On June 28th and 29th, 2021, the Final Dissemination Meeting of the EUSCREEN Study took place in Zaandam. As a hybrid event, the meeting was streamed live to a worldwide audience. Fortunately, the limitation of 50 participants for the meeting because of the COVID-19 pandemic had been lifted on June 26th, but many countries still were ‘orange’ on the ECDC COVID-19 chart and, hence, the Country Representatives from these countries could not attend. Speakers and other participants who could not be present in person were able to join the meeting and take part in the discussions online.
The meeting was opened on Monday morning by Zaandam’s mayor Jan Hamming, who congratulated the organizers and explained that Zaandam has a great history with preventive healthcare for children. The first school doctor in the Netherlands was appointed on April 1st, 1904. He worked in the Botenmakersstraat in Zaandam at 250 meters from the congress venue. In those days, many children had head sore, a very infectious fungus infection of the head. These children were sent home from school by the schoolteachers, but they often did not get better and they hung around in the streets for months and did not go to school. In 1901, primary school attendance became compulsory in the Netherlands, so now a school doctor was needed to assess the pupil, send it home from school and start treatment. In 1901 the Council of Zaandam declined a request for a school teacher, but in 1902 Carel Adolf Elias became mayor of Zaandam. At his PhD defendance in 1891 he had contended that “the appointment of factory and schooldoctors by the government would benefit the working class.” So in 1904 the Council of Zaandam, led by Elias, decided to appoint Cornelis Jan van der Loo as the first school doctor in the Netherlands. One of the council members who voted in favor was Johannes Simonsz, great grandfather of the EUSCREEN study coordinator. The tasks of the first school doctor were “to judge the physical condition of the pupils, to exam the pupil’s body posture, to detect infectious diseases, to check the number of class hours so that the children are not overloaded, to educate teachers about hygiene, and to determine the pupil’s eyesight: can the child see on the blackboard and where should the pupil therefore sit in the classroom.”
Pharmacist Marius Romijn from Zaandam explained the history of the ‘Baptist Admonition,’ the wooden church where the meeting took place. In the 17th century the Baptists, like other groups outside the Dutch Reformed Church, were subject to restricted toleration in the Netherlands, so their churches were not allowed to be conspicuous from the outside.Citation1 This church was built in 1687 after a merger of several Baptist communities in the region. It is inconspicuous from the outside but beautifully ornamented from the inside. The organ was built in 1784 by the famous Johannes Petrus Künckel from Rotterdam.
Preparation of the EUSCREEN study
The program kicked off with an introduction about the preparation of the EUSCREEN Study by the study coordinator Huib Simonsz (Erasmus MC, Rotterdam). He discussed the first cost-effectiveness model for vision screening in the Netherlands. This micro-simulation model was developed in 2008 by the engineer Frans-Willem Goudsmit to calculate the effectiveness of a vision screening program, based on estimates of age-specific incidence of amblyopia, age-specific sensitivity of examination technique and age-specific effect of occlusion treatment,Citation2 derived from observational data such as the RAMSES study.Citation3,Citation4 The calculations of the cost-effectiveness model in an analysis of the extensive vision screening program in the Netherlands in 2008 led to the first-ever disinvestment study of vision screening in children that was carried out by Frea Sloot (Erasmus MC, Rotterdam).Citation5 This study compared two consecutive birth cohorts (N = 11,000 in total) that both received visual-acuity screening at the age of 3–4 years, but only the first cohort was screened with orthoptic tests between the age of 6–24 months (before the age that visual acuity can be measured routinely). The study has shownCitation5 that the effectiveness of vision screening is not reduced by omission of screening with orthoptic tests at the age of 6–24 months. On the basis of these results, the national vision screening program in the Netherlands has been reduced when it was revised in 2019.
EUSCREEN survey of hearing and vision screening programs
The EUSCREEN Study developed a model (miscan.euscreen.org) to enable cost-effectiveness analysis of pediatric vision and hearing screening programs in all countries in Europe. To populate the model with data, the extensive EUSCREEN Survey was first conducted among Country Representatives, one appointed for vision screening, one for hearing screening and one for general screening, in 49, mostly European, countries.
