People living with young-onset dementia (YOD) face unique challenges and may experience dramatic changes to social status, identity, and family roles (Kitwood, 1997; Rossor et al, 2010; Smeybe and Kirkevold, 2013). YOD often manifests when people are in their 40s and 50s, meaning most will be in active employment and meeting familial and financial obligations (Sikes and Hall, 2018). YOD often has more atypical features (Snowden at al, 2011). There is a wider range of early dementia symptoms and differential diagnosis (Draper et al, 2016). Early changes—for example, non-amnestic changes, or changes in mood (Draper et al, 2016)—mean that timely diagnosis of YOD is a challenge (Tellechea et al, 2018).
Once diagnosis is received, planning pathways of care remains problematic, because many of the typical dementia supports are either not available or not appropriate (Rossor et al, 2010; O'Shea et al, 2018). Because dementia is equated with advancing age, services are designed with this population in mind, and so models of care for those under 65 are often overlooked (Withall et al, 2014; Carter et al, 2018). In addition, the lack of clear and age-appropriate models of services means that the unique needs of the person, and their family is often overlooked (Bakker et al, 2010; Harris and Keady, 2009).
Research relating to dementia, psychosocial interventions and models of care largely focuses on the needs of older people (Rossor et al, 2010; Whitehead, 2016; Mayrhofer et al, 2018). Although there is some valuable research on YOD (Johannessen et al, 2017; Millenaar et al, 2017; Sakata and Okumura, 2017; Carter et al, 2018), as recently as 2018, the nursing literature has highlighted the need for more awareness and research in this area (Kobiske and Bekhet, 2018). According to Carter et al (2018) the continued lack of international policy focus for people living with YOD results in significant social and psychological distress for those impacted.
At present, there is little insight into how models of care are enacted for YOD, and how health professionals offer support to people living with YOD and their families, within community and dementia services. Increased understanding on the unique service needs for persons living with YOD may help inform how health professionals plan and deliver YOD support.
Aim
The aim of this study was to explore health professionals' perceptions of the key challenges faced by people living with YOD and their families, and how they enact support for this group within community and dementia models of care.
Method
Study design
This study used a qualitative approach. Semi-structured interviews were held with health professionals, and interpretative phenomenological analysis (IPA) methodology was used. IPA uses in-depth data collection methods (such as individual interviews) with the researcher collecting detailed, reflective accounts from the participant, as well as acknowledging their own reflections or preconceptions in the process (Volpato, 2018). IPA was chosen as it facilitates the exploration of specific social contexts (Johnson, 2016), the subjective experience of the individual, and how interviewees make sense of that experience (Shinebourne, 2011). IPA has been used previously when exploring the experience of people living with dementia (Johnson, 2016). It has also been used successfully to explore the perspectives of health professionals who work in challenging health service contexts (Volpato et al, 2018).
Sample and setting
Convenience sampling was used to recruit health professionals from public, voluntary and private sector services within the Republic of Ireland. The Alzheimer's Society of Ireland acted as gatekeeper. Initial potential participants (n=14) were identified and invited to participate. Of these, nine agreed to participate (five either did not respond or cited lack of availability). Within this final sample, five were purposively selected, owing to their professional role working with people with dementia/YOD. The remainder (n=4) were recruited via snowballing sampling, having been informed of the study by other participants via their professional networks. To meet inclusion criteria, participants needed to be working as a health professional, to have a familiarity of dementia and community services, and to have more than 3 years' experience of working with individuals living with YOD and families. The IPA methodology allows for richness, pragmatism and the formation of a sample group for whom the research question holds relevance (Pietkiewicz and Smith, 2014). In accordance with this approach, the sample was not selected for representativeness, rather on the ability to represent a perspective on a unique phenomenon (YOD models of care) and to provide a rich description of the context and experience under exploration (Eatough and Smith, 2008; Boland and Levack, 2012).
Ethical considerations
The institution's Research Ethics Committee granted ethical approval for this study before commencement of data collection. Permission to approach participants was sought and granted by the relevant services a priori. The interviews were conducted by the first author who had experience of IPA interviewing, and there was no prior or existing relationship with participants. Participation was voluntary. Participant information leaflets were supplied prior to interviews, and written, informed consent was obtained from all interviewees.
