Equality in cleft and craniofacial care

Introduction

The European Union (EU) currently consists of 27 countries with a combined population of 446 million inhabitants^[1]. There are approximately 637,000-743,000 individuals living with a cleft lip and/or palate (for convenience referred to hereafter as “cleft”) in Europe and many others with other craniofacial conditions. Throughout their childhood and into their adult life, these individuals may benefit from the expertise of and care from a variety of specialists including surgeons (who may perform primary cleft surgery, revision surgery and further aesthetic surgeries) but also dentists, orthodontists, geneticists, nurses, paediatricians, psychologists, speech and language therapists, but their access to healthcare can vary greatly. For the general population in Europe, there is a significant variance in access to healthcare. When looking at the health expenditure per inhabitant in 2016, the countries with the lowest health expenditure were Romania (€400), Bulgaria (€600) and Poland (€700). In contrast, the countries with the highest expenditures were Luxembourg (€5600), Sweden (€5100) and Denmark (€5000)^[2]. The difference in resources can have an impact on the provision of care for cleft and craniofacial conditions and can therefore be important in determining health and social outcomes. In other words, the health and social outcomes of an individual with a cleft or craniofacial condition are dependent on the country in which they were born.

It is established that access to effective treatment for cleft and other craniofacial conditions varies widely throughout Europe, meaning that many children born with these conditions are never given the opportunity to realise their full potential. The concept of a comprehensive multi-specialist-team approach to care is not universal. Furthermore, infants with clefts are still institutionalised in some EU countries. A survey carried out for UNICEF in Bulgaria in 2011 showed that, in up to 40 per cent of cases where babies were born with a cleft, the parents were advised (in most cases by health professionals) to leave their baby in an orphanage. Via a fruitful collaboration, the European Cleft Organisation (ECO) and The Association of Children with Facial Anomalies and their Parents in Bulgaria (ALA) has helped reduce these numbers to 28 per cent through educating and training “front line” healthcare providers and families with children with facial anomalies. The work is ongoing^[3].

Clinicians and patients across Europe, especially in those regions that have limited healthcare expenditure, state that access to the necessary care pathways during the treatment span (from the moment of diagnosis to adulthood) is fragmented or in some cases non-existent in their countries. The importance of the early and adequate provision of information and treatment of clefts has been emphasised by the 2015 report of the European Committee for Standardisation (CEN)-Early Care Services for Babies Born with Cleft Lip and/or Palate. This involved input from 12 European countries and detailed the first agreed set of European guidelines in early cleft care^[4]. The minimum standards of care and best practice models presented in this document led to a European Science Foundation funded conference held in Bucharest in October 2015, attended by nurses working in the field of cleft in 17 European countries. Key outcomes of the meeting were the urgent demand for a Europe-wide programme of implementation of the guidelines together with protocols on how to evaluate progress, especially in Eastern Europe. In these respects, the outcomes mirrored those of the earlier Eurocleft project, involving 30 countries and 201 centres, which recommended the adoption of a common set of policy statements governing clinical practice for European cleft teams and practice guidelines detailing minimum standards of care^[5]. These recommendations, however, were never implemented on a consistent, Europe-wide basis.

Further evidence of the urgent need to improve cleft and craniofacial care in these countries was presented at a subsequent European Science Foundation funded meeting at CEN in Brussels in March 2016 attended by user groups, politicians and doctors from 13 European countries. It was emphasised that there is a need to adhere to evidence-based guidelines in order to develop national protocols. It was noted that there is still a lack of coordinated general measures in cleft care at a national level in many European countries including prenatal diagnosis, genetic advice, national registry development, equal access to treatment and transparent rules for treatment. There is a lack of coordinated multidisciplinary team protocols, deficiencies in information provision, both for professionals and parents, and deficiencies in long-term management. For example, it has been shown that the intervention of multidisciplinary team management in cases of women whose foetuses were diagnosed with cleft lip/palate on prenatal ultrasound screening was associated with these women being more likely to decide to continue their pregnancy than would have been expected if such intervention was not received^[6].