André Goedegebure (Erasmus MC, Rotterdam) presented the results of the EUSCREEN Survey concerning neonatal hearing screening programs in Europe. Neonatal hearing screening has been implemented in most European countries. Most infants are screened in hospitals, mostly by nurses within three days from birth. Interestingly, the extent and complexity of neonatal hearing screening programs are primarily related to the countries’ health expenditure and Human Development Index score.Citation6
Inger Uhlén (Karolinska Institute, Stockholm) discussed the use of quality measures in neonatal hearing screening: coverage, referral rate and lost-to-follow-up rate. Data on these quality measures were not available in many countries. Coverage rates were reported from 26 out of 47 countries or regions, referral rates from 23, follow-up from 12 and detection rates from 13 countries.Citation7
Allison Mackey (Karolinska Institute, Stockholm) reported the EUSCREEN Survey results regarding childhood (preschool or school-age) hearing screening. There is no consensus concerning the efficacy of childhood hearing screening, and its provision across countries in Europe is very diverse. Cost-effectiveness analysis could add insight, but data on important determinants needed for that are insufficiently collected, reported and shared.Citation8
Jill Carlton (University of Sheffield) presented the results of the EUSCREEN Survey regarding vision screening in 47, mostly European, countries. Many different professionals screen, related to the age of the child. The number of tests per screen varies between 1 and 19. The number of measurements of visual acuity per child varies between 1 and 32. Most vision screening takes place between age 3 and 7.
Implementation of neonatal hearing screening in three provinces in Albania
Birkena Qirjazi (University of Medicine, Tirana) discussed the implementation of neonatal hearing screening in three provinces in Albania, from her perspective as supervisor of neonatal hearing screening. She concluded that a neonatal hearing screening program can be successful only when the personnel involved is well trained, supported and monitored along the whole process. Raising the awareness of the general population is important in reducing the number of cases lost to follow-up. Raising the awareness of the government is important for continued support of an effective neonatal hearing screening program.
Martijn Toll (Erasmus MC, Rotterdam) presented the results from the perspective of the implementation of the neonatal hearing screening program in Albania. He concluded that neonatal hearing screening performed by nurses and midwives in maternity hospitals before discharge worked very well. Coverage was very high in the first screening step when the infants were still in the hospital after birth. However, loss to follow-up was significant in the second and third screening steps. It is imperative to keep track of infants and make sure follow-up appointments are made and attended. The rates of loss to follow-up differed considerably between screening nurses, indicating that supervision should be adequate and work overload should be avoided.Citation9
Implementation of vision screening in Cluj County, Romania
Jan Kik (Erasmus MC, Rotterdam) discussed the implementation study from the perspective of the implementation of the vision screening program in County Cluj in Romania. Altogether 12,866 4- and 5-year-old children had been screened. In the city of Cluj-Napoca, 74% of eligible children were screened, in the small cities 77% and in rural areas 47%. Of all children screened, 1,505 (12%) were referred. Only 43% of rural family doctors’ nurses had followed the screening course and only 26% screened children, none of the family doctors screened. In rural areas, parents often did not bring their children to the family doctors’ offices for screening. Later, the nurses went to the kindergartens to screen, but found few children there. After one year, halfway the study, screening had taken place in only 21 of 75 rural communes. A traveling screening nurse was employed to increase coverage in rural areas who then screened 805 children in 35 additional rural communes within eight months, substantially increasing rural coverage.
Maria Fronius (Goethe University, Frankfurt) presented the EUSCREEN protocol for measurement of visual acuity in children aged 4–5 for vision screening. This protocol has been agreed upon by all partners involved in the implementation of vision screening and is recommended for vision screening in the whole of Romania.
The implementation of the vision screening program for 4- and 5-year-olds in Cluj County was discussed by Mihai Mara (UMF-Cluj, Cluj-Napoca) from his perspective as supervisor of vision screening. Screening went well in urban areas, but was difficult and less successful in rural areas. As described, the employment of a traveling screening nurse brought a solution here. For implementation of screening in the whole of Romania, it is recommended that 4- and 5-year-old children will be screened by kindergarten nurses in urban areas and, in principle, by family doctors or their nurses in rural areas. Conditions should be improved for rural family doctors and their nurses, enabling them to include vision screening in their tasks. Until these conditions are improved, a traveling screening nurse should be employed in each county in Romania to screen children in rural areas where family doctors and their nurses currently cannot. The EUSCREEN protocol for measurement of visual acuity for vision screening should be used. A training program for orthoptists at one or more universities in Romania could be considered, as is in place in most European countries, to guarantee sufficient treatment capacity to treat all children with amblyopia detected by vision screening. Finally, treatment of amblyopia in young children with glasses and eye patches should be reimbursed by the national health care insurance.
