Services for people with young onset dementia: The ‘Angela’ project national UK survey of service use and satisfaction

Young onset dementia is associated with distinctive support needs but existing research on service provision has been largely small scale and qualitative. Our objective was to explore service use, cost and satisfaction across the UK.


| INTRODUCTION
Although dementia predominantly affects older people, an estimated 5.5% of those diagnosed have young onset dementia (YOD); where symptoms develop <65 years. 1 UK numbers are estimated as 42,325. 2 Those living with YOD have distinctive and wide-ranging needs due to age, life stage, [3][4][5][6] and the range of rare diagnoses. 7 Specialised, tailored, well-integrated YOD services, that provide continuity of care, are widely seen as central to addressing these challenges. [8][9][10][11][12][13] Available evidence, however, indicates problematic issues and gaps in provision. Delays to diagnosis and misdiagnosis are common 8,11 due to the rarity of YOD and the heterogeneity of presenting symptoms, which leads to multiple pathways to diagnosis. 13,14 In addition, effective post-diagnostic services are lacking. 15 In a recent UK-wide survey of healthcare professionals involved in services for YOD, 54% reported no access to a consultant with a special interest, 28% reported no post-diagnostic support and only 25% reported access to age-specific post-diagnostic support. 16 Consultation with people with YOD and family carers 17,18 highlights key barriers to post-diagnostic support, including lack of: ageappropriate services information to allow timely access and poor service continuity in the transition from diagnosis to post-diagnostic support.
Existing studies on post-diagnostic YOD services have tended to be small-scale and qualitative, with a lack of information on service costs. 8,11,12 This paper reports the findings from a large-scale, UKwide survey of people living with YOD and carers. The aim was to gather baseline information on current YOD service delivery and explore how patterns of care link to quality of care, user satisfaction and costs, in order to inform commissioning and service provision.

| METHODS
A cross-sectional UK-wide survey was conducted, gathering data from people with YOD and/or an involved family member or supporter (referred to from hereon as 'family member') on sociodemographic characteristics, service use, satisfaction and positive experiences of support. This article reports the service use, cost and satisfaction data.

| Measures
The survey (Data S1) was developed by the research team and The opening question of the survey filtered respondents to four different completion pathways (person with YOD alone, with family member or with paid carer or family member alone). Wording was customised accordingly (e.g., referring to 'you' for person with YOD and 'the person with YOD' when a family member was the respondent).

| Participants
Those with a confirmed YOD diagnosis and family members/ supporters were eligible to participate. People with dementia related

Key Points
� In the absence of detailed information on service use, costs and satisfaction for people with young onset dementia (YOD), a large national cross-sectional, selfreport survey was conducted of people with YOD and/or close supporters, receiving 233 usable responses � Nearly 60% of family members/supporters provided over 5 h of care a day for the person with YOD, with family members'/supporters' care being valued at over 20 times the cost of support from formal services � The majority of respondents were mostly or very satisfied with their care from services with higher satisfaction being associated with being managed by a specialist service and knowing who to contact with questions; reporting nobody or a general practitioner alone managed care on a regular basis was associated with lower satisfaction � Specialist YOD services performed better than other types of service on quality indicators, including provision of support in the immediate period after diagnosis, continuity of services and providing care plans and key workers 412 - to Down's syndrome, HIV, traumatic brain injury, Huntington's disease or alcohol-use were excluded, as these populations usually access different services. Participants were recruited through 14 UK National Health Service sites, purposively selected to include different diagnostic pathways (specialist YOD services, neurology, generic dementia services). A wide range of third-sector organisations also advertised the survey and information was distributed via Join Dementia Research, a UK National Institute of Health Research register of potential participants. Sample size was not pre-defined but we aimed to recruit as many participants as possible.  Costs of care (British pounds 2018) were calculated over a 3-month period, based on reported service use, in five groups: nurses and allied health professionals, medical, hospital inpatient, social care, family care (Table S3 gives details and unit costs).

| Analysis
Multiple linear regression was performed for three outcome variables: satisfaction with care, total cost of health and social care, informal caring hours. Predictor variables included the characteristics of the person with YOD and the services they received (see Results for full list). Backward elimination was used to derive an initial model. This was then rerun with just the statistically significant predictor variables, to maximise the number of observations used, owing to the presence of scattered missing data.

