Health and Social Care in the Community (2016) 24(4), 375–385

doi: 10.1111/hsc.12225

Review What is known about the subjective needs of people with behavioural variant frontotemporal dementia? A scoping review 1,2,a € Claudia Dinand MScN1,2,a, Sabine Ursula Nover Dr rer pol, Dipl Okon (Sociology) , Daniela Holle 1,2,a 1 1,2 , Matthias Zischka BScN and Margareta Halek Dr rer medic, MScN MScN

Dr rer medic,

1

Witten, Research Group: Care Interventions, German Center for Neurodegenerative Diseases (DZNE), Witten, Germany and 2Faculty of Health, School of Nursing Science, Witten/Herdecke University, Witten, Germany Accepted for publication 10 February 2015

Correspondence Claudia Dinand Research Group: Care Interventions German Center for Neurodegenerative Diseases (DZNE), Stockumer Straße 12 58453 Witten, Germany E-mail: [email protected] a

Equal contributors.

What is known about this topic

• •



Behavioural variant frontotemporal dementia (bvFTD) is an early-onset dementia and causes specific challenges in everyday life. Pharmacological interventions are not always effective, psycho-social interventions are not evidencebased, and care services are mostly designed for people with dementia in later life, but not for people with bvFTD. Psycho-social interventions are most effective if they match the specific needs and demands of the person concerned.

What this paper adds

• • •

Identifies a research gap in the literature regarding the selfexpressed needs of people with bvFTD. Highlights the lack of appropriate methods to explore the needs of people with bvFTD. Identifies a need to develop and test psycho-social interventions for people with bvFTD.

© 2015 John Wiley & Sons Ltd

Abstract Behavioural variant frontotemporal dementia (bvFTD) is an early-onset and progressive neurodegenerative disease associated with strong changes in judgement, behaviour, personality and emotions. These changes can cause significant problems in everyday life for people with bvFTD and their families, and have implications for health and society. Currently, there are no suitable evidence-based specific interventions for people with bvFTD. This scoping review aims to identify the selfexpressed needs, demands and coping strategies of people with bvFTD. Identifying these issues is the first step towards the development of needbased psycho-social interventions for people with bvFTD. A comprehensive literature research was conducted of German and English scientific articles published between January 2000 and October 2014 using the databases MEDLINE, CINAHL, PsycINFO, PSYNDEX, SocINDEX, GeroLit, the Cochrane Library, ProQuest, the German National Library and additional search strategies in terms of a scoping review. Articles were identified by combining search terms related to ‘frontotemporal dementia’ with terms related to ‘self-expressions’, ‘needs/demands’ and ‘coping’. After excluding duplicates, two independent reviewers screened the titles and abstracts of 2317 records for eligibility. Because eligibility could not be assessed from the titles or abstracts of 28 articles, those articles were assessed using the full text. One poster abstract met our research question and a few articles were related, but no article met all of the inclusion criteria. This lack of scientifically based knowledge concerning the perspective of people with bvFTD is discussed with reference to the search strategy and the research questions, diseaserelated aspects such as changes in behaviour or language and emotions and the difficulties in researching this topic. Recommendations are formulated for future research considering the perspective of people with bvFTD and that will involve the development of appropriate data collection methods. Subsequently, specialised interventions must be developed. Keywords: coping, demands, frontotemporal dementia, needs, scoping review, self-expressions