Photoscreening and autorefraction used for screening
Bart van Overmeire (Agentschap Opgroeien, Brussels) discussed the vision screening program in Flanders. After having used a photoscreening device for several years, in 2018 Flanders switched to a smartphone app. A study among 12-month-olds will be carried out to determine the sensitivity and specificity of the smartphone app.
Parinaz Rostamzad (Erasmus MC, Rotterdam) studied the photoscreening that was in place in Flanders until 2018. Between 2012 and 2017, when the photoscreening was in place, the number of 4-year-old children wearing glasses increased from 4.7% to 6.4%, but it remains unknown how many cases of amblyopia have been prevented by prescribing more glasses at age one and two.Citation10
Zanda Ruskule (Children’s Clinical University Hospital, Riga) discussed the problem of the low coverage of vision screening in children in Latvia (30%) because there are too few ophthalmologists to screen all children. Vision screening in Latvia would be more accessible by involving optometrists in vision screening. This would lead to higher coverage and earlier discovery of vision problems.
Anna Horwood (University of Reading) presented the results of her review of photoscreening and autorefraction used for screening. Photoscreening has many advantages, but also many drawbacks and the subject is controversial. The main choice is the decision whether to invest in early, automated screening or in high quality training so that visual acuity can be tested accurately and efficiently in later childhood, but still within the critical period. The latter option is likely to be more cost-effective.Citation11
Annelies Bruinenberg (Erasmus MC, Rotterdam) presented her new randomized controlled trial, the Early Glasses Study, of the effect of glasses given to children at age one for high refractive error on the development of amblyopia, assessed at age four. In the current preparatory phase, many child healthcare centers, youth healthcare physicians and nurses, and research-orthoptists in the Netherlands have committed themselves to this study in order to assess (1) the association between refractive error at age one and visual acuity and amblyopia at age four, and (2) whether the prescription of glasses at for high refractive error at age one can prevent the development of amblyopia.
Treatment and demographics of amblyopia and other vision disorders in children
Sjoukje Loudon (Erasmus MC, Rotterdam) presented the first results of a randomized controlled trial comparing treatment of amblyopia with dichoptic games with conventional treatment with occlusion therapy, carried out by Aveen Kadhum (Erasmus MC, Rotterdam).Citation12 In this study among children with newly detected amblyopia, half of the children failed to complete the treatment with dichoptic games, mainly due to their young age. In countries with nationwide vision screening with high coverage such as the Netherlands, amblyopia is detected at age 4 or 5 and, hence, the applicability of treatment of amblyopia with dichoptic games is limited.
Jan-Roelof Polling (Erasmus MC, Rotterdam) presented the ophthalmological findings in a large population birth cohort, the Generation R Study. Among 6,690 children (average age 6.2 years, range 4.8–9.1) the most frequent findings were refractive errors, followed by strabismus. Because vision screening in the Netherlands has very high coverage and orthoptic treatment is good and widely available, the prevalence of undetected or insufficiently treated amblyopia in this large population birth cohort was low.
Dongsheng Yang (Shandong Liankang Eye Hospital, Jinan, Shandong) presented a new binocular therapy for refractory amblyopia. The new therapy consists of placing a neutral density filter in front of the better eye to reduce luminance, but not contrast for that eye. This improved visual acuity in many cases of refractory amblyopia. Further studies will be performed to assess whether this treatment can be recommended for routine use.
Implementation of vision and hearing screening programs in other countries
Luisa Monteiro (Hospital Lusiadas, Lisbon) reported on the implementation of a universal newborn hearing screening program in Portugal. Although universal neonatal hearing screening is now fully implemented in Portugal, it still lacks central monitoring and quality control. A working group was recently appointed to regulate the neonatal hearing screening program and to implement a preschool hearing screening program.
Lisa Rubin (Public Health Service, Jerusalem) presented the neonatal hearing screening program in Israel that was started in 2010. The program is considered a success as it has decreased the age of diagnosis and age at entry to rehabilitation of very young children and has also increased equality by lessening the differences in outcome of quality markers of screening between Arab and Jewish populations. However, the program is still in the need for a sustainable reporting system to monitor performance.
Csilla Serfőző (Heim Pal Childrens Hospital, Budapest) discussed vision screening in Hungary, where screening is performed by health visitors. While the program’s coverage is very high (nearly 99%), its effectiveness is not monitored. Also, there is no training program for orthoptists, the number of pediatric ophthalmologists is low and glasses with a strength less than six diopters spherical power are not reimbursed by the health care insurance.
