Children and young people's experience of parental dementia: A systematic review

Abstract Objectives Most studies have been concerned with the experiences and needs of spouses/partners and adult children of people with dementia. In this review, children and young people's lived experience of parental dementia was investigated. Findings will inform both researchers and professionals in the area of dementia care. Design A systematic literature search was performed in CINAHL, PsychINFO, PubMed, Scopus, and Web of Science. A rigorous screening process was followed, and a checklist for qualitative and observational studies was used to evaluate the methodological quality of the studies. Narrative synthesis of the selected articles was carried out. Results Twenty‐one studies were included and a synthesis of the literature revealed six themes. The first theme concerned the difficulties in dealing with the diagnosis which was often preceded by a long period characterized by uncertainty, confusion, family distress, and conflicts. The second theme discussed changes in family relationships in terms of the role of children and young people in supporting both parents and keeping family together. The third theme described the impact of caring on children and young people who struggled to balance caring tasks and developmental needs. The fourth theme showed consequences on children and young people's personal lives in terms of education/career and life planning. The fifth theme illustrated main adaptation models and coping strategies. The last theme discussed the need for appropriate support and services based on a “whole family” approach. Conclusions The included studies provide the basis for knowledge and awareness about the experience of children and young people with a parent with dementia and the specific needs of support for this population.


| Young carers
In the last decades, apart from dementia, the issue of living and/or caring for a sick parent at young age has received increased attention due to recent changes in society. 14 Nowadays, there is no universally agreed international definition of the youth age group, and youth is a more fluid category than a fixed age-group. 15 Actually, increasing numbers of young people tend to live in family home at their 30s, and are still financially dependent on their parents, they attend university courses or have temporary jobs. 16,17 In the case of parental disease, young people may be more vulnerable due to their age and developmental needs that require them to face the new life challenges (e.g., getting a job, attending university, starting their own family) without guidance and support by their parents. [18][19][20] Statistics indicate that between 4% and 10% of young people care for an ill or disabled parent. 21,22 The term "carer" (also known as caregiver) refers to anyone who carries out, on a regular and unpaid basis, significant caring tasks for a friend or family member who cannot cope alone because of an illness or other condition. 23 Caring responsibilities include practical tasks (e.g., cooking, housework, shopping); physical and personal care (e.g., helping someone out of bed, get dressed or with intimate care); emotional support and supervision (i.e., safety and health monitoring). These tasks are often carried out by young people without supervision and assistance, and, in many cases, are comparable to those fulfilled by adult carers. 24 Furthermore, young people living and/or caring for a sick parent tend to have more mental health problems and more difficulties in behavioral, psychosocial, and academic adjustment than their peers without an ill parent. 23,24 Since caring can be viewed as a natural extension of family relationships, young people often do not receive adequate support from public authorities, social policy, health, and social services. 23,24 Despite these findings, most recent studies on the positive impact of caring found that a higher sense of self-mastery, self-worth, maturity and empathy are mitigated by the level of support that young carers are provided with. 21,25 Therefore, if adequate support is available, positive aspects may coexist with the adverse outcomes of caring at young age. 26

| The present study
While most research to date has focused on spouses/partners and adult children of people with dementia, less empirical evidence exists on children and young people's experience of parental dementia as told by themselves. Based on studies with adult samples, parental dementia in families with children and young people leads to greater social and psychological impairment, damage, tension, hardship, and family breakdown than it does in families with adult/middle aged children. [27][28][29][30][31][32][33] The few available reviews [9][10][11] focused on the experiences of family caregivers of a relative with YOD. Specifically, the review by Van Vliet et al. 11 did not include studies on children and/or young people, while in two reviews 9,10 results were discussed without differentiating among adult and young offsprings. Hence, the aim of this review is to fill this gap by systematically ascertaining the

| Search strategy
The review follows PRISMA guidelines 34  The screening process consisted of three stages: duplicate removal, titles/abstracts screening, and full-text scrutiny. The titles and abstracts of all studies were assessed independently by two reviewers (I. Chirico and M. Valente), and any discrepancy was resolved by a third reviewer (G. Ottoboni) through discussion until an agreement was reached. Finally, full-paper articles of any relevant titles/ abstracts were obtained and reviewed independently by two members of the review team (I. Chirico and M. Valente) with reasons for exclusion annotated; again, any discrepancy was resolved by a third reviewer (G. Ottoboni).

