Co-creation of a family-focused service model living with younger onset dementia

Abstract

Families living with younger onset dementia face unique social challenges, which services do not currently address. There is a gap in understanding the experiences of families affected by younger onset dementia, including the interactions and relationships within the family units, and with existing services and supports. In this qualitative research study, semi-structured interviews were conducted across Australia with seven health and social care providers, five persons living with younger onset dementia, six spouse/carers, and 17 children. A thematic analysis was conducted using a theoretical lens drawn from the combined perspectives of the social model of disability and family systems-illness model. Two central themes emerged in the data: (1) Understanding the social demands on the family – lifecycle challenges living with younger onset dementia from a social and family context; and (2) Interactions of health and social care providers with families living with younger onset dementia – opportunities and challenges in providing support and services geared to a whole family approach. In order to provide families for those living with younger onset dementia with optimal assistance and support, co-creation of a family-focused services model is proposed. Its purpose is to support effective therapeutic interactions and service development among service users, service providers, and stakeholders.

Keywords

family systems-illness model younger onset dementia service design and delivery co-creation social model of disability

Introduction

There is an international consensus that current health and social care services for people living with dementia and their families are predominantly designed for the older population, and do not meet the specific needs of younger people living with dementia (Cations et al., 2017; Greenwood & Smith, 2016; Millenaar et al., 2016). A recent systematic review reported little ‘service development’ for people living with younger onset dementia (YOD) and their families during the last 26 years, even with YOD diagnoses increasing worldwide (Mayrhofer, Mathie, McKeown, Bunn, & Goodman, 2017, p. 7). Although there are many types of dementia with differing presentations, particularly in younger people, Alzheimer’s disease (AD) is considered the most prevalent (Carter, Oyebode, & Koopmans, 2017; Rossor, Fox, Schott, & Warren, 2010). Dementia is considered an unexpected illness before the age of 65 years with social, emotional, psychological, and financial consequences for the individual and family members associated with unique social challenges compared to late onset dementia (LOD) (Roach, Keady, Bee, & Williams, 2014; Withall, Draper, Seeher, & Brodaty, 2014). To date, few research studies have considered the experiences of the whole family living with YOD and their interactions with existing services and support (Beattie, Daker-White, Gilliard, & Means, 2002; Gelman & Greer, 2011; Luscombe, Brodaty, & Freeth, 1998; Roach, 2010; Roach, Drummond, & Keady, 2016; Svanberg, Spector, & Stott, 2011). These studies corroborate that services should become more socially aware to ensure they target the complex needs of the individuals and family members living with YOD.

Understanding the way services are designed and delivered is a fundamental prerequisite in meeting the specific needs of families living with YOD. Shaping services and support based on the experiences of families living with YOD is crucial in informing the development of such services (Barca, Thorsen, Engedal, Haugan, & Johannessen, 2014; Bate & Robert, 2006; Evans-Roberts, Weatherhead, & Vaughan, 2013). Research on ‘families’ tends to focus on the perspectives of and impacts on spouses with dementia and spouse caregivers, with little consideration of other family members (La Fontaine & Oyebode, 2014; Roach et al., 2014). This research approach does not depict whole families’ experiences, internal dynamics, and relationships. The few studies exploring the impact on whole families living with YOD indicate that every member and their relationships with one another are impacted by complex challenges (Millenaar et al., 2016; Roach et al., 2014, 2016; Svanberg et al., 2011). Families’ experiences living with YOD are unique in many ways, particularly related to the developmental stage of the family, change in family members’ roles and responsibilities, and financial commitments (Rolland, 1999). Typically, these needs are not reflected in the design of services or policy development, suggesting a lack of focus on ‘family-centred paradigms’ in dementia research (Roach et al., 2014, p. 1416).

Currently, the way services are designed and delivered do not reflect whole family needs, nor consider the person with YOD as an ‘integral member’ of the family (Barca et al., 2014; La Fontaine & Oyebode, 2014; Roach et al., 2014, p. 1399). Even with an increase in the diagnosis of YOD, there remain few age-appropriate formal services available, particularly when the variety in forms of dementia and presentation of symptoms in YOD is considered (Millenaar et al., 2016). This results in an increasing reliance on informal family support (Sansoni et al., 2016); often over a longer time frame than with LOD (Carter et al., 2017). Moreover, families often find it difficult to relinquish care of the younger person with dementia due to this lack of fit between need and service provision, isolating families further from other support opportunities (Cations et al., 2017; Millenaar et al., 2016). Subsequently, informal caregivers across a range of ages can experience adverse effects on their emotional and physical well-being, because they are inadequately supported in their role (Hutchinson, Roberts, Kurrle, & Daly, 2016; Millenaar et al., 2016; van Vliet, de Vugt, Bakker, Koopmans, & Verhey, 2010). Receiving necessary support can assist caregivers’ capacity to manage their daily roles and commitments, particularly those linked to their phase of life such as employment, financial responsibilities, education, family relationships, and dependents (Brodaty & Donkin, 2009; Hutchinson, Roberts, Kurrle, et al., 2016; Millenaar et al., 2017; van Vliet et al., 2010).

Many health and social care providers in dementia care agree that identifying and engaging with family members of all ages and developmental stages is essential in YOD. However, this does not routinely occur even though family members are known to be influential on experiences and management of dementia (Gelman & Greer, 2011; La Fontaine & Oyebode, 2014; Roach et al., 2014). There is a dynamic interplay between complex family relationships, specific roles, and approaches to managing challenges which have implications for practice (La Fontaine & Oyebode, 2014; Roach et al., 2014). Frequently the concept of families has been based on the traditional view of family structure, a basic ‘social unit’ including parents and children (Murray & Barnes, 2010, p. 535). This view often ignores the functional relationships and social diversity of families including ‘ethnicity, sexual orientation, and divorce' (Clarke & Hughes, 2010; La Fontaine & Oyebode, 2014, p. 1268; Murray & Barnes, 2010).

Formal care providers face many challenges engaging with and supporting both the younger person with dementia and their families. Providers report they are working with a lack of resources and that there are few age appropriate services that cater for the complex and diverse needs of families living with YOD (Carter et al., 2017; Mayrhofer et al., 2017; Pratt, Clare, & Kirchner, 2006). It is also reported that working within service models that are focused on LOD is frustrating and contributes to families feeling ‘marginalized within existing service provisions’ (Beattie et al., 2002, p. 205), stigmatised and discriminated against (Millenaar et al., 2016). Also, the failure to receive a timely diagnosis can frequently be a barrier to accessing appropriate information, interventions and services (Pratt et al., 2006).

An opportunity to address these gaps in the literature arose on a larger doctoral research project exploring the impact of YOD on the individual and families. This research aims to develop a theoretically informed appreciation of the relationships and interactions of those living with YOD within current dementia care provision in Australia, with the view of informing the co-creation of appropriate services.

Theoretical framework

Two theoretical approaches were considered in this study; the social model of disability (Oliver, 1983, 2009) and the family systems-illness model (Rolland, 1990). The development of a socially focused theoretical framework requires a collaborative research approach with all family members and service providers that share the necessary experience to co-create family-centred services (Clare & Cox, 2003; Mayrhofer et al., 2017). Such an approach allows family members to regain an element of control in the way they are managed, providing a number of benefits for those living with impairments, and those family members living alongside them (Clare & Cox, 2003; Mayrhofer et al., 2017).