Data collection
Semi-structured interviews were undertaken with health professionals (n=9). All interviewees were interviewed alone and in a setting of their choice. All interviews were tape-recorded with permission and transcribed verbatim by the first author. A semi-structured interview schedule was used, however, in accordance with IPA guidelines (Eatough and Smith, 2008; Pietkiewicz and Smith, 2014), this was not prescriptive. The schedule was flexible, with open-ended questions, and was used only as a guide to ensure richness of interview content. Items included questions on diagnostic processes, post-diagnosis pathways, barriers and facilitators to good models of care. Probes were used to encourage elaboration, and interviews allowed time for pause or reflection. Interviewees were offered the opportunity to review transcripts for comment or correction. Duration of interviews ranged from 40 to 60 minutes. Eight interviews were conducted face-to-face and one was via telephone. A reflective diary was kept in order to record details of where and how emergent interpretations originated. All interviews were conducted by a health professional with experience in conducting qualitative research.
Enhancing trustworthiness
The authors aimed to enhance trustworthiness as outlined by Guba (1981) with the following quality strategies (Shenton, 2004; Korstjens and Moser, 2018). Credibility was optimised by emphasising the interviewer's independent status of researcher, and ice-breakers were used to establish rapport at outset. The authors aimed to enhance transferability by offering a rich description of participant accounts, and the research process. Dependability was optimised by ensuring the primary author and one coauthor (an experienced researcher) reviewed the coding process and analysis. An audit trail was maintained throughout the research process in order to try to optimise confirmability (Korstjens and Moser, 2018).
Data analysis
Data analysis was conducted using IPA guidelines (Smith et al, 2009), which helped to ensure structure and reliability. Analysis was conducted using a layered, inductive approach. Analysis was undertaken manually and involved a process of reading interview transcripts line by line, for immersion in the dataset. Early coding consisted of reading and recording notes within the transcript margins, to highlight emergent concepts. An initial list of emerging themes was compiled for each transcript. Once all transcripts were reviewed, related themes were reviewed together, and broad themes constructed. As with other IPA analyses (Pietkiewicz and Smith, 2014), this process involves several revisions. With each iteration, themes were refined and consolidated, and analysis continued throughout time of write up. On completion, analysis resulted in the final construction of hierarchical primary themes and contributing subthemes.
Results
Participant characteristics
Nine health professionals from a variety of professional disciplines were interviewed: nursing (n=2), medicine (n=1), psychology (n=1), academia (lecturer/practitioner) (n=1), primary care (n=1), private sector (n=1) and dementia services staff (n= 2). All interviewees had experience of working directly with people living with YOD or their families. The sample captured health professionals who work with people across the trajectory of the illness from time before diagnosis (in the community) to mid-stage dementia (day care, community care) and as the illness progressed (long-term care). Findings provided an overview of some of the key challenges that arise in YOD service provision. These relate to challenges obtaining timely diagnosis, impact of change on individual and family, and challenges enacting YOD-relevant post-diagnostic support. Findings are discussed across three themes: ‘beginning the journey’, ‘working with constraints’, and ‘new future’ (Figure 1).
Beginning the journey
Interviewees discussed their experience of supporting people with YOD during the initial stages of diagnosis. Two subthemes were constructed, and these related to initial diagnosis challenges, and to post-diagnosis pathways. Interviewees shared their own experiences of the emotional impact of diagnosis.
Barriers to diagnosis
Diagnosis in YOD was described as complex and multi-factorial. The conceptualisation of dementia as an older person's condition was seen to impede timely diagnosis. In addition, limited awareness resulted in long and difficult pathways to diagnosis. This was reflected in the following excerpts.
‘Diagnosis is slower to be reached for young people … One of my patients … for about 3 or 4 years were misdiagnosed. The assumption was because they were young, they didn't have … early-onset dementia.’
‘People [health professionals] don't have a high index of suspicion for that diagnosis in younger people.’
