On Monday 28th and Tuesday 29th, 2021, the Final Dissemination Meeting of the EUSCREEN Study took place in Zaandam. As a hybrid event, the meeting was also streamed live to a worldwide audience. Fortunately, the limitation of 50 participants for the meeting had been lifted on June 26th, but many countries still were ‘orange’ on the COVID-19 chart and, hence, the Country Representatives from these countries could not attend. 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 told the attendees that the first school doctor in the Netherlands, appointed April 1st 1904, worked at 250 metres from the congress venue in Zaandam, making Zaandam the best place to hold this meeting about childhood screening.
Next, Marius Romijn explained the history of the ‘Baptist Admonition’, the wooden church where the meeting took place. The Baptists, like other groups outside of the Dutch Reformed Church, were subject to restricted toleration in the Netherlands in the 17th century, so their churches could not be conspicuous1. This church was built in 1687 after a merger of several Baptist communities in the region. The organ was built in 1784 by the famous Johannes Petrus Künckel.

 

Preparation of the EUSCREEN study

The programme kicked off with a session about the preparation of the EUSCREEN Study by study coordinator Herb Simonsz (Erasmus MC, Rotterdam). He discussed the first cost-effectiveness model for vision screening in the Netherlands, a micro-simulation model developed in 2008 by Frans-Willem Goudsmit (Erasmus MC, Rotterdam) to calculate the effectiveness of a screening programme based on estimates of age-specific incidence of amblyopia, age-specific sensitivity of examination technique and age-specific effect of occlusion treatment 2, derived from observational data such as the RAMSES study3 4. In addition, Herb Simonsz presented the results of the OVAS study by Frea Sloot (Erasmus MC, Rotterdam)5, a Dutch disinvestment study involving two consecutive birth cohorts (N=11,000) who received regular visual acuity screening but only the first cohort was screened with orthoptic tests between the age of 6-24 months. This study showed that the effectiveness of vision screening was not influenced by the omission of orthoptic tests at the age of 6-24 months. Refractive and bilateral amblyopia were found almost exclusively by visual acuity measurement, whereas strabismic amblyopia was detected mainly outside the screening by parents and others.

 

EUSCREEN Survey of hearing and vision screening programmes

The EUSCREEN Study developed a model to perform comparative, cross-border, cost-effectiveness analysis of paediatric vision and hearing screening programmes in all countries in Europe and beyond. To populate the model with data, a survey was conducted among experts from 49 mostly European countries.
Opening the second session, André Goedegebure (Erasmus MC, Rotterdam) presented the results of the EUSCREEN Survey pertaining to neonatal hearing screening programmes in Europe. He found that neonatal hearing screening has been implemented in most European countries, where most infants are screened in hospital by nurses within three days from birth and finally concluded that the extent and complexity of neonatal hearing screening programmes are primarily related to countries’ health expenditure and Human Development Index score6. Inger Uhlén (Karolinska Institute, Stockholm) continued on this subject. She concluded that referral rates are related to age, test method and number of screening steps, as well as that quality measures were not available for many neonatal hearing screening programmes. Coverage rates were reported from 26 out of 47 countries or regions, referral rates from 23, follow-up from 12 and detection rates from 137.
Allison Mackey (Karolinska Institute, Stockholm) reported on the EUSCREEN Survey of childhood (preschool or school-age) hearing screening. She concluded that there does not appear to be consensus concerning the efficacy of childhood hearing screening, given the diversity of its provision across Europe. A cost-effectiveness analysis and comparison could add insight, but data on important determinants are not collected, reported or shared8.
Jill Carlton (University of Sheffield) presented an inventory of the survey of vision screening in all countries in Europe and a few beyond. The key findings she presented were that the content of screening programmes varies enormously, there are countries or regions where no vision screening occurs and, where there are vision screening programmes, the data show differences in who, what, when and where.