For families of those living with YOD, the social model of disability (Barnes & Mercer, 2011; Oliver, 2009, 2013) can add valuable research insights into the significant impacts of societal perceptions, whilst also focusing on interactions and relationships of both family members and health and social care providers, including the consequences (Chappell, Goodley, & Lawthom, 2001; Hutchinson, Roberts, Daly, Bulsara, & Kurrle, 2016, Hutchinson, Roberts, Kurrle, et al., 2016). With a focus on the experience of the service user, a social model perspective illustrated the way in which interventions are currently designed around health professional roles and patient diagnosis, and have unintended impact on families living with YOD (Gilliard, Means, Beattie, & Daker-White, 2005; Hutchinson, Roberts, Daly, et al., 2016). The social model has value in informing and guiding service design and delivery because it is not diagnosis-specific but socially inclusive, and supports shared involvement of people living with complex needs and their families (Clare & Cox, 2003). Utilizing the social model can help reframe dementia to offer ‘alternatives to current responses and approaches’ (Mental Health Foundation, 2015, p. 29).

From a family context, Rolland's family systems-illness model (Rolland, 1987, 1990, 1999) proposes a structure which advances understanding and knowledge of families’ ‘changed life-cycle demands’ (Rolland, 1990, p. 229), in order to help anticipate interventions and facilitate problem-solving. This model has been applied to families living with YOD (Roach et al., 2014) and other complex conditions (Rolland, 2005). This model provides a guide to managing challenges across the trajectory of family experiences. Understanding the impact, demands, and uncertainties of parental illness or disability on the intergenerational family unit are essential in appreciating need. Relating understanding to individual and family life cycles; openness of communication between family members; and family beliefs is beneficial. Also, knowing family backgrounds, family history, and family functioning is vital to appreciate the whole of the family circumstances (Roach et al., 2014).

The family system-illness model considers three phases: (a) the illness progression and psychosocial impact, (b) phase of illness and interface with individual and family developmental stage , and (c) elements and variables of family functioning (Roach et al., 2014; Rolland, 1990, 1999, 2005).

The onset of YOD is usually gradual, with cognitive changes occurring over a lengthy timeframe prior to a formal diagnosis. The time of the formal diagnosis can be described as the initial crisis, a ‘highly emotional and vulnerable period’ (Rolland, 1999, p. 231), with families sensing their loss of control, ultimately threatening usual family life. Overall a distressing time for the individual and family, especially around age of onset of YOD, work and family related disruptions (van Vliet et al., 2011). The views and attitudes of healthcare providers at this time can impact the future outlook on life, their access to necessary information, and vital services (Barca et al., 2014; Pratt et al., 2006). The progressive course of dementia creates a great deal of uncertainty and challenges at a socially significant time and developmental stage, generating tensions and disruptions within the whole of the family, and their lived community (Roach et al., 2014). The unpredictable and often lengthy chronic phase can place psychological and physical strain on families as they continually adapt to changing lifestyle, roles and responsibilities, and they are frequently inadequately supported (Cations et al., 2017). The outcome of death causes family members to experience a range of emotions over the trajectory, dealing with ‘anticipatory loss’ of the person living with YOD (Rolland, 1999, p. 229). Families may become ‘more present focused rather than future focused’ (Rolland, 1999, p. 233), particularly during this terminal phase. Understanding the ‘interface of the illness, the individuals and the family life cycle’ (Roach et al., 2014, p. 1401) alongside the historical and biographical functioning of a family unit, as well as societal influences around the perception of disability, would be valuable for health and social care providers. In considering the interplay between the two models, the social model of disability (Oliver, 1983, 2009) has recently been used to explore the ‘social position’ and rights, living with dementia (Shakespeare, Zeilig, & Mittler, 2017; Thomas & Milligan, 2018, p. 117). The social model, therefore, can help explain families’ experiences over the three phases living with YOD, by relating their experiences to the social landscape they live in (Barnes & Mercer, 2011; Gilliard et al., 2005; Hutchinson, Roberts, Kurrle, et al., 2016; Oliver, 2009). Therefore, considering data from the perspective of the social model of disability and family systems-illness model may lead to more nuanced theory building, and also inform current practice and service provision collaboratively at the level of both service user and their families (La Fontaine & Oyebode, 2014; Mayrhofer et al., 2017; Nolan, Ryan, Enderby, & Reid, 2002).

In this context, we aimed to develop a theoretically informed appreciation of the relationships and interactions of those living with YOD within current dementia care provision in Australia to determine the critical factors that need to be addressed in co-creating appropriate family-focused service.

Methods

This qualitative study was undertaken in New South Wales (NSW), Australia, based on active interactions between researcher and participants in families living with YOD, and with health and social care professionals (Fontana & Frey, 2005). Ethics approval was obtained from the University of Sydney.

Participants

Participants were recruited through advertising with non-governmental organisations, Alzheimer's NSW and Young Carers NSW, and via snowball sampling. A total of 26 family members participated, including people living with YOD (5), spouse caregivers (6), and children and young people in families (17), who were aware of their parents’ diagnoses. The age of the children and young people ranged from 9 to 33 years, with the older participants in this group describing retrospective experiences, adding the benefit of maturity (see Table 1). Seven health and social care providers who had experience supporting families living with YOD, including children, were recruited. These included one geriatrician, nurse, occupational therapist, psychologist, and counsellor, and two YOD key workers (see Table 2).

Table 1.

Demographic characteristics of participants (YOD = 5, spouse/carer = 6, children/young people = 17).

Family	Diagnosis	Relationship to person with dementia	Age at interview	Age of participant at family member diagnosis	Other immediate family members not interviewed	Person with YOD marital status & resides with
1	Alcohol related dementia	Self	49	46	3 children	Separated blended family, lives with ex-wife and 3 children
		Ex-wife	46	44
		Daughter	10	7
2	Alzheimer’s disease	Daughter	19	19	Mother (AD), I child	Divorced, lives with daughter (19)
2	Alzheimer’s disease	Daughter	22	22		Divorced, lives with daughter (19)
3	Alzheimer’s disease	Son	22	14	Father (AD), 3 children	Married, lives in nursing home
3	Alzheimer’s disease	Wife	68	58		Married, lives in nursing home
4	Alzheimer’s diseases	Daughter	30	24	Mother (AD), 1 child	Divorced, lives with daughter (30)
4	Alzheimer’s diseases	Daughter	33	28		Divorced, lives with daughter (30)
5	Alzheimer’s disease & Lewy body dementia	Self	43	42	Husband, 1 child	Married, lives with husband and 4 children
		Daughter	9	8
		Son	10	9
		Daughter	15	14
6	Frontotemporal dementia	Wife	48	44	Father (FTD), 1 child	Married, lives in nursing home
6	Frontotemporal dementia	Son	13	9		Married, lives in nursing home
7	Alzheimer’s disease	Wife	57	49	Father (AD)	Deceased
7	Alzheimer’s disease	Daughter	28	21		Deceased
8	Frontotemporal dementia	Daughter	24	23	Mother, father (FTD), 2 children	Married, lives with wife
9	Semantic dementia	Daughter	25	18	Mother, father (SD), 2 children	Married, lives with wife and 2 children
10	Alzheimer’s disease	Daughter	26	16	Mother(AD)	Divorced, lives in nursing home
11	Huntingdon’s disease	Daughter	27	Teenager	Mother (HD) in NH	Single, lives in nursing home
12	Posterior cortical atrophy & Lewy body dementia	Self	51	49	3 children	Alone
13	Alzheimer’s disease	Wife	53	51	Father (AD), 2 children	Married, lives with wife
14	Frontotemporal dementia	Self	58	52	3 children	Married, lives with husband
14	Frontotemporal dementia	Husband	57	51		Married, lives with husband
15	Alzheimer’s disease	Daughter	24	20	Mother (AD), stepfather, 1 child	Remarried, lives with husband
16	Alzheimer’s disease	Self	52	57	Husband, 2 children	Married, lives with husband
16	Alzheimer’s disease	Daughter	21	16		Married, lives with husband

AD: Alzheimer’s disease; FTD: frontotemporal dementia; SD: semantic dementia; HD: Huntingdon’s disease; NH: nursing home.