Other barriers to diagnosis were a lack of clarity among community-based health professionals about the correct pathway for referring persons under the age of 65 for assessment, and where the expertise and responsibility lay. Some health professionals had a remit for assessing and monitoring early issues or changes in their patients, and described it as ‘a very hard area to broach’ (health professional 7). Interviewees were vocal about the unique emotional context, voicing their own personal and professional reluctance in naming the dementia.
‘As a GP, I know these people … I feel devastated for them. I often feel inadequate in … dealing with it … it's not the diagnosis you make on the spot … Even using the word “dementia” with the families and the patients … it's like saying “cancer” to them … I would often feel uncomfortable.’
Interviewees described witnessing difficult early periods experienced by the individual and the family. There was a sense that this time awaiting diagnosis was characterised by missed opportunities to offer clarity, and to start decision-making.
‘In the meantime … people are left in this awful limbo, they've huge levels of stress, not knowing what's going on.’
Post-diagnosis pathways
In many instances, even when diagnosis was in place, care planning was problematic. In the early stages, along with information, models of care consist of signposting and referrals; however, here it was evident that specific YOD services were not readily available. This triggered frustration among professionals who felt unable to relieve the stress experienced by their clients. A psychologist compared this with diagnosis of other life-altering conditions.
‘If somebody gets a diagnosis of cancer … a huge support network kicks in and everybody … professionals and society at large. There's a sense of people rallying round, providing all kinds of input.’
Interviewees were conscious of the implications the diagnosis would bring, and what they themselves should be doing to support the person. They were aware of what a good model of care might look like but were also frustrated that this could not be enacted.
‘People might become very depressed post diagnosis, or become very anxious …We should be trying to help people … come to terms with (the diagnosis) … Maximising their strengths and abilities and working with them to have a good quality of life.’
There was a consensus that timely diagnosis and clear access to appropriate specialties was needed, and the starting point was better awareness in primary and community care services. There was a consistent view that the disclosure of dementia was only the beginning, and that properly resourced models of diagnosis and post-diagnosis pathways were required.
Family transitions
Discussion around family elicited both a personal and professional emotional response from health professionals. Health professionals discussed the practical changes they saw occurring within families, such as the impact on employment, driving, and independence.
‘A word that comes to mind for me would have been “loss”, at a time in one's life when they might not have expected to have lost these things for another 20 years.’
Some health professionals had a direct role in supporting family caregivers, but a lack of dedicated supports caused high levels of stress. This was particularly evident in the context of families with young children.
‘Emotional living grief … a feeling of (carers) were very much struggling with it on their own … feeling not well supported.’
Health professionals felt a sense of professional responsibility towards both clients and their families especially where there were younger dependants. This was in tension with the lack of supports for the families involved.
‘(These cases) are just much more affecting, because it's not a timely illness … Particularly where there (are) … dependent children under the age of 18. Particularly stressful … even for the professionals involved.’
Working with constraints
As need increased, health professionals felt greater responsibility to help families engage with services and access entitlements. However, they felt constrained by a lack of relevant and accessible services. The health professionals experienced dilemmas as they tried to decide how best to enact support. This theme is described in two subthemes: suboptimal choices and regulatory barriers.
Suboptimal choices
Dementia-specific services were described as excellent in many respects, but populated by an older demographic. This generated difficult decisions and even dilemmas for interviewees. When planning care, the choice seemed to be between referring to services that they considered inappropriate, or not to refer and ‘do nothing’. In some cases, neither option felt right. In order to do something for the person and their family, some health professionals referred people to mainstream dementia services.
‘They are a minority group … they're going to be on their own at that particular age, they feel … fearful that they're going to be an awful lot younger than the rest of the people.’
Knowing that the services options were not appropriate, others chose (eg a GP and a community nurses) not to refer to dementia services.
‘Sending somebody that's 45 into our day care centre where the (typical) age may be 75 … it's really not appropriate.’
It was apparent that health professionals were uncomfortable with the lack of relevant services. Interviewees voiced frustration with a service context whereby the person is ‘left to fend for themselves’ (health professional 9). Rather than seeing the person and their family supported, it was ultimately felt that the unique needs of the person was overlooked.