Lieke Gouma (Rwanda Charity Eye Hospital, Kigali) discussed the obstacles to implementation of childhood vision screening in Rwanda. Currently there is no vision screening and there are also very few ophthalmologists. Before a vision screening program is implemented, several questions need to be answered: who could perform vision screening, where do referred children go for treatment, who will examine them for diagnostic assessment, who would prescribe glasses and eye patches and who would pay for the treatment?
Zia Chaudhuri (University of Delhi, New Delhi) discussed the new government program for all of India Vision 2020: The Right To Sight, which is primarily aimed at correcting refractive error at the age of nine to fourteen, which is myopia in most cases. With the current program, amblyopia is not detected at an age that it can be treated effectively: Treatment with patching the better eye and with glasses for amblyopia should start at age 6 or before. Therefore, with the EUSCREEN model for cost-effectiveness of vision screening (miscan.euscreen.org) it was calculated how much the additional costs would be when vision screening would start at age 5 or 6 instead of age 9. For the calculation it was assumed that 20% of children in one birth year will be included in the screening programme, 5.6 million children approximately; 10% of these 5.6 million children will already be wearing glasses and will not need screening: For the remaining 5,040,000 children to be screened the estimated budget would be €886,364 or €0.17 per screen test per birth year. The costs of diagnostic evaluation and treatment would be €355,068 in one birth year, in addition to the €886,364 for vision screening, together approximately €1,241,432 in one birth year or €6,207,160 in five birth years from 9 to 14 years. For the aim of detecting amblyopia at an age that it could still be treated, 5 or 6 years, the vision screening program including the costs of screening, diagnosis and treatment, would cost an additional €1.1 million per birth year or approximately €4.5 million to the existing costs of €6,207,160 for vision screening in children between 9 and 14 years of age. This would increase the scope of holistic screening and treatment of amblyopia in children between 6 to 14 years of age along with vision screening and correction of refractive errors with glasses in children between 9 and 14 years.
Conditions and cost-effectiveness of vision and hearing screening
Eveline Heijnsdijk (Erasmus MC, Rotterdam) presented the EUSCREEN cost-effectiveness model for childhood vision and hearing screening. She explained how the model works, as well as the differences between the vision and hearing models, and demonstrated this by showing the input and results of several model simulations.
Anna Horwood (University of Reading) discussed the costs of vision screening and costs of treatment of amblyopia, concluding that small changes in programs can dramatically change the costs. The main drivers of costs are tests, equipment, sensitivity and specificity, positive and negative predictive value, age and testability at screening, availability and costs of skilled screeners, number of children testable in a session and the costs of diagnostic assessment.Citation13 Costs post-screening are particularly influenced by the number of visits between referral and discharge, and the professional they see, so early screening and referral are generally much more costly long-term, for relatively small improvements in outcome.
Jan Kik (Erasmus MC, Rotterdam) looked at the current monitoring and data collection in vision and hearing screening programs in Europe as assessed with the EUSCREEN Survey and concluded that monitoring and data collection take place in relatively few countries. In a second step, the variables that are needed for cost-effectiveness analysis of vision and hearing screening program were identified and the results of the EUSCREEN Survey were screened for availability of data on these variables. Again, it was found that, for vision and hearing screening programs in most countries, little data is available for variables essential for cost-effectiveness analysis of screening programs. The EU should request collection of these data when stimulating implementation of new vision and hearing screening programs in member states.
Jill Carlton (University of Sheffield) reviewed the acceptability of childhood screening and concluded that this an under-researched area. Acceptability is often determined by assessing parental knowledge and understanding of the screening process, the testing procedure(s) and consent. The emotional impact of childhood screening is associated with maternal anxiety levels associated with the timing of the screening process and the impact of any false referral.Citation14
Cost-effectiveness model ‘jam session’
The final session of the meeting was a ‘jam session’ with the EUSCREEN cost-effectiveness model, moderated by Harry de Koning (Erasmus MC, Rotterdam). Several model simulations that were run by Country Representatives in the two weeks before the meeting were discussed that illustrated how the model works, how it should be used and, also, how it should not be used. Harry de Koning walked the attendees through simulations from Romania, Israel and Latvia. The simulations showed that the model is not always easy to operate, but that it does work. Even if users may have difficulties supplying values for important variables like specificity, as long as they use the same estimated values, comparisons between different programs with, for example, different screening professionals or different screening ages are still valid. The model demonstrations were followed by a lively discussion that included online participants and clearly illuminated the purpose and workings of the model. This proved to be a fitting end to a highly successful meeting.