Table 2.

Health and social care provider interview information (n = 7).

Roles	Sex	Sector	Years working with dementia	Years working with families living with YOD	Youngest child supported (years)	Most challenging age to support (years)	Interview (min)
Counsellor/ coordinator of early intervention	F	NGO	9	8	15	20–24	72
Geriatrician	M	Health	8	5	8/9	5–9	78
Case manager/YOD key worker	F	NGO	19	9	4	10–19	67
YOD key worker	F	NGO	12	3	7	15–19	72
Registered nurse	F	Private	>25	11	9	10–14	65
Occupational therapist	F	NGO	1	1	12	10–14	74
Psychologist	F	Health	7	7	7	15–19	72

Data collection

Interested participants had initially contacted the first author (KH), and those eligible for inclusion received initial study information via email, with a follow-up phone call once consent to proceed was received. Basic demographic information was also collected before the interview, which prompted discussion of family background and helped judge how best to conduct the interview (Irwin & Johnson, 2005; Murphy, Jordan, Hunter, Cooney, & Casey, 2015). KH discussed the interview process prior to obtaining written consent to alleviate any fears and concerns, ensuring that confidentiality and the rights of the participants were considered at all times (Murphy et al., 2015). To manage any risk to the participants’ emotional wellbeing, KH explained her duty of care as a health professional and a researcher. The participant had to agree that if they became extremely distressed or concerned during the interview, a referral would be made to an appropriate clinician for further support. No referrals were required after the interviews. Additional written consent to safeguard the interests of children under 16 years and adults living with YOD was obtained from an adult (parent or guardian). Important ethical considerations ensured probing, open ended questions were conducted in a ‘sensitive and considerate manner’ (Roach et al., 2016, p. 28).

With the younger participants, coloured pens and paper were provided and they were given a choice to draw a picture of their family, which helped to generate discussions and improved comfort levels to participate (Irwin & Johnson, 2005). Also, KH carefully considered questioning techniques with younger participants and those living with dementia to build trust and rapport. They were given the option to have someone remain with them throughout the interview, which some accepted. After the interview, a resource list of support and services was provided to participants.

Data were predominantly collected via individual semi-structured interviews with family members, but on four occasions they were conducted in pairs. Observations and field notes at interviews were also considered as part of the data collected. Interviews were audio-recorded by KH in participants’ preferred venue, by Skype or by telephone due to distance. Data were gathered through the sharing of stories, aided by the use of probing questions associated with the lived experiences of family members impacted by YOD over the trajectory of YOD, and health and social care provider's experiences supporting families over this time. After every interview, each participant was thanked and advised of the importance of their contribution. The sensitivity of the subject was acknowledged, and the interviewer debriefed with each participant afterwards to establish their emotional well-being.

Data analysis

Interviews were transcribed verbatim by a professional agency, anonymised by first author (KH), and participants were provided with a pseudonym. A framework analysis (Ritchie, Lewis, Nicholls, & Ormston, 2013) was used, being a flexible analytical tool that can assist in generating inductive themes that emerge from qualitative data (Gale, Heath, Cameron, Rashid, & Redwood, 2013). Initially there was a period of familiarisation with the data, and each author independently analysed three transcriptions. Significant key issues and themes were identified, highlighting a relationship between family members’ interactions and their social world. This focused coding of transcriptions on the socio-cultural underpinnings of families, health and social care provider's experiences, interactions, and relationships that shaped family functioning, service and support provision. The authors then collaboratively discussed and negotiated to cluster codes and develop a theoretical framework that resonated with our preliminary themes and sub-themes. Refinements were made to the theoretical framework that captured this relationship until all the authors reached an agreement, and then KH applied the framework to the whole dataset. Inductively the combined paradigm presented by the social model of disability and the family systems-illness model created a theoretical framework that resonated with our central and sub-themes.

Results

Two major themes emerged from the data. The first theme: Understanding the social demands on the family – described lifecycle challenges and pressures on families living with YOD, within a social context. The second theme: Interactions of health and social care providers with families living with younger onset dementia – depicted opportunities and barriers experienced in service provision including advocating for change in the health and social care system. The three sub-themes underpinning each central theme will be explored.

Understanding the social demands on the family

When a family member receives a diagnosis of YOD, there are a number of impacts, or social demands, on relationships and interactions between immediate family members, extended family and friends, and health and social care providers. These social demands are illustrated by the following three sub-themes: Social challenges for people living in the family with young onset dementia, Coping with the unexpected as a child of a parent with younger onset dementia, and Balancing the demands between dual roles as a parent and caregiver. Each of these three sub-themes will be described in detail below.

Social challenges for people living with young onset dementia

People living with YOD provided rich descriptions of the complex social challenges they experienced within a family context. Brian diagnosed with alcohol-related dementia at the age of 46, describes himself as a ‘loner' and a consumer of large quantities of alcohol, which contributed to his impairment. Although separated from his wife Jane, they continued to live together. Jane became his main carer post-diagnosis and described their life together as stressful, particularly with three children aged between 10 and 21 years. Challenges of family life were compounded with Brian's forced termination of employment and being unable to contribute financially to support his family. He ‘felt like the world could end,' and was not coping well with the loss of his sense of purpose associated with his unemployment. Throughout this early phase post-diagnosis, while he was ‘fighting for the pension,' he began increasing his alcohol intake. This had been his consistent response to difficult situations in the past, but in this instance he went onto attempt suicide. It was only after this crisis did Brian and Jane receive any services, but no formal support or information was offered to their children.

Another participant, Sylvia, married with four children aged between 9 and 18 years, was diagnosed with YOD at 42 years. She recalls feeling ‘the floor was giving way beneath’ when the GP mentioned the possibility of dementia. Sylvia was about to start a new career and described dementia as the ‘biggest hit in the guts.’ She then felt in ‘limbo’ anticipating the formal diagnosis, which took some time to confirm. Some family members refused to accept her diagnosis and her other social networks diminished, stating ‘fantastic friends … they’ve all just disappeared.’ Sylvia believed these ‘painful' reactions were based on dementia and she was concerned about the impact of reduced socialising may have on her children, ‘as we don’t see each other my kids miss out.' Exposure to negative attitudes and responses added to her sense of loss and exclusion, leaving Sylvia apprehensive and vulnerable informing her children about dementia. Believing they had already lost so much, Sylvia wanted to protect her children and so delayed telling them about her diagnosis, not wanting to add to her children's ‘emotional baggage.’ She was unsure how to approach the conversation, as was evident during the interview, ‘What do you say? How do you tell them (children)?’