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Introduction Frontotemporal dementia (FTD) is the clinical term for a pathological, heterogeneous set of neurodegenerative disorders that together are known as frontotemporal lobar degenerations (FTLDs). These disorders impact behaviour and language in various ways and are associated with atrophy of the frontal and temporal lobes (Otto et al. 2011, Warren et al. 2013). Characteristically, the onset of FTD is early (prior to the age of 65) and has a wide range (from the third to the ninth decade) (Johnson et al. 2005). The disease shows a progressive course, with a life expectancy of 1.3–6.5 years after diagnosis (Brodaty et al. 2012). Based on a current review, prevalence rates varied widely, with figures ranging from 2 to 31 people per 100,000 aged between 45 and 64; figures for incidence ranged from 2.7 to 4.1 people per 100,000 younger than 70 years old (Onyike & DiehlSchmid 2013). Knopman and Roberts (2011) have estimated a total number of 20,000–30,000 people with FTD in the entire population of the USA (Knopman & Roberts 2011). There is an overlap between main clinical subtypes of FTLD, classified as behavioural variant frontotemporal dementia (bvFTD), semantic dementia (SD), primary progressive aphasia (PPA) and other atypical syndromes (Otto et al. 2011, Warren et al. 2013). Therefore, differential diagnosis remains difficult, and the terminology used in the literature is often not distinctive and inconsistent (Neary et al. 2005, Brun & Gustafson 2011). In the past, the behavioural variant was often referenced more generically as FTD. This article is focused on people with bvFTD, and we use the term bvFTD if there is a clear reference to this subtype in the papers quoted. We use the more general term FTD if people with the behavioural variant were included in samples but no clear differentiation was made. BvFTD is thought to be the most common subtype of FTLD (Rabinovici & Miller 2010, Onyike & DiehlSchmid 2013). The early stage of the disease is associated less with impairment of memory and more with various strong changes in judgement, behaviour, personality and emotions (Rankin et al. 2005, Piguet & Hodges 2013). The disease tends to affect individuals at a time of life when they have a job and/or a family to attend to (Mendez 2009), and the neuropsychological and functional abilities in people with FTD are more severely affected than in individuals with other types of dementia (Piguet et al. 2011). For people with bvFTD, the co-occurrence of prominent symptoms (e.g. disinhibition, apathy/inertia, loss of 376

sympathy/empathy, perseverative/compulsive behaviours, hyperorality or dysexecutive neuropsychological profiles) (Rascovsky et al. 2011) has a strong and disruptive impact on everyday life and creates specific challenges for families and society. These challenges include financial problems, substantial caregiver burden (Nunnemann et al. 2012) and a decrease in health-related quality of life (Riedijk et al. 2006). Furthermore, health and social care services for people with dementia are primarily designed for older people (e.g. individuals with Alzheimer’s disease), and there is evidence that these services do not fit the needs of people with FTD and their families (Beattie et al. 2004, Nunnemann et al. 2012, Shnall et al. 2013). Currently, no specialised interventions are available for people with FTD (Massimo & Grossman 2008, Mendez 2009, Piguet et al. 2011). There is inconsistent evidence of the benefit of pharmacological interventions for this group, and psycho-social interventions, which are postulated to be the most effective intervention for dementia, have not yet been either developed systematically or studied using people with FTD (Piguet et al. 2011, Warren et al. 2013). Because psycho-social interventions are most effective if they match the specific needs and demands of the persons concerned (Ballard & Corbett 2010), more information about the particular needs, demands and coping strategies of people with bvFTD from their own perspective is needed. Research in this area should particularly investigate whether existing dementia-specific psycho-social interventions are suitable for people with bvFTD, whether they should be adapted or if new FTD-related interventions should be developed.

Methods This review was created as a scoping review following the framework of Arksey and O’Malley (2005) and enhanced procedures (Levac et al. 2010, Daudt et al. 2013). It aims to be the first step towards the development of a psycho-social intervention for people with bvFTD based on the framework of the Medical Research Council for developing complex interventions (Craig et al. 2008). A scoping review offers the opportunity to describe the process of exploring and mapping existing literature without a conventional quality appraisal (Daudt et al. 2013, Armstrong et al. 2011). This presentation focuses on examining the extent, range and nature of research activity and identifying research gaps in the existing literature regarding the © 2015 John Wiley & Sons Ltd

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self-expressed needs and demands of people with bvFTD, which equates to the first and fourth reasons within the framework of Arksey and O’Malley (2005). This framework included five key phases: (i) identifying the research question; (ii) identifying relevant literature; (iii) study selection; (iv) charting the data; and (v) collating, summarising and reporting the results. Consultation of stakeholders is an optional phase and was conducted during the entire process described within the phases. Identifying the research question An interdisciplinary research group with an academic background in nursing science and sociology with content and methodological expertise in person-centred health service research for people with dementia designed the search strategy. Initially, a broad research question was defined: What is known about the needs and demands from the perspective of people with frontotemporal dementia in everyday life? In this stage, national and international websites (Table 1) were consulted frequently since November 2010 to identify research activities and trends to find stakeholders and to become familiar with the research field. An initial literature scan in MEDLINE [PubMed] between November and December 2010 showed

some inconsistencies with the wording and the terminology of FTLD and underlying syndromes. The authors contacted stakeholders, such as physicians, healthcare providers or carers of people with FTD, via e-mail or at national and international FTD conferences to discuss possible strengths and faults of the research questions and the search strategy. The research question was further defined by formulating the following three sub-questions and focusing on the behavioural variant of FTD.