| Inclusion/exclusion criteria
In this review, as to reflect current situation in the society, a broad definition of the term "young people" was adopted. 14,15 Specifically, the term "children" refers to individuals aged 6-10 years, while the term "young people" includes adolescents (11-18 years) and young adults (19-35 years). 16,17 Articles were included if they focused on: (a) children (aged 6-10 years) and/or young people (aged 11-35 years) (i.e., population of interest); (b) children and/or young people living and/or caring for a parent with dementia (i.e., context of interest); (c) children and/or young people's experience of parental dementia as told by themselves (i.e., outcome of interest); and (d) they reported results of peer-reviewed research based either on quantitative, qualitative, or mixed method studies. Studies where only a subsample met the eligibility criteria were included if outcomes focused on the population of interest were separately considered and analyzed.
Articles were excluded if they focused on: (a) only spouses/ partners and/or adult children (i.e., not population of interest); (1b) children and/or young people living and/or caring for a relative other than their parent (i.e., not context of interest); (2b) children and/or young people living and/or caring for a parent without disease or with other diagnosis than dementia (i.e., not context of interest); (c) causes of dementia, prevalence and incidence, medical considerations, evaluation and assessment of interventions (i.e., not outcome of interest); and (d) they did not report empirical findings. Due to the exploratory nature of this review, there were no restrictions on the type of data to look for and extract.

| Quality appraisal
The methodological quality of the included studies was assessed using a quality checklist for observational studies with 23 criteria, 35 and a quality checklist for qualitative studies with 12 criteria. 36 If the criterion was met, it was rated with "a+" and unmet with "a−," and when the criterion was not completely met, it was rated with "+/−." If the criterion was not applicable, it did not receive any rating. The quality appraisal was carried out independently by two members of the review team (I. Chirico and M. Valente), and interrater reliability was substantial with a Cohen's kappa of 0.75. 37 After a consensus meeting with a third reviewer (G. Ottoboni) both raters reached full agreement on the quality ratings.

| Data extraction and synthesis
A standardized data extraction form 38 was used to examine the main characteristics (i.e., author/s, year, country, design, population, measures) and key results of the included studies. By using this form, key issues of each study were identified. 39 An inductive thematic analysis 40

| Study characteristics
After duplicate removal, the search identified a total of 651 hits ( Figure 1). Of these, 628 records were rejected based on the title or abstract, and two papers 27,41 were not eligible based on the full-text scrutiny. Hence, the final sample included 21 studies.
The relevant features of each study (i.e., author/s, year, country, design, population, measures) and key outcomes are presented in Table 2. All studies took place in the West: United Kingdom (n = 10), United States (n = 4), Norway (n = 3), Australia (n = 2), Netherlands (n = 1), and Sweden (n = 1). As regards the study design, 18 papers used a qualitative approach (10 cross-sectional and 8 longitudinal), -983 As shown in were used in qualitative studies: semi-structured (n = 10), unstructured (n = 7), and focus group (n = 1); the only quantitative study used an online survey while the remaining two studies used a mixedmethodology consisting of questionnaires and semi-structured interviews.