Sylvia also felt disconnected from her family and that communication within the family was limited. This altered the family dynamics, with each family member taking on new roles and responsibilities without her input. Sylvia lamented the loss of meaningful activities and a feeling of connection related to her historical place in the family.

I tend to stay at home, my license has been suspended. My family is busy and I don’t have friends that come around.

Sylvia had insight into the ways in which her cognitive impairment impacted her, but also the way attitudes of others was disabling. For example, she tried attending school meetings to organise more support for her children. She experienced disbelief when she disclosed her diagnosis, causing her to lose confidence and want to disengage from any social situations. Fortunately, the involvement of a health care professional that she trusted supported her to re-engage with the schools resulting in a more favourable outcome. A significant person, either within or outside the family, to openly communicate and connect with, can help counter vulnerability or manage uncertainties. An unfavourable alternative for those living with YOD, is to keep things hidden based on their experiences of misunderstanding, and discriminatory behaviours. Support services need to anticipate and better manage families living with YOD as ‘blocked communication can fuel anxieties' (Rolland, 1999, p. 257).

Coping with the unexpected as a child of a parent with YOD

The experiences of children and young people in families living with YOD demonstrate complex challenges frequently related to their age, which are often not understood or recognised. Evie was 11 years old when she noticed some behavioural changes in her mother, Freda. Evie’s story demonstrates some socially significant experiences over the progression of her mother’s dementia, throughout her own challenging developmental period. As an only child with divorced parents, she divided her time living with both parents. However, she did not have a good relationship with her father, therefore, was reluctant to burden him, leaving her frequently unsupported in caring for her mother.

I don't know what it was, but I just had people around me that just didn't care or didn't want to get involved. I think I'd just been a bit unlucky.

Six years later, Freda was formally diagnosed with YOD when Evie was 17. Evie’s initial experience of formal services at this time was in a group format on ‘how to take care of someone with Alzheimer's’. She learned useful strategies but recalled standing out wearing her ‘school uniform.’ A sense of belonging and inclusion is important for young people in accessing support and services. As Evie recalls, ‘I'm glad I didn't (attend YOD support groups) because that would have made me feel more of an outcast.’ However, a few years later, Evie attended a specific group for young people with a parent with dementia. She believed being connected to people of a similar age was extremely beneficial.

Evie received dementia-specific counselling, but this ‘sometimes could give me more of a feeling of hopelessness than before I came in,’ which she felt was focused around a medical model that was fixated on the tragedy of her age and situation. At 18, Evie’s situation deteriorated; a recognised significant developmental stage in a young adult's lifecycle. Increasing demands on her caring role and school work resulted in a ‘nervous breakdown.’ She sought help from her GP, who advised her to leave home, but she had nowhere to go and no other support options were offered by her father or formal services. Throughout this difficult time, it was arranged by a family friend for her to obtain the enduring power of attorney on her 18th birthday, something not often contemplated as applicable to this age group. Consideration of the social diversity and background of families is crucial. She describes the enormity of taking on this role for her mother at her stage in life; ‘it was like everything I was doing, it was setting up for something horrible.’

After many years caring for her mother, Evie moved to another city to live her own life. Not long after her departure, Freda was admitted to a nursing home. Evie recalls feeling ‘worried about, the fact that I had the responsibility of choosing when I had to put her in a home, and when do you know how horrible is horrible?’ This level of responsibility in navigating complex processes is daunting for most adults but additionally so for young people, being unexpected at their stage of life.

John was the youngest sibling out of four and 14 years old when his father, Trevor, was diagnosed with YOD. He described feeling lost and alone in the family while his mother Flora was ‘kind of too deep in her pain to kind of be aware of it (his distress),’ so he hid his emotions for years, demonstrating a lack of openness, not wanting to add to his family’s distress. He reached a crisis in his early 20s when he self-harmed. This crisis was related to his feelings of guilt with the permanent placement of his father in residential care. Certain events or transitions can spark an emotional or family crisis, like the situation John described. Young people portray different coping mechanisms at times of crisis, which include ‘escaping’ the family home like Evie, engaging in risk-taking behaviours such as drug and alcohol abuse to ‘make myself happy’, or self-harm, revealed by John. This could all be anticipated and managed by service providers with open communication; improved connectivity with families (Hutchinson, Roberts, Daly, et al., 2016); knowledge and understanding of potential responses to life cycle challenges; knowing potential impact on the family through transition phases of dementia; and more access to age-appropriate family-focused services.

Balancing the demands between dual roles as a parent and caregiver

There are many challenges experienced being both a caregiver and a parent. This results in a deviation in their life trajectory as a consequence of changed family circumstances, associated with partner's diagnosis of YOD. Ann and her husband, David, were living with their two young sons (one in the first year of school and the other in year 3), when he was diagnosed with YOD at 50 years. Although challenged with her dual role, Ann believed throughout the upheaval of their family's day to day life:

… the thing that's really kept me going has been the boys. I think that's been my motivation to keep on going. Because it would have been really easy just to really go into victim mode here … knowing though that if I fell over then it was going to fall over for everybody.

Acknowledging the family was now dependent on her, Ann took control of her own emotional wellbeing in the early stages by self-sourcing and connecting with a psychologist. Taking care of everyone’s needs and juggling the many appointments required regular travel, living in a rural community. She gave up work for a couple of years, something her employer sympathetically supported. Although facing many challenges, Ann lived in a supportive, small community and believed ‘the medical support has just been amazing. I couldn't have asked for anything better.’ Access to a psychologist and psychiatrist directly to obtain support at times of need, improved her confidence and ability to deal with complex situations. Being bulked-billed – not being charged for medical services – also alleviated some family financial concerns being unable to work. Positive interactions and attitudes towards her situation helped to reduce her vulnerability and offered her some control over the situation.

However, Flora, John’s mother, described her life as a parent and caregiver as ‘hell on a stick.’ This metaphor could convey feeling trapped in an adverse situation, ‘hell,' all directed onto Flora the ‘stick.' After Trevor's diagnosis of YOD, John at 14 years developed a mental illness, which required ongoing treatment. Flora also shared her own mother's care with her sister, prioritising everyone's care needs over her own. She described how her

[Son] was my priority because he was the child – if I needed to make a choice between anyone my duty as a parent I felt he had to be the number one. But I was coping with the other two as well.

So the next few years became fairly difficult. I certainly was very, very, very depressed that first year. Did consider suicide, but I did finally get some treatment. I've realised that it was just so much pressure that I couldn't handle it.

Flora experienced a health crisis some years later, being admitted to a hospital, which triggered input from formal services. This crisis enforced her to reconsider her situation. She arranged permanent residential care for Trevor so she could better manage her health and parental responsibilities.

Interactions of health and social care providers with families living with YOD

This theme explores the perspectives of health and service providers to understand better the social factors impacting their role in supporting families living with YOD. Three sub-themes were used to further explain: Barriers to providing services and support to families, Working collaboratively can make a positive difference to family life, and health care and service provider's contribution to changing practice.