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What are the self-reported needs and demands of people with bvFTD? What are the subjective experiences and perceptions of people with bvFTD? How do people with bvFTD cope with the disease in everyday life?

Moreover, this definition led to the decision to extend the search and to systemise the search strategy in further databases using key terms related to the categories ‘self-expressions’, ‘needs/demands’ and ‘coping’ in combination with key terms related to ‘frontotemporal dementia’ (Table 2). Clarification of concepts The key terms used in the search were chosen according to a systematic review of the subjective experiences of persons with dementia (von Kutzleben et al. 2012) focusing on people with bvFTD. All terms were

Table 1 Consulted websites Link National German websites http://www.deutsche-alzheimer.de/die-krankheit/frontotemporale-demenz.html http://www.ftld.de http://www.knd-demenzen.de/die-verbuende/verbund-ftld/projektbeschreibung.html http://www.wegweiser-demenz.de/frontotemporale-demenz-pick.html http://www.demenz-support.de

Association

German Alzheimer Association German Consortium for FTLD German Network of degenerative dementia, Association of FTLD Federal Ministry for Family Affairs, Senior Citizens, Women and Youth Demenz Support Stuttgart gGmbH

International websites http://www.theaftd.org/ http://www.theaftd.org/life-with-ftd/i-have-ftd/my-experience http://www.ipa-online.org http://www.alz.co.uk/ http://www.alz.org/ http://www.alzheimer-europe.org

International Psychogeriatric Association Alzheimer’s Disease international Alzheimer’s Association Alzheimer Europe

Blogs http://earlydementiasupport.blogspot.de/ http://www.adementiavoice.com http://theFTD/DementiaSupportBlog http://LivingwithDementia http://bvftd.blogspot.de/mypersonalaccountofbvFTD

FTD/Dementia Support Blog A dementia voice A blog by Howard Glick A blog by Bruce Bane A blog by Lee Cerny

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Association of Frontotemporal Degeneration (AFTD)

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Psychological adaptation [MeSH] # OR Self care [MeSH] # OR Sense of self [tiab] # OR Sense of coherence [tiab] # OR Utilization of service [tiab] # OR Coping [tiab] # OR Self efficacy [MeSH] Health services needs and demand [MeSH] # OR Personal satisfaction [MeSH] # OR Patient satisfaction [MeSH] # OR Patient preference [MeSH] # OR Quality of life [MeSH] # OR Personal requirements [tiab] # OR Needs [tiab] # OR Need perception [tiab] # OR Wants [tiab] # OR Care [tiab] # OR Support [tiab] # OR Help [tiab] # OR Preferences [tiab] # OR Demand [tiab] # OR Need assessment [MeSH] Self concept [MeSH] # OR Self disclosure [MeSH] # OR Self assessment [MeSH] # OR Communication [MeSH] # OR Identity crisis [MeSH] # OR Self experience [tiab] # OR Subjective experience [tiab] # OR Personal experience [tiab] # OR Inner experience [tiab] # OR Self perception [tiab] # OR Self handling [tiab] # OR Self report [tiab] # OR Values [tiab] # OR Suffering [tiab] # OR Experiencing illness [tiab] # OR Awareness [tiab] # OR Stressor [tiab] # OR Discoveries about self [tiab] Frontotemporal lobar degeneration [MeSH] # OR Frontotemporal lobar degeneration [tiab] # OR Frontotemporal dementia [MeSH] # OR Frontotemporal dementia [tiab] # OR Pick disease of the brain [MeSH] # OR FTLD [tiab] # OR FTD [tiab] # OR bvFTD [tiab] # OR Frontal lobe dementia [tiab] # OR Early onset dementia [tiab]

3 2 1

Frontotemporal Dementia (1) #AND (Self-expressions (2) # OR Needs/Demands (3) # OR Coping (4))

Table 2 Major search terms and related sub-terms for MEDLINE [PubMed]