Regulatory barriers
During the course of their work, health professionals encountered frustration with services they felt impeded timely and equitable access. Interviewees felt unsure how to proceed, eg a GP uncertain about referral pathways for assessment, owing to age. The same issue often applied to certain state entitlements and allowances. Knowing that these services could provide benefit triggered a response in health professionals. A particular example was a public health nurse who tried to organise respite care for a family, but was unable to.
‘The barriers in the statutory service … the whole “65 cut off ”, it really does feel like people are being punished for getting a diagnosis before their time. Access to the service should be based on your need rather than your chronological age.’
The age barrier to services was deemed to be a central policy decision that warranted consideration. This would ensure existing services were inclusive and equally accessible.
‘I'm not sure … that it would be high on the list of government priority … the numbers are small, but the needs for the family and individual are different. In the ideal setting there should be specialist services, some of the bureaucracy lifted. Access to services that would traditionally be given to over-65-year-olds.’
New future
Although challenges in the current context were acknowledged, health professionals identified some local, innovative improvements they had seen. They discussed recommendations for YOD-relevant models of care, presented here in two subthemes: ‘community focus’, and ‘working with what we have’.
Community focus
Local community health services were seen as a priority for focus, particularly raising awareness among primary care teams and community groups. Interviewees advocated for education and training of community-based health professionals, in order to improve the challenging time around diagnosis.
‘Somebody to explain the diagnosis to them properly, and what supports are out there for them… Psychologically, definitely you need to be in the right place at the start…. with the proper supports in the community.’
In addition to formal services, interviewees emphasised the importance of developing social models of care. They discussed community social initiatives that might enhance resilience among caregivers, such as peer support and family counselling. Extending beyond this, health professionals called for YOD models of care that supported personhood, responsive to individual choice and preference.
‘You have people who become isolated from friends they have had for long periods of time … (social) clubs or advice sessions or support groups are useful to people because (they aim to address) the sense of social isolation.’
Working with what we have
There was a consensus that the role of a YOD dementia advisor would be of benefit to families, to assist with advice and signposting tailored to the unique needs of YOD. It was felt that current dementia services could be modified in small ways; if services became more flexible, this might increase engagement. Health professionals emphasised an inclusive and needs-led approach to planning future models of dementia care.
‘We need to have a serious think of having specific services for the under-65s that are appropriate, and that they will get benefit from.’
Interviewees called for a more flexible approach to mainstream dementia service arrangements, such as day centre services with weekend or evening options. Related suggestions included the development of a dedicated space or time for people with YOD within current day centre services, and a review of current social, supportive interventions attached to dementia centres.
Discussion
This study drew from the experiences of health professionals to explore models of care for YOD. For those interviewed, the experience of providing care for people with YOD was characterised by uncertainty, frustration and concern for individual and family wellbeing. Health professionals described barriers to timely diagnosis and missed opportunities for support in the early stages. After diagnosis, when trying to enact support and instigate referrals, health professionals were faced with a lack of age-appropriate post-diagnosis models of care, and rigid health service eligibility criteria. This in turn created professional tensions for those who work in the area, who felt frustrated by the constraints they faced.
The literature on YOD is limited compared with typical age dementia. However, some of the themes identified in this paper are reflected in the published literature, particularly in relation to diagnosis pathways, service inequities and the family context (Bakker et al, 2010; Rossor et al, 2010; Bakker et al, 2013; Barca et al, 2014). As far back as the mid-1990s, Keady and Nolan (1994) called for community services, especially nursing, to develop YOD models of care. This call is equally true today, but there remains a lack of relevant service models, and a lack of resources for frontline practitioners to help families live with YOD. Chaston (2011) used the phrase ‘between a rock and a hard place’ to describe the context of YOD service models. This accurately reflects the sentiments of health professionals in the current study, who experience ethical dilemmas as they make professional decisions and provide services that fall short of meeting individual and family needs.
The findings align with recent research calling for additional psychological support for families in YOD (Kobiske and Bekhet, 2018). Health professionals witnessed the effect on families and were mindful of a lack of additional support for partners and children in the community. Nonetheless, health professionals retained a sense of optimism that change was occurring. Interviewees drew attention to the main strategies that might best meet the challenges, starting with timely diagnosis and early intervention. Pragmatic suggestions such as flexible service arrangements, awareness raising initiatives, and social models of care were cited.