| Methodological aspects
Most qualitative studies scored high and the main weaknesses were concerned with the limited description of the sample and/or sampling method (Table 3). Specifically, the inclusion and exclusion criteria were not always specified, and not every study explicitly described For what concerns the three observational studies, several methodological concerns need to be raised (Table 4). Participants were not representative of the population, sample sizes were small and inclusion and exclusion criteria were not always fully described. 48,50 Moreover, there was a lack of control for possible confounders. The articles often included children of only parents with specific diagnoses such as Alzheimer's disease 50 or frontotemporal dementia 48 and it was unclear how the diagnosis was established.
Research hypotheses were always omitted thus making it difficult to interpret the results. Finally, the type of study was never mentioned and outcomes were sometimes neither validated 49 nor clearly described as means and standard deviations were not reported, 50 and response rates were not available in two studies. 49 as their parents gradually lost interest in them, exhibited aggressive and embarrassing behaviors and they did not know how to react.
Participants experienced confusion, disorientation, fear, and sadness which rose, in some cases, to the level of emotional trauma. 51,56,58 They disliked the parent who had become, and some spoke of "relief" for them and "release" for their parent when death finally came. 53,55 The relationship among participants and their parents with dementia was usually characterized by "latent grief," and an ongoing and unmitigated loss as the condition worsened. 45,48,49,51,52,58,59 Parents were physically present but emotionally absent, and they were no longer available in any meaningful parental way. 45,46,[53][54][55]58 Participants told about their unique experience of grieving the loss of their parents due to a disease that, although fatal like other illnesses, society stigmatized creating an aura of uncaring. 59 Several concerns for the well parent were raised and they often assumed the role of "protectors" in the attempt to reduce the burden on their healthy parents. 45,51,52,54,57 In the study of Lövenmarck 60 they became orphans with parents, that is, parents to both of their parents in order to support and keep them healthy for as long as possible.
In the study of Hall and Sikes 43

| Caring
Participants had extensive caring responsibilities including physical support of the parent such as helping with feeding and ambulating, household chores but also companionship, talking with healthcare professionals, driving for errands. 48,54 They provided not only instrumental care, but also emotional care to their healthy parents as to comfort them, mediate conflicts and keep the family together. 57,59 Caregiving took an emotional toll on the participants as they struggled to find a balance between being a young person and a caregiver. 48,51,52,54,59 The impact of caring varied from ongoing stress to a medically diagnosed mental health condition that then compounded their disability such as depression, anxiety, psychosis, obsessive-compulsive disorder, self-harm, alcohol and substance -987 addiction, suicide ideation. [49][50][51]59 Some positive aspects associated with the experience of caring were also reported such as increased maturity and the opportunity to spend time with their parent. 48,54 Levels of participants' burden were different depending on the previous family relationships, the family reorganization after diagnosis as well as the nature and severity of parental symptoms and the speed of decline. 45,52,58 They were higher in families where participants lived with a single parent with dementia or when families were denied social security, the healthy parent struggled to find a job or was dealing with his/her own emotional issues. 59 On the contrary, levels of burden were lower in families where the well parent was the primary caregiver, and managed the situation in ways participants found reasonable, or when family members like siblings collaborated and shared the tasks. 45,48 Furthermore, those who lived outside of the home seemed to be less influenced by their parent's illness than their peers living at home. 45

| Personal life
Parental dementia strongly affected participants' choices, time perspectives and life planning in relation to education/career, mobility and personal lives. 43,46 Participants felt their lives were in "limbo," in "betwixt and between" 46 , p. 245. For some, the future was a source of deep anxiety, while others were unable to contemplate life beyond their parents' illness, and life was put on hold. 52 They had fewer possibilities to focus on their development and were not able to develop an identity as they would have liked during their youth. 52,56,58,60 Participants' concerns and lack of concentration sometimes hindered progress in their studies and career; some had interrupted their studies or work to return home and take care of their parents. 45 Educational choices were done, at least partly, because of parental dementia or decisions not to move were based on the need to stay close to their family and spend time with the parent with dementia. 47 Many participants at school pointed out the disparity in being acknowledged as young carers due to the lack of awareness about YOD among teachers. 47,61 When the well parent informed the teaching staff about the situation at home, schools often did not accommodate the timetable to students' needs by distance learning for example. In most cases teachers did not understand participants' experience, the schools did not sensitively match their response to students' needs through the provision of professional help like a counselor. Furthermore, when the condition was known, students could have been premature. Indeed, some participants did not see themselves as carers, and did not feel they had sufficient responsibilities to warrant this label. 45,51 As regards coping strategies, in the study of Allen et al. 51 young people reported mainly emotion-focused coping, but also problemfocused coping in that they helped with care, supporting siblings, and obtaining formal help. Maladaptive strategies were denial, social and emotional withdrawal, smoking, abuse of alcohol and self-harm.
In Aslett et al.'s 52 young people employed problem-focused coping strategies when duties were tangible, while a lack of mastery or control was associated with feelings of hopelessness. Fears over genetic risk were managed by adopting mindful coping strategies that focused on living in the moment. 51,52 Some participants grew up stronger, excelled in school, involved into academic and extracurricular activities as ways of coping and distraction; others told that 988this experience reinforced their faith which, in turn, helped them to cope better. 49,50,58 Other supportive activities included being with friends, sports, music, staying busy, video games, and staying away from home. 48 Millenaar et al. 57 found that, especially at the beginning, young people adopted avoidant ways of coping because they did

