Developing research priorities for palliative care of people with intellectual disabilities in Europe: a consultation process using nominal group technique

There have been mounting concerns in recent years about inequalities in the provision of healthcare for people with intellectual disabilities [1‐5]. People with intellectual disabilities are more likely to have poorer health outcomes than the general population, for reasons including poor access to healthcare services [3], poor quality healthcare [6], lack of advance care planning [6], lack of staff understanding of their needs [7‐9], and diagnostic overshadowing (where the symptoms of physical ill-health are attributed to the presence of intellectual disability, and therefore not treated or managed [10]).

Intellectual disability affects approximately 1–3% of the population [11]. The term covers a wide range of abilities and disabilities, skills and limitations, but always includes the following three aspects: (a) a significantly reduced ability to understand new or complex information and to learn and apply new skills (impaired intelligence); (b) a significantly reduced ability to cope independently, expressed in conceptual, social, and practical adaptive skills (impaired adaptive functioning); and (c) early onset (before adulthood), with a lasting effect on development [12, 13].

The specific palliative care needs of people with intellectual disabilities have only recently become a focus of research attention. Early work in this field was concerned with the impact of bereavement on people with intellectual disabilities [14]. The earliest reported research within the English-language literature related to intellectual disability, death and dying date from the 1980s and looked at patterns of morbidity and mortality within institutionalised populations [15]. The first large scale mortality studies that compared data of people with intellectual disabilities with those of the general population came at the turn of the century from Finland [16] and Ireland [17], where national registers of the population of people with intellectual disabilities made this possible. Countries without such registers face significant methodological difficulties in making such comparisons [18, 19]. Most such studies reported earlier deaths among people with intellectual disabilities.

The first case reports of adults with intellectual disabilities who were dying and in need of palliative care were published in the 1990s [20, 21], but research studies in this field did not appear until this century. A literature review in 2007 revealed that of the 45 identified journal papers, book chapters and web-based articles with a primary focus on palliative care issues for people with intellectual disabilities, only seven of the materials were research reports [22]. Although research activity in this area has been growing steadily in the past decade, there is still only limited empirical evidence about applying the principles of palliative care to people with intellectual disabilities who have palliative care needs [23]. It has been argued that the lack of research interest in death, dying and intellectual disability stems from concerns that it is too emotive, as well as from the fact that the lives and deaths of people with intellectual disabilities have been hidden from society until the time of deinstitutionalisation [24, 25].

The palliative care needs of people with intellectual disabilities may be no different from those of the rest of the population, but they often present with unique challenges and circumstances that make it more difficult to meet those needs. A recent review of both USA and European literature found that people with intellectual disabilities face unique barriers in accessing hospice care, including delays in diagnosis of life-limiting conditions, the hospice providers’ knowledge of this patient group, issues around communication, and ethical dilemmas including issues around informed consent [26]. Difficulties with communication and comprehension affect the assessment and treatment of pain and other symptoms [27, 28]. The complex issues around ethics, decision making and patient involvement have become a more recent focus for research [29‐31]. There is evidence that care at the end of life is of poorer quality for people with intellectual disabilities compared with the general population [32].

A significant proportion of the research studies to date is focused on the experiences of staff providing palliative care to people with intellectual disabilities, highlighting the training needs of palliative care professionals who may have limited experience of providing care for this population and the importance of collaboration between intellectual disability services and palliative care services [8, 26, 33‐36].

There are a number of challenges in researching the palliative care needs of people with intellectual disabilities. One of the difficulties, especially in an international context, is the lack of consensus on terminology and variation in service models of palliative care [37, 38] and intellectual disability [39]. In some countries, for example, the definition of intellectual disabilities may or may not include people with mild intellectual disabilities or people on the autistic spectrum who don’t have additional intellectual disabilities. There are various ways in which the population of people with intellectual disabilities might be identified, such as through access to specialist services or medical records; each have their limitations. Some countries have highly developed palliative care services; in others, no such services are available even to the general population [40]. This makes it difficult to make international comparisons.

In order to understand the issues involved, much of the research has used samples of services, staff and carers for interviews or surveys. Investigating the perspectives of people with intellectual disabilities themselves presents significant methodological and practical difficulties [41], although these are not unsurmountable [42, 43]. Another difficulty has been the lack of understanding and agreement on what constitutes good palliative care for people with intellectual disabilities and a lack of common outcome measures for assessing this. Finally, in assessing the current state of knowledge on an international level, there is clearly a limitation in only accessing English-language literature, which is likely to skew insights towards those of English-speaking countries.

Setting a research agenda for palliative care of people with intellectual disabilities is important for a number of reasons. Firstly, it addresses a gap in national strategies for palliative care and research, where there appears to be a distinct lack of focus on the needs of people with intellectual disabilities. There are research gaps for the general population too, but people with intellectual disabilities are often explicitly excluded from research populations, including palliative care research. Secondly, obtaining a consensus on the most important areas for research ensures that existing research expertise in this area is well used and may encourage researchers new to these topic areas to help expand the knowledge base. Thirdly, it informs and encourages international collaboration, and stimulates mutual sharing of expertise. This is particularly important in a European context where research and clinical expertise are unevenly distributed, with most developments originating in North West Europe. International collaboration in research has a wide range of benefits, including knowledge transfer, developing culturally relevant palliative care services, facilitating successful funding applications and establishing networking mechanisms that enable multi-country comparative studies [44]. Finally, identifying consensus research priorities may improve the possibilities of attracting funding in areas where it is most needed.