National dementia strategy
More than 30 countries now have national dementia policies, and the World Health Organization has declared dementia a ‘public health priority’ (Patterson, 2018). Since this study was conducted, Ireland has launched its National Dementia Strategy (Department of Health, 2014). Compared with strategy documents in the UK, Norway, the Netherlands, France (Carter et al, 2018), the Irish strategy lacked a specific focus on YOD (O'Shea et al, 2017). However, there have been significant positive developments overall; the National Dementia Strategy and the National Dementia Office have acted as a catalyst for numerous priority action areas. It is likely that the new national objectives will benefit people living with YOD. One such example will support primary care clinicians in providing integrated, collaborative dementia care responses. Furthermore, there have been a small number of local innovations specifically targeting YOD.
Moral distress
A further contribution of this research is the health professional perspectives of how they support people living with YOD, in the absence of dedicated services. There was a pervasive sense of concern among interviewees that resource limitations would reduce resilience in people living with YOD and caregivers. In their daily work of referrals, and signposting, they expressed frustration and encountered ethical dilemmas, as they tried to match the unique needs of individual and family with services that were not relevant or readily accessible. These professional constraints experienced by health professionals reflected elements of the literature on moral distress (Austin, 2012; Rathert et al, 2016), which occurs ‘when one knows the right thing to do, but institutional constraints make it nearly impossible to pursue the right course of action’ (Jameton, 1984: 6). Moral distress has also been identified among dementia health professionals previously, as staff try to alleviate client distress and enhance quality of life, but encounter constraints that prevent this (Pijl-Zieber et al, 2018). The current study extends these findings, providing insight into how staff experience such ethical constraints specifically in YOD. Health professionals tried to navigate services that were not appropriate and not accessible, all the while worrying that the situation might erode individual and family coping and resilience. This frustration at YOD service context triggered a personal emotional response in the health professionals. This suggests that health professionals could benefit from psychological supports, especially in community and dementia care settings, where there is a limited tradition of formal peer supervision or debriefing to manage complex ethical issues.
Implications
This study holds implications for future practice, education and research. People living with YOD fall outside of many of the traditional services, and so may not access professionals with the requisite expertise to offer support. In light of the findings, there is a need for practice education and awareness specifically on the area of YOD, for health professionals and particularly for community staff. Staff working in community care settings are ideally placed to support early-stage YOD, to provide timely referrals and post-diagnosis support.
Health professionals who work with people living with YOD should ideally be afforded adequate resources to carry out their role. The model of clinical supervision as espoused in the field of mental health nursing may be an ideal intervention for staff are working in complex or unique settings. An evaluation of the National Dementia Strategy is being completed and should identify any positive changes that have occurred across services.
Limitations
Although it was not a focus of this study, future research should include the person living with YOD, and family caregivers. A potential limitation of this study may be the small sample size, and the use of non-probability sampling. Findings may only transfer to the settings and professional roles that were studied. Because the authors sought to capture individual professional experiences of participants well placed to describe the particular phenomenon under discussion, generalisability was not sought (Smith and Osborn, 2015).
Representation from senior health service management, and also senior clinicians involved with diagnosing was sought but not achieved in the final sample on account of their work and time commitments.
Conclusion
This study aimed to explore health professionals' perceptions of the challenges faced by people living with YOD and their families, and how they enact support for this group. Findings outline some of the key challenges, especially relating to diagnosis, early-stage post-diagnosis support, and service access. The study highlights the unique needs and considerations of YOD, for example, services that are relevant to the person and their time of life, and support options that are responsive to the needs of working families, and children.
A further contribution to the evidence base is the knowledge that health professionals working in YOD experience professional constraints. The National Dementia Strategy was an important policy document and although YOD was not included as a specific priority action plan, it will likely generate positive outcomes, particularly in relation to timely diagnosis and integrated post-diagnostic models of care. Future research on optimal models of YOD services is warranted. Furthermore, it may be of benefit to explore ways to support health professionals working in challenging care contexts.