At the same time, the European Pillar of Social Rights states that “Everyone has the right to timely access to affordable, preventive and curative healthcare of good quality” p.21^[7]. There have been significant improvements in health in Europe due to advancing healthcare systems; however, access to healthcare remains uneven across countries and social groups, according to an individual’s socioeconomic status, place of residence, ethnic group and gender^[8]. Furthermore, it has been established that the lack of coverage and provision of certain types of care, such as cleft and craniofacial care, means that significant inequality remains in Europe. It is extremely important to acknowledge, however, that this inequality is not just between countries, but also within a country.

Inequalities are more present in socially marginalised groups; they are, for example (but not limited to), individuals who are unemployed, experience mental health problems, migrants or refugees and have disabilities. Belonging to a socially marginalised group also increases the risks of health inequalities, poverty and social exclusion. In 2017, 22.4 per cent of the European population (112.8 million people) lived in households at risk of poverty or social exclusion. In Bulgaria, Romania and Greece, more than a third of the population was at risk^[9]. When it comes to children, figures for 2018 show they are the group at highest risk of poverty or social exclusion in Europe^[10].

A fundamental factor that explains inequalities in healthcare is that the lower the income of an individual or family is, the more self-reported unmet healthcare needs they have. A descriptive example is that in Greece those who are in the lowest income quintile report 34.3 per cent unmet healthcare needs while those in the highest quintile report only 0.4 per cent^[8]. Currently, there exists evidence that large income differences result in poorer health and more negative social consequences^[11] and health inequities are increasing in many countries^[12]. One of the factors that contributes to belonging to a group that is at risk of being in poverty and social exclusion is an individual’s level of education^[13], since higher educational attainment often correlates with higher employment rates and higher earnings^[14]. It also depends on which region an individual resides, since the 2018 unemployment rates in EU regions vary from 1.3% to 35.1%^[15].

At the same time, research shows that individuals with cleft are at an elevated risk of poor educational outcomes in comparison to their peers^[16-19]. This carries the potential to influence the long-term outcomes for individuals with cleft, since they are then hypothetically at a higher risk of poverty and/or social exclusion if they do not succeed in their education.

Experiencing inequality, poverty or social exclusion is also associated with mental health problems. Currently, in Europe, more than one in six people experience mental health problems^[20], and, when it comes to mental health problems for individuals with cleft, there are population-based studies from Denmark and Sweden that show a significantly increased risk for having psychiatric and neurodevelopmental disorders^[21,22] as well as increased risk for being prescribed psychotropic medication^[23,24].

When individuals with a cleft or craniofacial conditions are seen by their healthcare providers, based on EU data for the general population, approximately 22 per cent are at risk for poverty or social exclusion^[25]. However, the real figures are probably higher since the research introduced above shows that some individuals with a cleft are at significant risk of not doing well academically and at risk of mental health problems, which are associated with a higher risk of experiencing inequality, poverty and/or social exclusion. The impact is also influenced by the access to healthcare, as well as the economic situation in the country in which an individual with a cleft or craniofacial condition resides.

The European Cleft and Craniofacial Initiative for Equality in Care (ECCE) is a pan-European multidisciplinary network with participants from hospitals and research institutions located in 27 European countries and beyond. ECCE is a 4-year project funded by the European Cooperation in Science and Technology (COST), promoting research and the development of innovation networks by providing support for arranging meetings, training schools and short-term scientific missions (STSMs) within the focus of the ECCE Action.

The ECCE Action is focusing on sharing scientific knowledge on treatment, research methods and organisational implementation in the different disciplinary groups in cleft and craniofacial care between the participants in the Action. The Action is made up of working groups to provide a structured environment within which mentoring and guidance will be offered to all the participants, with a particular focus on the early career investigators (ECIs) and researchers hailing from Inclusiveness Target Countries (ITC). This is aimed to facilitate the gaining of experience of leadership by participating in working group meetings and plenary sessions. In addition, as part of the COST framework, the Action organises conferences, short-term scientific missions and training schools targeting different aspects of equality in cleft and craniofacial care.

The european cleft and craniofacial initiative for equality in care

The activities that have been and are being undertaken as part of the ECCE Action are now introduced in order to consider how they contribute to the goals of the Action.