Barriers to the provision of services and support to families

Health and social care providers report their primary role and responsibilities are with the person with dementia and primary caregiver. They confirm a lack of engagement with the children or young people in families, reporting ‘we mostly go through the families and they filter out the information.’ A significant challenge is ensuring that this information is communicated to younger family members whilst recognising they are often isolated and not connected to any support. However, ‘trying to figure out how to give parents information to connect them (young people) to services so that they can provide them with information’ is an ongoing challenge. Parents were sometimes described as ‘gatekeepers' limiting or denying service providers engagement with younger family members. This was believed by some as a combination of parents' protection of their offspring, not being aware of their children's need for support and information, or their perception of support being a parental or family responsibility. However, a lack of open communication could in effect be more destructive, contributing to isolation, misinformation, and exclusion of younger members from the family (Hutchinson, Roberts, Daly, et al., 2016).

Health and social care providers face many challenges that oppose their values and beliefs in providing age-appropriate support to families living with YOD. They describe ‘frustration’ with restrictive guidelines in their roles; difficulty navigating and sourcing age-appropriate services; limited opportunities to be flexible and responsive to need; challenged by the aftermath of changing government and funding structures; limited time and opportunities to develop interagency collaboration; and dealing with the general lack of understanding and negative attitudes towards YOD. Teresa, a healthcare professional, discovered ‘that with a lot of services I had to educate them before they knew enough about dementia to help support’ the family as a whole and understand their needs.

Limited opportunities for upskilling to work with younger families and lack of family-minded policy and procedures can result in ethical challenges and dilemmas, being constrained in the provision of responsive services. One service provider described guidelines as ‘prescriptive’ and another as ‘very focused on the person with dementia, not their children.’ Without a social change, the way services are designed and delivered will continue to challenge professional values and responsibilities supporting whole families. This could be a possible factor in moral distress (Pauly et al. 2012) and high turnover of staff, an ongoing problem, which affects the development of strong connections and relationships with families.

Kate, a service provider working over an entire state in Australia, is expected to be responsive to and engage with families diagnosed with YOD. She reported ‘burnout’ in many colleagues, another factor causing high staff turnover. Kate is not alone, feeling:

Helpless. I feel like it's hopeless and its difficult for me to turn up to an assessment with underlying hopelessness … because I intend to provide that person with a sense of, you have dementia but you can live well with dementia and this is how, but I – there's a big gap. There's help but we can't access too much and that's what I struggle with.

Jan, another service provider, highlighted in many cases there is ‘no opportunity to get funding' to access necessary services. They felt disempowered in meeting their professional responsibilities to the family, which disables the family from moving on with their life.

Working collaboratively can make a positive difference in family life

Providing support to the whole family is deemed a necessary action by most, but can be challenging with limited resources and unsupported by policy and procedures. Ideally, developing strong links with many sectors and agencies is crucial but currently not routine practice. This justifies the importance of understanding the family’s social position as well as their historical and biographical functioning as demonstrated by Fran.

Fran, a service provider, supported a single mother, Sophie, living with YOD. Sophie struggled to get her teenage child, Annie, to school on time, which caused her to be bullied. Fran attended a school meeting as Sophie’s advocate, obtaining prior consent from Sophie to talk about the home situation. Fran describes the school as being initially ‘process based,’ until the whole family situation was explained and some understanding of the complexity of the situation was acknowledged. The school instigated ‘band-aid solutions' for Annie, but Sophie remained unsupported in her parenting role, which was the main issue. Annie developed depression and anxiety, feeling alone, having ‘no one to talk to.’

Fran obtained relevant family background information. Sophie, diagnosed with YOD, lived alone with Annie, 13-years-old. Annie's father lived far away from the area, and her other siblings had left home and reportedly ‘abused alcohol and drugs … being physically aggressive with each other and very well known to the police.’ This information was helpful in Fran's role as she was aware that the social interactions with other family members would not be a useful resource. Sophie's parental judgments were impacted by the dementia, and Annie urgently needed access to age-appropriate supports. After many frustrations, a favourable outcome for the family was eventually achieved. Fran did highlight some ethical considerations, ‘recognising I‘m not trained in that field, and I'm not trained to counsel and nor am I employed to do that.’

Teresa worked with complex family situations living with dementia, and routinely gained knowledge about the family. In her role she identified specific family needs before implementing ‘multi-system case reviews,’ bringing relevant agencies and family members together to develop services and support that addressed the specific family situation. It was time-consuming, with many obstacles and processes in the way particularly related to the way services were funded. But ‘at the end we'd always walk away with a really good package that would meet the needs of the people’ in the family. This service model discontinued due to a lack of funding.

Healthcare and social care provider’s contributing to changing practice

Raising awareness and understanding of the challenges families living with YOD experience is necessary for advocating for social change. Teresa, a health care professional, has adopted this as

… part of my role now is to build the capacity within our staff to actually understand and see the importance of younger people within the families.

I think we need to be doing assessment around what all the family members need, not clinical assessments but what are the needs of each family member.

Stan, a healthcare professional specialising in aged care, stated medical training focuses on ‘diagnosis, managing and treating patients' but identifies inadequacies in this approach concerning families with YOD. Even with his limited experience with children and young people he appreciated ‘if I don’t know what these young people are going through, I’m not even going to connect with them.’ He demonstrates a moral obligation through self-learning to invest in the whole family over the long term, and his responsibility ‘to prepare this person and their family members.’

He acknowledged the way dementia is framed can have long-term repercussions on the whole family, advocating for the inclusion of all family members in sharing information and connecting them with appropriate support. But he admitted this is not the case with other work colleagues, as many take the attitude ‘dementia means there is nothing can be done.’ The adoption of these negative views often results in a failure to provide vital information and support opportunities, which can have detrimental effects on family members as already highlighted. A significant shift in thinking and approaches taken by health and social care providers is essential.

Stella, a healthcare professional, suggests a ‘buddy system’ across sectors, which could improve confidence with children and young people through

learning from another youth-focused clinician on how he or she is doing that, and they would be learning from me with dementia-specific stuff.

Attitudes and communication styles adopted by health and social care providers can have a lasting effect on whole families’ approach to living with YOD and future interactions. Those with a willingness to learn, explore own strengths, weaknesses, beliefs, and values to engage and understand values, hopes, and fears of those in the midst of challenging situations exhibited sensitivity, positive, and non-judgmental attitudes, which improved job satisfaction and relationships with families (Rushton, 2016).

Discussion

Our findings have illuminated a number of the relationships and interactions of those living with YOD within current dementia care provision in Australia. The ability of the family to surmount the daily challenges of living with dementia can be dependent on the quality of relationships, openness of communication, emotional connectedness, and the level of understanding of dementia (La Fontaine & Oyebode, 2014; Patterson, Clarke, Wolverston, & Moniz-Cook, 2017). The research concurs that poorer family relationships and stress were linked to less positive adaptation to dementia, ineffective communication, and lack of connectedness for all family members, which has been demonstrated here in this paper (Keady & Nolan, 2003; La Fontaine & Oyebode, 2014). Unfavourable interactions and lack of informal and formal support can compound these experiences. Moving the focus of service provision from the individual with YOD to the whole family is supported in this research. Advocating for the entire family’s involvement in early communications about dementia could significantly impact the family's future functioning and interactions. It is vital to obtain early knowledge about families’ background, history, and beliefs to help gauge service interventions, and reduce the risk of a family crisis is consistent with other research (McDonald et al., 2016; Rolland, 2005). Knowing the developmental stage of families is essential, with individuals across different ages juggling roles and responding to family demands. This can affect their ability to look after their own and other's needs. However, the more vulnerable and less confident members of the family, the parent living with YOD, and younger family members often have a less powerful voice in society and within the family. Opportunities to communicate and connect with significant individuals safely are crucial (Hutchinson, Roberts, Daly, et al., 2016; Patterson et al., 2017).