| Care and support
Participants felt neglected by family, friends, health, and social professionals. They experienced discrimination and marginalization due to the way services were designed and delivered. [44][45][46][47][48][49]52,54,56,[59][60][61] On the one hand, they felt isolated from the community but, on the other hand, they often did not reveal their situation as not to draw attention of their situation to social services and to avoid the stigma. 59,61 Participants were mainly concerned about the needs of their parents, and they were more likely to ask for help after watching their healthy parents do the same. 54 Assistance with activities of daily living and mobility was needed as well as a better support for their family. The latter one should allow the well parent to retain his/ her role, and children and young people to assume more age appropriate roles and be successfully engaged into school and work. 45,49,54 Since the lack of personal experience acted as a barrier to communication, participants preferred to talk with at least one professional who was familiar with their situation and with knowledge of the disease and available services. This was preferred to support provided by sporadically visiting healthcare professionals who were felt too impersonal. 54,60 Similarly, being involved with peer support groups who had personal experience of dementia was considered valuable in managing stress, burden, guilt and increasing their understanding of dementia. 45,52 Participants often used the Internet and social media to obtain information and advice, and to make contact with others with similar experiences. 44 At the onset of parental symptoms and, throughout the disease trajectory, participants were "lost in the chaos" 51 and accepting the diagnosis was difficult. Over time participants had increasing responsibilities as adults, while they experienced a "latent grief" 54 as their parent was no longer emotionally available although physically present. At the same time, they provided instrumental and emotional care to both parents as to keep the family together. In this scenario, they experienced similar levels of emotional distress to those observed in the adult carers. [49][50][51]59 Participants had to deal with the unpredictability of dementia and their plans in relation to education/career, mobility, and personal lives were strongly influenced by their parent's condition.
Some had worsened in school or had even given up their studies. At school they were stigmatized and their educational needs in relation to parental dementia were neglected. 44 At the same time support from extended family and friends was greatly disturbed. 56,62 Hence, their feelings of neglect and marginalization were similar to those experienced by young carers of parents with other mental disorders. 59,61 It is important to note that caring at young age was sometimes viewed as a natural extension of family relationships. 49 Participants described the existing services as silo-based and inappropriate for their needs. A "whole family" approach should be based on a strong cooperation among adult, child, health, and voluntary sectors. This joint work should be facilitated by a case manager with knowledge of the family situation, and capable to organize specific care as to alleviate the burden on the well parent which, in turn, would decrease the pressure on their children. 60 Professionals should have appropriate interdisciplinary training with CHIRICO ET AL.
-989 knowledge of dementia, consequent changes in family dynamics, and children and young people's developmental needs. [44][45][46]52,56 It is nevertheless important to point out that not all of them will experience these difficulties depending on the availability of support. 47,58,59,61 Broader societal views and a greater public understanding may promote increased support at the policy level, and better emotional and practical support for children, young people and their families. 45,58,63 Psychoeducation, face-to-face groups, online forums and/or blogs may suit the needs of support for this population. 52,54

| Limitations
Although the review was rigorous, the gray literature was not

| Conclusions
Findings should contribute to raising awareness about the peculiar and often "invisible" experience of young people with a parent with dementia. Furthermore, they should inform research, practice, program development, and policymakers in the area of dementia care.
Around the world, there are already some good practices (nondisease specific) including young carer groups, forums, some awareness campaigns, ICT and web-based interventions. 25 Preventive actions should avoid the negative consequences of caring at young age which, in turn, would save society the costs of increased healthcare. 25,64,65 Since dementia caring can be variable over time,