreening was a stand-alone, single, test event in 71% of projects. In contrast, 25 of 45 EUSCREEN Country Reports showed that if adopted, photoscreening often supplements other tests in established programmes and is rarely used as a stand-alone test. Reported costs varied widely and evidence of cost-effectiveness was sparse in the literature, or in international practice. Only eight (13%) papers compared the diagnostic accuracy or cost-effectiveness of photoscreening and VA testing, and when they did, cost-effectiveness of photoscreening compared unfavourably. DISCUSSION: Evidence that photoscreening reduces amblyopia or strabismus prevalence or improves overall outcomes is weak, as is evidence of cost-effectiveness, compared to later VA screening. Currently, the most cost-effective option seems to be a later, expert VA screening with the opportunity for a re-test before referral.