This research highlights that health and social care providers’ roles are currently organised around the biomedical model, with a focus on the person living with YOD and primary caregiver. The life cycle stage of the family and individual members with their specific needs are often overlooked in the way services are designed and delivered (Cations et al., 2017; Millenaar et al., 2016). The lack of shared responsibility, cross sectorially, to support these families can disable them in managing life challenges, and affects healthcare and social care providers’ ability to maintain core values and commitments to the families. This can affect relationships with the families, being unable to fulfill professional obligations with associated narratives of frustration, hopelessness, and challenge. Accessing resources and services to address family-specific issues and concerns is currently time-consuming due to the limited cross-sectorial collaboration opportunities available, but can produce favourable outcomes for everyone as demonstrated in this research. The research verifies that health and social care providers’ ability to adapt and respond to challenges, explore and manage own perceptions of dementia are favourable. Developing the capacity to establish a positive relationship with the family can bring about the change needed for greater ‘understanding, acceptance and adjustment,' benefitting all (Fallowfield & Jenkins, 2004, p. 317).

Implications

How then can these critical factors in the lived experience of families living with YOD be synthesized into a framework for the co-creation of appropriate family-focused services. In considering first the theoretical framing, we propose the combination of the family systems-illness model (Rolland, 1987, 1990) and the social model of disability (Barnes & Mercer, 2011; Oliver, 2009) as an innovation in providing explanations as to why the family needs should be at the centre of service design and planning process (Cations et al., 2017; Mayrhofer et al., 2017). The co-creation of a family-focused service model underpinned by the theory of these two models can inform policy change and guide clinical practice, appreciating the dynamic interplay between all family members living with YOD, health and social care providers, and the broader community (Mayrhofer et al., 2017).

This co-creation of family services model shifts the focus of interventions from the biomedical model to the social model, fostering social engagement and a change in clinical practice, and the promise of better service user outcomes. In the initial phase of YOD, a particular time of family turmoil, early engagement with the whole family by a community liaison person or similar role, can help identify a family’s social position, functional background and beliefs. The way in which these early encounters are framed can set the precedence for future interactions. Fostering the whole family’s engagement in joint discussions conveys the importance of inclusion in the process of change and adapting expectations, which can ultimately help direct focus away from experiences of loss (Millenaar et al., 2016; Rolland, 1999). Facilitating crucial family discussions helps to reduce the risk of crisis, alleviate fears and isolation, and improves resilience to manage life's challenges. Ideally, those in this community liaison role would ‘work, communicate and learn together' (Robinson & Cottrell, 2005, p. 558) with GPs, medical specialists, nursing and allied health professionals across sectors to identify these families as early as possible and engage promptly with the whole family. In this liaison role it would be possible to develop strong links cross sectorially with health and social care providers, and connect with the whole family, which could reduce family vulnerabilities and improve confidence to manage challenging situations. Collaborative options for improved communication, routine information transfer, and stronger links and learning across agencies and sectors could be facilitated through ‘electronic databases and single points of entry to dementia services' (Robinson et al., 2009, p. 141). The family's access to age-appropriate services and support would then be simplified and more responsive, with more established cross-sectorial links.

Considering the whole family approach to service provision as the norm is crucial over the progression of dementia. Being mindful of family vulnerability and emotional manifestations that can vary with stages of dementia, socially significant and developmental ages of family members and the community they reside. Routine engagement and connectivity with services and support that understand these family’s needs would help foster positive relationships and interactions. The adoption of this model could assist in the identification of potential transition periods or life events that result in negative implications on family members as demonstrated in this research, and prompt scheduling of family reviews over the chronic and into the terminal phases (Rolland, 2005). In clinical practice, being able to conceptualise and action prompt responses to challenging circumstances could prevent a crisis, alleviating further anxieties and worries. However, the enablement of responsive actions is a cross-sectorial responsibility, fostering seamless links and partnership with other sectors and agencies, irrespective of funding source. Improving the capacity to respond to need could improve current job satisfaction and promote mutual learning opportunities. As reported by Cations et al. (2017, p. 12) having access to ongoing sustainable, ‘unique’, coordinated, and flexible ‘good’ services are clearly recommended by the family members. Their inclusion in service design gives them some control over their life and is empowering (Mayrhofer et al., 2017).

The co-creation of a family-focused services model can help inform service design, taking into consideration the dynamic relationships and interactions between the person living with YOD, family members and service providers. This model encourages more attention to interpersonal relations through adopting a socially inclusive approach. In principle, ‘citizens are looked at as co-creators of their own well-being' (Freire & Sangiorgi, 2010, p. 2). Family members are best placed to contribute to the design and development of services that are applicable and responsive to individual and family orientated solutions, around need (Clarke & Hughes, 2010; Edvardsson, Tronvoll, & Gruber, 2011; Freire & Sangiorgi, 2010; Mayrhofer et al., 2017; Sansoni et al., 2016). Considering the co-creation of services, where the users are ‘central not only to the design of services but also to their production and continuous development' (Freire & Sangiorgi, 2010, p. 3) is empowering, and improves social value and overall well-being (Mayrhofer et al., 2017; Mental Health Foundation, 2015). The theory underpinning the co-creation model draws attention to the societal influences on complex family's ‘cohesion, adaptability, and communication' (Rolland, 1990, p. 2). Raising awareness of societal influences living with YOD (Thomas & Milligan, 2018) could promote constructive attitude change towards a sense of hope (Surr, Smith, Crossland, & Robins, 2016); improve sensitivity and understanding of complex issues; break down barriers between healthcare, service providers, and family members, to assist in the adoption of more socially orientated practices. Challenging one's issues and beliefs in this process, including societal expectations and norms, can make valuable contributions to the successful shaping of enabling practices (Edwards, Voss, & Iliffe, 2014).

Adopting the framework for the co-creation of family-focused services model would ensure services are tailored to the unique needs of families (Cations et al., 2017; Mayrhofer et al., 2017), taking into account lifecycle and psychosocial demands (Mental Health Foundation, 2015; Rolland, 1990,1999; Thomas & Milligan, 2018), over the progression of dementia. Individual and family history, and biography and developmental stages would be routinely contemplated along with the fundamentals and variables of how families’ function within the community they reside (Oliver, 2009; Roach et al., 2014; Rolland, 1990). Families’ contribution to the design of future services would support equity for all, irrespective of geographical location, availability of appropriately skilled staff and specific services. Embracing technological platforms (Robinson et al., 2009) where health and social care providers, and family members come together to receive or deliver appropriate services, support, information and education opportunities is vital in this changing social world.