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discussed and adapted by the team related to the research questions. The first category of key terms in the list represents the operationalisation of the term ‘self-expression’. ‘Self-expression’ is defined in this review as a verbal, non-verbal or written demonstration/statement by people with FTD themselves, including gestures, facial expressions, narratives, letters or standardised questionnaires, or protocols. The next key terms cover the two concepts ‘needs’ and ‘demands’. The concept of ‘need’ is anchored traditionally in a psychological context but has been adapted and modified for use in economic and health sciences. Within the context of this review, ‘need’ refers to an implicit, communicated or perceived state of deprivation or desire. ‘Demands’, often discussed in relation or in contrast to needs, are self-expressions that directly express or refer to either what is expected or what can be done to fulfil the needs (Naidoo & Wills 2000, van der Roest et al. 2007). The key terms related to ‘coping’ include strategies for dealing with the disease and the utilisation of services by people with bvFTD. Although this review focuses on bvFTD, broader terms such as ‘FTLDs’ or ‘frontal lobe dementia’ were included to compensate for the difficulties of differential diagnosis or inconsistencies in the terminology used by different authors. The sub-term ‘early-onset dementia’ was also included because people with FTD are often subsumed into the group of people with early-onset dementia. Identifying relevant studies The comprehensive search strategy was performed in several literature sources using electronic databases; websites of existing networks, relevant organisations and conferences; and reference lists of relevant literature, in addition to hand-searching procedures (Figure 1). Electronic databases A systematic literature search was conducted between February and March 2012 and was updated between October and November 2014 in seven peer-reviewed databases: MEDLINE [PubMed], CINAHL [EBSCO], PsycINFO, PSYNDEX, SocINDEX, GeroLit, and the Cochrane Library, along with two grey literature databases – ProQuest and the German National Library – using defined key terms. The literature search was limited to German and English publications of human studies published between January 2000 and October 2014. No limitations were made based on type of paper, type of study design, and stage of disease or setting. Each key term category was operationalised using different sub-terms, which were adapted for each © 2015 John Wiley & Sons Ltd

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Records identified through other sources

Records identified through database searching

Identification

Peer reviewed

Grey

Referencelists (7)

MEDLINE [PubMed] (966) CINAHL (173)

German National Library (3)

PsycINFO (890)

ProQuest (224)

Abstract books ICFTD 2008 (82), 2010 (94), 2012 (128), 2014 (122)

PSYNDEX (20)

German Symposia FTD

SocINDEX (15)

Handsearching procedures

2005 (4), 2006 (8), 2009 (6), 2010 (2), 2013 (6)

GeroLit (6) Cochrane Library (112) (n = 23)

(n = 227)

(n = 2182)

Screening

Websites Conferences Stakeholders

(n = 452)

(n = 3)

Records screened based on title and abstract (n = 2317)

Records excluded

Eligibility

(n = 2287)

Poster abstracts (n = 1)

Full-text articles assessed for eligibility (n = 28)

Full-text articles excluded, with reasons

Included

(n = 28) Relevant abstracts related to the research question no full-text available

Studies included in qualitative synthesis or quantitative synthesis

(n = 1)

(n = 0)

Figure 1 Flow diagram of the scoping review.

database or literature source and set forth in a search protocol. The MeSH terms [PubMed] and thesaurus or subject headings in each database (e.g. PsycINFO, CINAHL) were used as much as possible. If attainable, all key terms were searched using the title and abstract function (tiab). Table 2 shows the operationalisations of the key terms applied in the MEDLINE [PubMed] database. The search consisted of two steps. First, an OR relationship was used to identify all papers that contained words related to either ‘self-expressions’ or ‘needs/demands’ or ‘coping’. Then, an AND relationship was used to search these papers for terms related to ‘frontotemporal dementia’ (Table 2). Websites, conference proceedings and other literature sources All available abstract books (2008–2014) of the International Conference on Frontotemporal Dementias (ICFTD) were screened based on their titles and abstracts, and the documentation of the German Symposium on FTD (2005, 2006, 2009, 2010, 2013) was screened based on the full text for relevant research activities using the search function for all key terms. © 2015 John Wiley & Sons Ltd