For over a decade, researchers in this field have shared their findings at dedicated “Death and Dying” symposia during international conferences, in particular through the European Association of Palliative Care (EAPC) and the International Association for the Scientific Study of Intellectual and Developmental Disabilities (IASSIDD). There have been several international research collaborations. In recognition of the barriers people with intellectual disabilities face in accessing palliative care services [45], the EAPC convened a Taskforce on Intellectual Disabilities in 2012, consisting of clinical and academic experts in the field. The taskforce developed consensus norms for palliative care of people with intellectual disabilities in Europe, which has been published as a White Paper [46]. Following this, a group of 16 researchers and clinicians in the field of palliative care for people with intellectual disabilities gathered for round-table workshops to share experiences and knowledge, assess the existing body of research, identify the challenges in conducting research in this field and strengthen international collaboration and networks. The meeting was convened in response to a desire by European researchers to develop joint projects. Part of this process was to work towards proposing a consensus research agenda. This paper reports on the outcome of one of the workshops, focusing on the following question that was posed: “What are the research priorities for palliative care of people with intellectual disabilities in Europe?” There are clearly limitations related to the small size of the project. However, the authors believe that it is important to share work of this kind in order to help researchers, policy makers and practitioners direct their focus towards some of the most pertinent issues. Resources in this area are scarce and need to be well-used.

NGT [47] uses a democratic process to form a set of collective priorities. The highly structured technique allows for the sharing of ideas from groups of people who have insight into a specific area of interest, which are then discussed and ranked by the group, producing consensus decisions [48]. NGT methodology has been used successfully to determine research priorities in healthcare [49‐53], including palliative care in under-researched areas [44]. Within palliative care settings, it has also been used to identify outcome indicators [54], patient and carer preferences for end of life care [55‐57], and staff perspectives on the information needs of family caregivers [58].

NGT has a number of advantages over other group methods. It prevents domination by more vocal or powerful members of the group and avoids conformity or social pressure [59, 60]. It is time and money efficient, as it is a single-occasion process that uses few resources to acquire a substantial amount of information in a relatively short time [48]. It requires little preparation by the participants and allows for immediate dissemination of results to the group, which makes it a highly satisfying method for the participants [48].

NGT in this project lasted approximately two hours and was facilitated by the first author, who was experienced in using the methodology [55]. Following explanation of the process, the NGT session was conducted in five steps.

Each participant allocated between one and five points to their five selected research themes, with their first preference receiving five points and their fifth preference receiving one point. All scores were added together, resulting in a group ranking of priorities. The number of votes each theme received was also noted, as this indicated not just the strength but the breadth of support for the theme.

Ethical issues around end of life decision making emerged as a key research priority. There is evidence that people with intellectual disabilities are not always sufficiently involved in end of life decision making, that assumptions are sometimes made about their quality of life and ability to cope with treatment, and that potentially life-saving treatments are not always offered because the person has intellectual disabilities [6, 62, 63]. The consequences are potentially extremely serious. They include, at worst, a risk of premature death; but there is also a risk of compromised quality of life if, for example, people are not offered treatments to ameliorate debilitating symptoms.

At present, most official systems and services, including palliative care services, do not usually identify which of their service users have intellectual disabilities. They are, in effect, an invisible population and as such, there is a danger that their specific needs are overlooked, not seen as a priority or even potentially perceived as a problem [46]. In addition, by being unable to extract data at local, regional or national levels, we have little idea about the patterns of use (relative to the general population) of palliative care services for people with intellectual disabilities. The issue is confounded by a lack of insight into where people with intellectual disabilities currently live and die, and who is involved in their care and support at the end of life. There are likely to be huge variations across Europe, affected in part by variation in societal attitudes to disability and variations in palliative care provision for the general population. Mapping the scale and scope of the issue is a prerequisite for engendering the system and policy changes needed to ensure that all people with intellectual disabilities have the opportunity to access appropriate palliative care support and services that meet their specific needs.

Research to date has highlighted unique challenges in achieving high quality palliative care for people with intellectual disabilities, as described at the start of this paper. There are real contextual differences between countries and cultures, but the extent to which this would influence models of best practice is uncertain. Investigating the quality of palliative care for people with intellectual disabilities in a wide range of European countries will enhance understanding of how good practice can be achieved, regardless of settings, circumstances or cultures. Researchers have started to investigate the perspectives and experiences of people with intellectual disabilities themselves, their families, care staff and professionals, but publications originate mostly from English-speaking countries.

Unless there are outcome measures and instruments for palliative care that are appropriate and relevant to the circumstances of people with intellectual disabilities, it will not be possible to assess and compare the palliative care received by people with intellectual disabilities (between settings, regions and countries as well as in comparison with palliative care for the general population).

It will be important to assess how the proposed research priorities align with the priorities of national agencies and governments across Europe (for example, with national palliative care research agendas, such as the UK Palliative and end of life care Priority Setting Partnership [61]) as this would give further leverage for obtaining research support. Careful consideration is then needed to determine which of the identified research themes (or sub-themes) need European-wide collaborations, and which specific research questions are best answered within national borders, given the wide variation in service provision and palliative care development [40]. Each of the four research priorities described above could usefully be addressed in a range of European countries and could benefit from international collaboration. Population studies benefit from greater numbers of participants; this is particularly salient in a field where numbers of people with intellectual disabilities within research studies might be too low for powerful statistical analysis. Analysis of palliative care provision across European borders, including aspects related to overall quality and to specific ethical issues and decision making, may highlight whether any barriers or issues are specific to local contexts, or whether they are experienced on a larger scale. This may help with the development of workable improvements. Research collaborations provide a powerful opportunity for sharing lessons learnt and thus enhancing practice.