Conclusion

It is well established that disparities and inequalities exist within healthcare in the EU and subsist within cleft and craniofacial care. These extend to health expenditure, access to effective and multidisciplinary healthcare following a clear treatment pathway or protocol, the provision of long-term case management and the operation of national data registries. Such disparities exist between countries and within subsets of a national population. In turn, this may prejudice patients’ social and economic opportunities and, in cleft care, compound the impact of their condition on educational attainment and socioeconomic status.

Whilst the activities of the ECCE Action are ongoing, it has already enabled over 135 researchers and clinicians from 30 countries to attend one or more of the events facilitated by the Action, including training schools, short-term scientific missions and conferences, all related to the topic of equality in care. It is believed by the authors that this exposure to the experiences and healthcare cultures of such a diverse group of researchers and clinicians can only serve to increase awareness of this topic amongst those researchers, clinicians and trainees involved in the Action. In addition to facilitating individual awareness and, potentially, enabling those working clinically to carry this awareness into their practice, those involved in the Action are committed to sharing their learning with colleagues and, gradually, instigating organisational change at a local level in order to combat healthcare inequalities where possible.

The activities of the working groups, including the surveys and questionnaires and GCM exercises being undertaken, the STSMs and the long-term collaborations forged in performing this activity together as well as during conferences and training schools, will also lead to contributions to the literature on this topic. This collaboration between participants in the ECCE Action has resulted in four ongoing research or pilot endeavours concerning the access to and provision of care in cleft and craniofacial conditions and has generated six further EU grants comprising a total of 16 different country partners and a combined grant sum of 1,372,000 Euros. In turn, it is hoped that the immediate outputs of the ECCE Action as well as those stemming from partnerships it has helped create will increase awareness and encourage reflection, action and application within the clinical, research and healthcare policy spheres. These outputs will also be important in establishing equality of access to cleft and craniofacial care as a critical issue going forward and maintaining a focus on the importance of equality of care.

These achievements should be understood against the background of it being a significant challenge to attract funding to enhance knowledge of relatively rare medical conditions such as cleft and other craniofacial anomalies. In this respect, COST provides networking opportunities for researchers and innovators to strengthen Europe’s capacity to address scientific, technological and societal challenges. The principal areas of activity are promoting and spreading excellence, fostering interdisciplinary research for breakthrough science and empowering and retaining young researchers and innovators. COST implements its mission by funding bottom-up, excellence-driven, open and inclusive networks in all areas of science and technology. Whilst COST does not fund research time, it provides support for networking activities carried out within Actions and, as has materialised with the ECCE Action, increases the possibility of the topic under consideration being the subject of further applications for research funding.

The fact that COST puts significant emphasis on helping early career researchers and clinicians grow professionally is both unique and very important for topics such as equality of care. This is because there exists an opportunity to influence and shape future approaches to cleft and craniofacial care and research in Europe. For these groups, the training schools and short-term scientific missions have been, and will continue to be, powerful tools that can act as an introduction to cross-border collaboration and highlight future possibilities. Furthermore, COST also prioritises clinicians and researchers from less research-intensive countries. This has been hugely beneficial in bringing together researchers, especially in those parts of Europe that have hitherto not had the opportunity to exchange knowledge and ideas with counterparts in other countries due to limited resources. Indeed, funding is not only open to Action members but also a limited number of colleagues and researchers in countries represented in the Action. Grants to attend the conferences and training schools and take part in STSMs are usually sufficient to cover most, if not all, of the costs of travel, accommodation and subsistence, which ensure networking opportunities are genuinely open to everyone eligible. In this sense, the ECCE Action has been able to encourage and foster opportunities and equality within its own members and its own activities, a critical requirement given the focus of this Action upon equality.

A vital component of the Action is to generate awareness about the inequalities of care that exist between and within countries in Europe and beyond. In addition to the activities detailed, members of the Action are participating in dissemination actives, including key conferences around the world and with the European Parliament. The ECCE Action network is, however, not closed but remains open. Interested clinicians and researchers, from the EU and beyond, who wish to learn more and/or collaborate on this project or on future projects are invited to contact the corresponding author and discuss these possibilities.