Selected websites listed in Table 1 were consulted, and relevant research activities and publication links were considered. The search varied in response to the different structures of the website itself. A web-newsletter was installed for the website aftd.org for a continuous update. The reference lists of relevant papers were screened for further papers and added by handsearching articles in Google/Google Scholar. Selection of the literature Based on the key term search strategy and following the dismissal of duplicate papers, two independent reviewers screened the title and abstract of each paper for eligibility according to the following criteria. In cases of disagreement, the decision of a third reviewer was recruited. Using the same procedure, the full texts of all potentially relevant articles were reviewed if it had not been possible to assess these papers from the title or abstract (Figure 1). If the full text of a relevant publication was not available, the corresponding author of the relevant abstract was contacted by e-mail. 379

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Inclusion criteria Publications were included if they focused on both of the following aspects:



The needs, demands or coping strategies of people with bvFTD or people with FTD, if no differentiation of subtype was made; and Self-expressions/self-reports from people with bvFTD or people with FTD, if no differentiation of subtype was made.



Papers that included statements from informal caregivers or professionals were only included if they reported the perspective of people with bvFTD. Exclusion criteria Publications were excluded if they were non-scientific or focused solely on one of the following aspects:

• • • • •

Pathology, pathophysiology and aetiology of FTD Prevalence of FTD Pharmacological interventions for FTD Diagnostic tests and procedures for FTD Healthcare services for family members of people with FTD Needs, demands or coping strategies of relatives of people with FTD or professional caregivers People with types of dementia other than bvFTD or without a specific dementia diagnosis People with early-onset dementia/young-onset dementia without specifying bvFTD.

• • •

Charting the data In this stage of a scoping study, the relevant literature must be sorted and analysed in a descriptive way, and the research team must determine whether the approach to data extraction is consistent with the research question (Levac et al. 2010). Therefore, we planned to extract the relevant data using a worksheet with either a qualitative or a quantitative approach and to screen the study design, sample, setting, data collection/analyses/ethics and key findings of the selected studies based on the research questions. In this step, identified full texts could be sorted in the literature if they: fit the research question exactly (self-reported needs/demands/coping of people with bvFTD); or were closely related to the focus on selfexpressions of people with bvFTD, but

1 2

• included other people with (early-onset) dementia in addition to bvFTD • did not address their needs and demands.

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Collating, summarising and reporting the results As a result of the comprehensive key term search strategy and after removing duplicates, 2317 papers were identified as relevant in the electronic databases, the reference lists, the abstract books, and the web sources database. All titles and abstracts were screened for eligibility. A further 28 articles could not be screened using only the title or abstract, and these papers were assessed for eligibility based on their full texts. The reviewers’ screening showed that none of these articles met both inclusion criteria. 1 We only found one poster abstract within the abstract books of the international conference on FTD, which was identified as relevant for the review, but the full study were not published in a journal until recently. The poster was titled ‘The subjective experiences of people with a diagnosis of behavioural variant FTD: Living in an alien world’ (Griffin et al. 2012). The aim of the study sought to elicit the views of people with bvFTD on their experiences of the illness. They used interpretative phenomenological analysis of in-depth interviews with five people with bvFTD living at home. Four themes were identified: ‘What’s the problem? Relationships, Threats to Identity and Coping’. Participants could describe their diagnosis without an emotional response or a problematic connotation. They experienced relationships as unchanged but noticed alterations in behaviour towards them. Restrictions and other consequences of the illness, such as loss of employment, were felt to be unfounded and interpreted as an attack. Withdrawing, blaming others and externalisation were described as coping strategies to protect aspects of self-identity. The authors summarised that the subjective experiences of the interviewed persons were congruent to the deficits of people with bvFTD named under Theory of Mind tasks, but the first-hand accounts could enable professionals to address affected individuals with greater empathy (Griffin et al. 2012). More detailed information could not be received by contacting the authors. 2 Two other research projects identified in the updated search of electronic database using videobased observation (Barsuglia et al. 2014, Mendez et al. 2014) were closely related to our question but did not fit exactly because of the focus on social behaviour instead of needs. In three articles, people with FTD were included in the survey of the needs of people with early-onset dementia, and interview reports were explicitly cited with reference to them (Harris 2002, Alzheimer’s Aus© 2015 John Wiley & Sons Ltd