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References
- Spaans J. Religious policies in the seventeenth-century Dutch republic. In: Hsia RP, van Nierop H, eds. Calvinism and Religious Toleration in the Dutch Golden Age. Cambridge: Cambridge: University Press; 2002:72–86. Chapter 5.
- Sloot F, Heijnsdijk E, Groenewoud JH, et al. The effect of omitting an early population-based vision screen in the Netherlands: a micro-simulation model approach. J Med Screen. 2017 Sep;24(3):120–126. doi:https://doi.org/10.1177/0969141316670422.
- Groenewoud JH, Tjiam AM, Lantau VK, et al. Rotterdam AMblyopia screening effectiveness study: detection and causes of amblyopia in a large birth cohort. Invest Ophthalmol Vis Sci. 2010 Jul;51(7):3476–3484. doi:https://doi.org/10.1167/iovs.08-3352.
- de Koning HJ, Groenewoud JH, Lantau VK, et al. Effectiveness of screening for amblyopia and other eye disorders in a prospective birth cohort study. J Med Screen. 2013 Jun;20(2):66–72. doi:https://doi.org/10.1177/0969141313497355.
- Sloot F, Telleman MAJ, Benjamins J, Sami A, Hoogendam JP, Simonsz HJ; Orthoptic Research Group. Effectiveness of routine population-wide orthoptic preschool vision screening tests at age 6-24 months in the Netherlands. Acta Ophthalmol. 2021. doi:https://doi.org/10.1111/aos.14859.
- Bussé AML, Mackey AR, Hoeve HLJ, et al.; EUS€REEN Foundation. Assessment of hearing screening programs across 47 countries or regions I: provision of newborn hearing screening. Int J Audiol. 2021 Mar 10;1–10. doi: https://doi.org/10.1080/14992027.2021.1886350.7.
- Mackey AR, Bussé AML, Hoeve HLJ, et al.; EUS€REEN Foundation. Assessment of hearing screening programs across 47 countries or regions II: coverage, referral, follow-up and detection rates from newborn hearing screening. Int J Audiol. 2021 Mar 9;1–10. doi:https://doi.org/10.1080/14992027.2021.1886351.
- Bussé AML, Mackey AR, Carr G, et al.; EUS€REEN Foundation. Assessment of hearing screening programs across 47 countries or regions III: provision of childhood hearing screening after the newborn period. Int J Audiol. 2021 Apr 9;1–8. doi: https://doi.org/10.1080/14992027.2021.1897170.
- Bussé AML, Qirjazi B, Goedegebure A, et al. Implementation of a neonatal hearing screening program in three provinces in Albania. Int J Pediatr Otorhinolaryngol. Jul 2020;134:110039. doi:https://doi.org/10.1016/j.ijporl.2020.110039.
- Bostamzad P, Horwood AM, Schalij-Delfos NE, Boelaert K, de Koning HJ, Simonsz HJ. Plusoptix photoscreener use for paediatric vision screening in Flanders and Iran. Acta Ophthalmol. 2020 Feb;98(1):80–88. doi:https://doi.org/10.1111/aos.14144.
- Horwood AM, Griffiths HJ, Carlton J, et al.; EUSCREEN Foundation. Scope and costs of autorefraction and photoscreening for childhood amblyopia-a systematic narrative review in relation to the EUSCREEN project data. Eye (Lond). Mar 2021;35(3):739–752. doi:https://doi.org/10.1038/s41433-020-01261-8.
- Kadhum, A, Tan, ETC, Levi, DM, et al. Barriers to successful dichoptic treatment for amblyopia in young children. Graefes Arch Clin Exp Ophthalmol. 2021. May 31. doi:https://doi.org/10.1007/s00417-021-05193-1.
- Horwood A, Lysons D, Sandford V, Richardson G. Costs and effectiveness of two models of school-entry visual acuity screening in the UK. Strabismus. Jul 2021;5:1–8. doi:https://doi.org/10.1080/09273972.2021.1948074.
- Carlton J, Griffiths HJ, Horwood AM, Mazzone PP, Walker R, Simonsz HJ. Acceptability of childhood screening: a systematic narrative review. Public Health. Apr 2021;193:126–138. doi:https://doi.org/10.1016/j.puhe.2021.02.005.