Strengths and limitations

Our study is one of the first to explore a theoretical framework to inform a family-focused service design model for individuals and family members living with YOD. Although the sample size is not predetermined in qualitative research, the limited numbers of people living with YOD, spouse carers, health and social care providers could be seen as a limitation. Further research is necessary to broaden understanding of family experiences through conducting interviews with all family members, ideally from diagnosis and at different times along the trajectory of dementia. Service providers supporting these families could also be interviewed at the same time to deepen understanding.

Conclusion

The co-creation of a family-focused services model for families living with YOD could provide a valuable foundation that underpins effective interactions, and informs service and policy development opportunities between service users, service providers, and stakeholders. The proposed model offers a socially focused theoretical paradigm for rethinking and fostering health and social care providers’ approach to design and development of services and support, incorporating cross-sectorial cooperation and education opportunities in collaboration with family members. This innovative model would focus on actions that foster enablement of the whole family unit to function well together as the dementia progresses. Enabling participation and engagement in the community, and appropriately investing in maintaining the emotional and physical well-being of the whole family to live and function well with dementia, is crucial. Further advancement and integration of this theoretically underpinned co-created, family-focused service model requires additional research but presents excellent opportunities for social change.

Footnotes

Author contributions

K Hutchinson designed the study, collected and analysed the data and wrote the first draft of the paper. C Roberts designed the study, supervised the data collection, analysed the data and assisted in writing the paper. P Roach analysed the data and assisted in writing the paper. S Kurrle contributed to the writing.

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

Karen Hutchinson is a physiotherapist and PhD research student with the University of Sydney conducting qualitative research into the impact of younger onset dementia (YOD) on self and the family. Her PhD thesis has recently been submitted for examination.

Chris Roberts is an academic general practitioner in the Faculty of Medicine and Health at the University of Sydney. His health services research focuses on the impact of complex interventions in primary and community care, including those living with chronic disease.

Pamela Roach is currently an adjunct assistant professor in the Department of Community Health Sciences at the University of Calgary, and also manages the Brain and Mental Health Research Clinics as part of the Hotchkiss Brain Institute, also at the University of Calgary. Her research foci include young onset dementia, family and patient experiences, qualitative methods, and using clinical registry data to improve care provision.

Susan Kurrle is a geriatrician at Hornsby Ku-ring-gai Hospital, New South Wales, Australia and Curran Professor in Health Care of Older People at the University of Sydney, Australia. She is the director of the Cognitive Decline Partnership Centre and has research interests in dementia care and service development, and elder abuse.

References

Barca

Thorsen

Engedal

Haugan

Johannessen

(2014). Nobody asked me how I felt: Experiences of adult children of persons with younger onset dementia. International Psychogeriatrics, 26(12), 1935–1944. DOI: 10.1017/S1041610213002639.

Barnes

Mercer

(2011). Exploring disability (2nd ed.). Cambridge: Polity Press.

Bate

Robert

(2006). Experience-based design: From redesigning the system around the patient to co-designing services with the patient. Quality and Safety in Health Care, 15(5), 307–310. DOI: 10.1136/qshc.2005.016527.

Beattie

Daker-White

Gilliard

Means

(2002). Younger people in dementia care: A review of service needs, service provision and models of good practice. Aging & Mental Health, 6(3), 205–212.

Brodaty

Donkin

(2009). Family caregivers of people with dementia. Dialogues in Clinical Science, 11(2), 217–228.

Carter

J. E.

Oyebode

J. R.

Koopmans

R. T. C. M.

(2017). Young-onset dementia and the need for specialist care: A national and international perspective. Aging & Mental Health, 1, 6. DOI: 10.1080/13607863.2016.1257563.

Cations

Withall

Horsfall

Denham

White

Trollor

Draper

(2017). Why aren't people with young onset dementia and their supporters using formal services? Results from the INSPIRED study. PLoS ONE, 12(7), 1–15. DOI: doi.org/10.1371/journal.pone.0180935.

Chappell

Goodley

Lawthom

(2001). Making connections: The relevance of the social model of disability for people with learning difficulties. British Journal of Learning Disabilities, 29, 45–50.

Clare

Cox

(2003). Improving service approaches and outcomes for people with complex needs through consultation and involvement. Disability & Society, 18(7), 935–953. DOI: 10.10180/0968759032000127344.

10.

Clarke

Hughes

(2010). Introduction: Family minded policy and whole family practice – Developing a critical research framework. Social Policy and Society, 9(4), 527–531.

11.

Edvardsson

Tronvoll

Gruber

(2011). Expanding understanding of service exchange and value co-creation: A social construction approach. Journal of the Academy of Marketing Science, 39(2), 327–339. DOI: 10.1007/s11747-010-0200-y.

12.

Edwards

Voss

Iliffe

(2014). Education about dementia in primary care: Is person-centredness the key?. Dementia, 13(1), 111–119.

13.

Evans-Roberts

Weatherhead

Vaughan

(2013). Working with families following brain injury. Revista chilena de neuropsicología, 8(2), 1–9.

14.

Fallowfield

Jenkins

(2004). Communicating sad, bad, and difficult news in medicine. The Lancet, 363(9405), 312–319. https://dx-doi-org.web.bisu.edu.cn/10.1016/S0140-6736(03)15392-5.

15.

Fontana

Frey

(2005). The interview. In N . Denzin

. Lincoln (Eds), Qualitative research (3rd ed., pp. 695–728). Thousand Oaks, CA : SAGE.

16.

Freire

Sangiorgi

(2010). Service design and healthcare innovation: From consumption to coproduction and co-creation. Paper presented at the Second Nordic Conference on Service Design and Service Innovation.

17.

Gale

Heath

Cameron

Rashid

Redwood

(2013). Using the framework method for analysis of qualitative data in multi-disciplinary research. BMC Medical Research Methodology, 13, 117.

18.

Gelman

Greer

(2011). Young children in early onset Alzheimer's disease families: Research gaps and emerging service needs. American Journal of Alzheimers Disease and Other Dementias, 26(1), 29–35.

19.

Gilliard

Means

Beattie

Daker-White

(2005). Dementia care in England and the social model of disability: Lessons and issues. Dementia, 4(4), 571–586. DOI: 10.1177/1471301205058312.

20.

Greenwood

Smith

(2016). The experiences of people with younger-onset dementia: A meta-ethnographic review of the qualitative literature. Maturitas, 92, 102–109.

21.

Hutchinson

Roberts

Daly

Bulsara

Kurrle

(2016). Empowerment of young people who have a parent living with dementia: A social model perspective. International Psychogeriatrics, 28(4), 657–668. DOI: 10.1017/S1041610215001714.

22.

Hutchinson

Roberts

Kurrle

Daly

(2016). The emotional wellbeing of young people having a parent with younger onset dementia. Dementia, 15(4), 609–628. DOI: 10.1177/1471301214532111.

23.

Irwin

Johnson

(2005). Interviewing young children: Explicating our practices and dilemmas. Qualitavie Health Research, 15(6), 821–831.

24.

Keady

Nolan

(2003). The dynamics of dementia: Working together, working separately or working alone?. In Nolan

Lundh

Grant

Keady

(Eds), Partnerships in family care; Understanding the caregiving career (pp. 15–32). Buckingham, UK: Open University Press.

25.