| Sample characteristics
There were 233 usable responses. 20 Data were obtained about the person with YOD from all responses (n ¼ 233) and from 185 family members (when a family member was involved in completion). In over half the responses, a person with YOD was involved in survey completion, alone (n ¼ 39, 16.7%), with a paid carer (n ¼ 9, 3.9%) or with a family member (n ¼ 84, 36.1%). There were 101 (43.3%) family members completing the survey on their own, meaning family members were involved in almost 80% of all responses. There were no statistically significant differences between survey completion pathways on any characteristic related to the person with YOD or family member (data not shown).
Those with YOD were evenly divided between women and men, with a mean age of 62 years. They reported receiving the diagnosis a mean of 3.8 years previously, at the average age of 58 years (range 37-65). Just over half had Alzheimer's disease and the next most common diagnosis (n ¼ 30, 12.9%) was a variant of fronto-temporal dementia; the rest reported a variety of diagnoses, with 18 (7.7%) not knowing or not reporting dementia type. They were taking on average over four prescription medications. A small number reported being in paid employment, living alone or in a care home; just over 10% reported having children living in the household. Just over a quarter of those with YOD were not comfortable to remain home alone at all; just under a quarter were comfortable to be alone for 24 h or longer (Table 1).
Most family members were spouses and female, with a mean age of 59 years. They were taking on average one prescription medication.
Over one third were in paid employment and more than half reported giving up work to provide care. Those in employment were, on average, younger (58.9 vs. 63.7 years) and there was less time since diagnosis (2.7 vs. 4.5 years; both, independent t test p < 0.0005; Table 1).
The annual household income reported for the person with YOD was low, with a third reporting this to be under £12,000 and almost a further third reporting income between £12,001 and £20,000 (data not shown). £18,400 has been calculated as the minimum for an acceptable standard of living. 21 STAMOU ET AL.

| Diagnostic services
Over a third of respondents received their diagnosis in a memory clinic, around a quarter in Neurology; with under a fifth in older people's mental health services or a specialist YOD service (Table 2).
Eighty-six percent of respondents reported that both brain scans and memory tests were used during assessment. Around 40% of respondents reported receiving no follow-up during the 6-weeks after diagnosis. Rates varied significantly between types of service, being lowest (22%) in specialist YOD services and highest (61%) in Neurology (See Table S2, for detail).

| Management of ongoing care
Care arrangements varied. About one fifth reported their care was managed solely by a specialist YOD service and another fifth solely by a general practitioner (GP). The rest reported a range of single service arrangements or shared care; around 16% reported nobody managed their care. Those in rural areas were more likely to be receiving care from their GP alone (Table 2).
Almost half of those diagnosed in specialist YOD services continued to receive ongoing care from those services, whereas under a quarter who were diagnosed in another type of service received ongoing care from that same type of service. The rest were discharged to GP care (alone or shared), referred to another service, reported no on-going management or didn't answer the question. Of those who did not know who regularly managed their care, 40% had been diagnosed in a memory clinic and a quarter in neurology. Nearly one third of all respondents reported having no routine follow-up appointments. Frequency for those who did have appointments varied widely. There was no significant difference between service types, urban or rural location, diagnosis or time since diagnosis regarding frequency of follow-up (Table 2).

| Professionals seen and activities attended
Approximately 39% reported that the person with YOD had seen no health professional in the last three months. The proportion was highest among those whose care was managed by GPs alone and lowest among those whose care was managed in a specialist YOD service. Sixteen different types of health or social care professionals were listed as involved in care, the most common being mental health nurses, social workers and psychiatrists. Of other service contacts in the last 3 months, just over half had attended social activities, around a quarter had a visiting home carer and an eighth had received a home visit from a voluntary agency (Table 3).

| Costs of care
Costs of care (£, 2018), over 3 months, are summarised in Table 4.  There was no statistically significant difference between the three diagnostic groups with regard to frequency of appointments with service/doctor seen the most (Kruskal-Wallis test: p ¼ 0.191).