 

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 the screeners’ perspective. She concluded that neonatal hearing screening is not only about devices, but more about people and systems. It is a dynamic process that has to be analysed, monitored and adapted in response to the factors that may influence its progress.
Martijn Toll (Erasmus MC, Rotterdam) presented the results from the perspective of the implementation study. He concluded that neonatal hearing screening performed by nurses and midwives in maternity hospitals before discharge worked very well. Initial screening coverage was high. However, loss to follow-up was significant. It is imperative to keep track of infants and make sure follow-up appointments are made and attended. Supervision should be adequate and work overload should be avoided9.

 

Implementation of vision screening in Cluj County, Romania

The implementation of vision screening in Cluj County was discussed by Mihai Mara (UMF-Cluj, Cluj-Napoca) from the screeners’ perspective. He concluded screening went well in the urban areas, but was less successful in the rural areas. For implementation of screening in the whole of Romania, it is recommended that children are screened by kindergarten nurses in urban areas and by family doctors or their nurses in rural areas. Conditions should be created to enable all family doctors or their nurses to screen and until then, a travelling screening nurse should screen where family doctors or their nurses cannot. The new EUSCREEN protocol for measurement of visual acuity should be implemented. Orthoptists should be trained to ensure children identified through screening can be treated. Treatment should be free of charge.
Maria Fronius (Goethe University, Frankfurt) presented the recommended procedure for measuring visual acuity. Based on the experiences with the implementation in Cluj, a procedure for visual acuity assessment has been agreed upon by all partners involved in the implementation, which will be recommend to the government in Romania for implementation of vision screening in the whole country.
Jan Kik (Erasmus MC, Rotterdam) discussed the implementation study from the perspective of implementation. Altogether 12,866 children were screened: in Cluj-Napoca 74% of eligible children, in the small cities 77% and in rural areas 47%. Of all children screened, 12% were referred; ophthalmologists’ diagnostic assessments were received for only 28%. Vision screening was considered acceptable by all stakeholders, but opinions differed on its sustainability. Screening was feasible in urban kindergartens because there are resident nurses, but not in rural areas, where there are no nurses in kindergartens. Adoption was good where personnel was available.

 

Photoscreening and autorefraction used for screening

First, Bart van Overmeire (Opgroeien, Brussels) discussed the vision screening programme 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 (N=500) will be carried out to determine the sensitivity and specificity of the smartphone app.
Parinaz Rostamzad (Erasmus MC, Rotterdam) presented the results of her study on the photoscreening that was in place in Flanders until 2018. She noticed that it remains unknown how many cases of amblyopia have been prevented by prescribing glasses at ages one to two and a half, whereas the number of children wearing glasses has increased considerably10.
Zanda Ruskule (Children’s Clinical University Hospital, Riga) discussed the low coverage of vision screening in children (30%) in Latvia because there are too few ophthalmologists to screen all children. Vision screening in Latvia would be more accessible by involving optometrists. This can lead to higher coverage and earlier discovery of vision problems, with less effort from parents.
Anna Horwood (University of Reading) presented the results of her review of photoscreening. 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-effective11.
Annelies Bruinenberg (Erasmus MC, Rotterdam) presented her randomised controlled trial (Early Glasses Study) on the effects of glasses given to children at age one for high refractive error on the development of amblyopia. Sufficient child healthcare centres, youth healthcare physicians and nurses and research orthoptists have committed themselves to this study in order to investigate whether the prescription of glasses at age one can prevent the development of amblyopia.

 

Treatment and demographics of amblyopia

Sjoukje Loudon (Erasmus MC, Rotterdam) presented the randomised controlled trial comparing dichoptic games with occlusion therapy for amblyopia, which was carried out by Aveen Kadhum (Eraasmus MC, Rotterdam)12. They found that in this study among children with newly detected amblyopia in the Netherlands, half the children failed to complete dichoptic treatment, mainly due to their young age. In countries with nationwide screening where amblyopia is detected before age six, the applicability of dichoptic treatment is limited.
Jan-Roelof Polling (Erasmus MC, Rotterdam) presented ophthalmological findings in a very large population birth cohort, the Generation R Study. The most frequent findings were refractive errors, followed by strabismus. Paediatric vision screening strongly reduced the prevalence of insufficiently treated or undetected amblyopia.
Dongsheng Yang (Shandong Liangkang Eye Hospital, Jinan) presented a new binocular therapy for refractory amblyopia. The therapy consisted of placing a neutral density filter in front of a the better eye to reduce luminance, but not contrast for that eye. This improved vision for refractory amblyopia, but further study is required before it can be routinely used.