La Fontaine

Oyebode

(2014). Family relationships and dementia: A synthesis of qualitative research including the person with dementia. Ageing & Society, 34, 1243–1272.

26.

Luscombe

Brodaty

Freeth

(1998). Younger people with dementia: Diagnostic issues, effects on carers and use of services. International Journal of Geriatric Psychiatry, 13, 323–330.

27.

McDonald

Patterson

White

Butow

Costa

Kerridge

(2016). Correlates of unmet needs and psychological distress in adolescents and young adults who have a parent diagnosed with cancer. Psycho-Oncology, 25, 447–454.

28.

Mayrhofer

Mathie

McKeown

Bunn

Goodman

(2017). Age-appropriate services for people diagnosed with young onset dementia (YOD): A systematic review. Aging & Mental Health, 1–9. DOI: 10.1080/13607863.2017.1334038.

29.

Mental Health Foundation. (2015). Dementia, rights, and the social model of disability. Policy discussion paper. London.

30.

Millenaar

Bakker

Koopmans

Verhey

Kurz

A., D.

Vugt

(2016). The care needs and experiences with the use of services of people with young-onset dementia and their caregivers: A systematic review. International Journal of Geriatric Psychiatry, 31(12), 1261–1276. DOI: 101002/gps.4502

31.

Millenaar

J. K.

Bakker

van Vliet

Koopmans

R. T. C. M.

Kurz

Verhey

F. R. J.

de Vugt

M. E.

(2017). Exploring perspectives of young onset dementia caregivers with high versus low unmet needs. International Journal of Geriatric Psychiatry, 33(2), 340–347. DOI: 10.1002/gps.4749.

32.

Murphy

Jordan

Hunter

Cooney

Casey

(2015). Articulating the strategies for maximising the inclusion of people with dementia in qualitative research studies. Dementia, 14(6), 800–824.

33.

Murray

Barnes

(2010). Have families been rethought? Ethic of care, family and ‘whole family’ approaches. Social Policy and Society, 9(04), 533–544. DOI: 10.1017/S1474746410000254.

34.

Nolan

Ryan

Enderby

Reid

(2002). Towards a more inclusive vision of dementia care practice and research. Dementia, 1(2), 193–211.

35.

Oliver

(1983). Social work with disabled people (1st ed.). Basingstoke, UK: Palgrave Macmillan.

36.

Oliver

(2009). Understanding disability from theory to practice (2nd ed.). Basingstoke, UK: Palgrave Macmillan.

37.

Oliver

(2013). The social model of disability: Thirty years on. Disability and Society, 28(7), 1024–1026.

38.

Patterson

Clarke

Wolverston

Moniz-Cook

(2017). Through the eyes of others – The social experiences of people with dementia: A systematic literature review and synthesis. International Psychogeriatrics, 1–15. DOI: 10.1017/S1041610216002374.

39.

Pauly

Varcoe

Storch

(2012). Framing the issues: Moral distress in healthcare. Healthcare Ethics Committee Forum, 24, 1–11.

40.

Pratt

Clare

Kirchner

(2006). 'It's like a revolving door syndrome': Professional perspectives on models of access to services for people with early stage dementia. Aging and Mental Health, 10(1), 55–62. DOI: 10.1080/13607860500307530.

41.

Ritchie

Lewis

Nicholls

Ormston

(2013). Qualitative research practice: A guide for social science students and researchers (2nd ed.). London, UK: Sage.

42.

Roach

(2010). A family-centred study of younger people with dementia (PhD Thesis). University of Manchester, UK.

43.

Roach

Drummond

Keady

(2016). Nobody would say that it is Alzheimer's or dementia at this age: Family adjustment following a diagnosis of early-onset dementia. Journal of Aging Studies, 36, 26–32.

44.

Roach

Keady

Bee

Williams

(2014). We can't keep going on like this’: Identifying family storylines in young onset dementia. Ageing & Society, 34(08), 1397–1426. DOI: 10.1017/S0144686X13000202.

45.

Robinson

Emden

Lea

Elder

Turner

Vickers

(2009). Information issues for providers of services to people with dementia living in the community in Australia: Breaking the cycle of frustration. Health and Social Care in the Community, 17(2), 141–150.

46.

Robinson

Cottrell

(2005). Health professionals in multi-disciplinary and multi-agency teams: Changing professional practice. Journal of Interprofessional Care, 19(6), 547–560. DOI: 10.1080/13561820500396960.

47.

Rolland

(1987). Chronic illness and the life cycle: A conceptual framework. Family Process, 26(2), 203–221. DOI: 10.1111/j.1545-5300.1987.00203.x.

48.

Rolland

(1990). Anticipatory loss: A family systems developmental framework. Family Process, 29(3), 229–244. DOI: 10.1111/j.1545-5300.1990.00229.x.

49.

Rolland

(1999). Parental illness and disability: Family systems framework. Journal of Family Therapy, 21, 242–266.

50.

Rolland

(2005). Cancer and the family: An integrative model. Cancer, 104(S11), 2584–2595. DOI: 10.1002/cncr.21489.

51.

Rossor

Fox

Schott

Warren

(2010). The diagnosis of young onset dementia. Lancet, 9(8), 793–806. https://dx-doi-org.web.bisu.edu.cn/10.1016/S1474-4422(10)70159-9.

52.

Rushton

(2016). Moral resilience: A capacity for navigating moral distress in critical care. Advanced Critical Care, 27, 111–119.

53.

Sansoni

Duncan

Grootemaat

Capell

Samsa

Westera

(2016). Younger onset dementia: A review of the literature to inform service development. American Journal of Alzheimer's Disease and Other Dementias, 31(8), 693–705. DOI: 10.1177/1533317515619481.

54.

Shakespeare

Zeilig

Mittler

(2017). Rights in mind: Thinking differently about dementia and disability. Dementia. 18(3), 1075–1088.

55.

Surr

Smith

Crossland

Robins

(2016). Impact of a person centred dementia care training programme on hospital staff attitudes, role efficacy and perceptions caring for people with dementia: A repeated measures study. International Journal of Nursing Studies, 53, 144–151.

56.

Svanberg

Spector

Stott

(2011). The impact of young onset dementia on the family: A literature review. International Psychogeriatrics, 23(3), 358–371.

57.

Thomas

Milligan

(2018). Dementia, disability rights and disablism: Understanding the social position of people living with dementia. Disability and Society, 33(1), 115–131. DOI: 10.1080/09687599.2017.1379952.

58.

van Vliet

de Vugt

M. E.

Bakker

Koopmans

R. T.

Verhey

F. R.

(2010). Impact of early onset dementia on caregivers: A review. International Journal of Geriatric Psychiatry, 25, 1091–1100.

59.

van Vliet

de Vugt

Bakker

Koopmans

Pijnenburg

Vernooij-Dassen

Verhey

(2011). Caregivers' perspectives on the pre-diagnostic period in early onset dementia: A long and winding road. International Psychogeriatrics, 23(9), 1393–1404. DOI: 10.1017/s1041610211001013.

60.

Withall

Draper

Seeher

Brodaty

(2014). The prevalence and causes of younger onset dementia in Eastern Sydney, Australia. International Psychogeriatrics, 26(Special Issue 12), 1955–1965. DOI: 10.1017/S1041610214001835.