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T A B L E 3 Support for person with young onset dementia (YOD) and carer in previous three months -417

| Quality and satisfaction with care
Specialist YOD services performed better than other services on all quality indicators. In specialist YOD services, almost all reported seeing the same professional at each appointment whereas this was the case for only just over half of those whose care was managed solely by their GP. Under a third of the overall sample reported having a care plan and the number with a key worker was not much higher (38%), though in specialist YOD services it was 76.3% (Table 5).
Around 60% of respondents reported that their care was mostly or very good whilst 24% felt their care was mostly or very poor, the rest being equivocal. The mean scores suggest people receiving care from specialist YOD services were most satisfied. Family members completing the survey alone expressed lower service satisfaction than was expressed through other completion pathways (Table 5).

| Regression modelling
Five variables explained 30.2% of the variation in satisfaction scores.
Knowing who to contact, being managed in a specialist YOD service and living in the south-east of England were positively associated with satisfaction; reporting nobody or a GP alone managing care on a regular basis were associated with lower satisfaction (Table 6).
Total costs of health and social care over three months were associated with the amount of time the person with YOD could comfortably be left alone (higher cost with less time person could be left alone), dementia diagnosis (Alzheimer's less cost than any other type) and region (higher cost in South-East England) but this model explained only 12% of variation in costs ( Table 6).     23 It is notable that our survey found lower service costs and higher family costs for YOD than calculated for older people with dementia. Regression modelling confirmed that costs were positively related to the level of independence of the person with dementia, proxied by the amount of time they could be left alone, as also found for all-age dementia care. 23 One unexpected finding was that those with YOD were being prescribed significantly more medications than family members/ supporters, even though they were similar in many aspects of social demography. Even though their medications may have included an anti-dementia medication, this difference raises an issue about the health and medication use of those with YOD which may be worthy of further exploration, particularly given previous research indicates high levels of psychotropic drug prescription for people with YOD living in the community. 24 The significant contribution of region, to respondents' satisfaction with services, service costs and amount of family care, implies possible geographical variation in provision. 16 This could imply the need to level up services across the UK or could be due to regional cultural differences.

| Strengths and limitations
The survey achieved a large sample with over half of responses directly involving people living with YOD. A reasonable representation across UK regions, types of dementia, age of onset, current life situation and degree of independence/dependence was achieved.
Although mean time since diagnosis (3.8 years) was relatively short, there was a relatively even split of those providing care for under, and over, 5 h per day (42.7% vs. 57.3%), and in the extent to which people with YOD could be at home alone, implying a reasonable degree of variation in extent of impairment due to dementia.
While this was the largest UK study focused on services for YOD to date, the numbers of those with rarer dementias were small, leading to a decision to combine a range of different diagnoses into an 'other' category for comparative analyses. Consequently, we were unable to discern different patterns between diagnostic groups. Furthermore, people with more severe cognitive impairment may have been under-represented. These areas deserve further investigation, in particular to consider the service profile and satisfaction of those with fronto-temporal dementia (FTD), given the distinctive impact of FTD diagnoses. In addition, the survey relied on self-report from those with dementia or their supporters so information may include errors. Compromises in the survey questions were made to achieve a balance between accessibility and comprehensiveness, which means some detail could not be precisely gathered.

| Clinical implications
The findings demonstrate that specialist YOD services provide higher quality care both by objective indicators and in satisfaction ratings by people with YOD and family members. This, in the context of wider evidence that interventions and activities provided in generic dementia services are inappropriate and unacceptable for those with YOD, 15,17,18 implies that local commissioners should seek to configure dementia services to include specialist YOD teams. Where diagnosis does not take place in a specialist YOD service, attention needs to be paid to transitional arrangements between diagnosis and post-diagnostic support.

| CONCLUSIONS
There is great variation within diagnostic care pathways and ongoing care management for YOD in the UK, leading to disparate experiences. Overall, our survey respondents received few services and absorbed few resources. Specialist YOD services were associated STAMOU ET AL.
-421 with better continuity, quality and satisfaction and appeared to be in the best position to meet care needs. However, respondents in receipt of specialist care were in the minority, indicating that further specialist services should be commissioned. Family members are providing significant amounts of care on a daily basis with little formal support, and meeting their needs is also a priority.