 

Implementation of vision and hearing screening programmes in other countries

Luisa Monteiro (Hospital Lusiadas, Lisbon) reported on the implementation of a universal newborn hearing screening programme in Portugal. Although universal neonatal hearing screening is fully implemented in Portugal, it still lacks central monitoring and quality control. A working group was recently appointed to regulate the neonatal hearing screening programme and to implement a pre-school hearing screening programme.
Lisa Rubin (Public Health Service, Jerusalem) presented the neonatal hearing screening programme in Israel, that was started in 2010. The programme is considered a success as it has decreased the age of diagnosis and age at entry to rehabilitation and also has increased equality by lessening the differences in quality markers between Arab and Jewish populations. However, the programme still needs a sustainable reporting system for monitoring performance.
Csilla Serfőző (Heim Pal Children’s Hospital, Budapest) discussed vision screening in Hungary, where screening is performed by health visitors. While the programme’s coverage is very high (nearly 99%), its effectiveness is not monitored. Also, there is no training programme for orthoptists, the number of paediatric ophthalmologists is low and glasses with less than six dioptres spherical lenses are not reimbursed by the health insurance.
Lieke Gouma (Rwanda Charity Eye Hospital, Kigali) discussed the obstacles to childhood vision screening in Rwanda. Currently there is no vision screening and there also are very few ophthalmologists. Before a vision screening programme could be implemented, several questions would have to be answered, such as who can perform screening, where do children go If they need a referral and who will examine them, who can prescribe glasses or an eye patch and who can pay for the treatment?
Zia Chaudhuri (University of Delhi, New Delhi) presented the possibilities of detecting amblyopia in India by screening at an earlier age than the new government programme for all of India Vision 2020: The Right To Sight does, which is mainly aimed at correcting refractive errors at ages nine to fourteen. Based on several assumptions, a calculation with the EUSCREEN model showed that the costs of diagnostic evaluation for amblyopia and its further management in one birth year would be €355,068 in addition to €886,364 for the existing vision screening programme, thus costing approximately €1,241,432 in one birth year/€6,207,160 in five birth years from 9 to 14 years. Further, if the amblyopia screening programme was started at an earlier age, say at 6 years of age, it would add approximately €1 million per birth year. Thus, the total costs of this additional programme for amblyopia screening, diagnosis and management would add approximately €4.5 million to the existing costs of €6,207,160 for refractive error screening in a cohort of children who are between 9 to 14 years of age. This would increase the scope of holistic screening and management of amblyopia in children between 6 to 14 years of age along with visual screening and refractive errors management of children between 9 to 14 years, as targeted in the existing visual screening programme.

 

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 programmes 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 assessment13. 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) discussed the current monitoring in paediatric vision and hearing screening in Europe and what more would be needed for comparative, cross-border, cost-effectiveness analysis. The EUSCREEN Survey showed that for most programmes, these data are currently not available. A standardised set of variables should be collected in all screening programmes, in a uniform manner, to facilitate this. Digital data collection is preferable as this is efficient, minimising costs and administrative workload. Digital safeguards can be employed to minimise errors and to enable real-time quality assurance.
Jill Carlton (University of Sheffield) presented the results of her review of the acceptability of childhood screening, concluding that this an under-researched area. Acceptability was often determined by assessing parental knowledge and understanding of the screening process, the testing procedure(s) and consent. The emotional impact of childhood screening explored maternal anxiety levels associated with the timing of the screening process and the impact of any false referral14.

 

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 clearly 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 programmes 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.

 

The EUSCREEN Study was funded by the European Union’s Horizon 2020 research and innovation programme under grant agreement No. 733352.

 

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