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tralia 2007, Armari et al. 2013). The researchers let them speak for themselves; although the sample included people with FTD, the results were not broken down by types of dementia. Interviews with stakeholders revealed that there is one ongoing PhD project based on the family perspective, in which people with FTD have been involved in a current study at the University of Birmingham, UK. This project was also found in the actual conference paper of the ICFTD 2014 (LaFontaine et al. 2014), but the study results have not yet been published. To date, the existing literature on the perspectives of people with bvFTD towards their own situation is non-scientific and only available in blog statements (e.g. early dementia support.blogspot.de) and one autobiographical report (Mobley 2011). These sources would have been evaluated by a secondary analysis, but such an assessment was not an aim of this review. In summary, one poster abstract was identified that met both inclusion criteria. Currently, nothing has been published on the self-reported needs, demands or coping strategies of people with bvFTD.

Discussion Only one poster abstract (Griffin et al. 2012), mentioned above, met all inclusion criteria, and a few studies were related to the focus of this review but had a different research question, e.g. social behaviour instead of needs and demands (Barsuglia et al. 2014, Mendez et al. 2014) or subsumed people with FTD in the group of early-onset dementia (Harris 2002, Alzheimer’s Australia 2007, Armari et al. 2013). There are two important results in our research: 1 These studies demonstrate that it is possible and sensible to analyse the self-expressions of people with bvFTD. 2 Analysis of the perspectives of people with FTD revealed completely new knowledge. For example, Griffin et al. (2012) found that people with FTD experience their environment as strange and assess the reactions they cause as inadequate and incomprehensible. It is obvious that there would be more results to discuss, but unfortunately, the publication by Griffin et al. (2012) is not yet available in a full-text article. In response to this striking result, it may be argued that the research questions were inadequate or the chosen search strategy was not appropriate. If not, there is an unexpected large research gap, and it © 2015 John Wiley & Sons Ltd

is necessary to discuss the reasons underlying the identified gap. In the following section, we will discuss this issue, the possible limitations of this review according to the ‘research question’ and ‘search strategy’, ‘disease-related aspects’ and note some ‘future perspectives’. Research questions The research questions comprised four essential components: people with bvFTD, self-expressions, needs/ demands and coping. Our focus on people with bvFTD reflects their current situations. The early disease onset and strong as well as radical changes in behaviour cause numerous problems for the people affected and their social environment. More than half of those with FTD are cared for at home, primarily by close relatives, but little is known about the challenges of coping with everyday life, and the related distress and burden appear to be enormous (Diehl-Schmid et al. 2012, Nunnemann et al. 2012, Oyebode et al. 2013). Research needs to focus on the expressed needs of those with FTD to learn how to provide suitable support (van der Roest et al. 2007). This approach is particularly important based on the finding that there is often incongruity in perspectives between people with dementia and their relatives, particularly regarding interpersonal relationships (von Kutzleben & Schmid 2012). To ensure that the self-expressions of people with dementia are heard, it has become an important and increasingly prevalent approach to involve people with dementia themselves in research activities. The needs and demands from the perspective of healthcare receivers themselves must be listened to if effective and appropriate programmes for social interventions and healthcare services are to be provided (Wilkinson 2002). This involvement has become a typical approach for data collection in many research fields. During the last decade, an increasing number of articles have focused on the subjective experiences of people with dementia; accordingly, people with dementia have been invited to scientific or political conferences and meetings specifically to share their perspectives in public. Therefore, people with dementia are able to express their needs, particularly in the early stage of their disease (Beattie et al. 2004, von Kutzleben et al. 2012). Initially, there does not appear to be any reason to treat people with FTD differently from other patient groups in collecting information on their needs and demands. Rather, the opposite phenomenon appears to be true: Because people with bvFTD have little or no insight into their symptoms, they may see no 381

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problems with their behaviour, particularly during the early stages of dementia. However, others may have a quite different view of the person’s behaviour and may consider it as being highly problematic and even embarrassing. If we want to uncover the various interests and needs of people with bvFTD, we must learn to understand those factors that are specific to them. Further research into the views and experiences of people with different types of dementia would enhance our knowledge of how to provide services for those with less common forms of the condition. The research projects mentioned above (Harris 2002, Alzheimer’s Australia 2007, Griffin et al. 2012, Armari et al. 2013, Barsuglia et al. 2014, Mendez et al. 2014) demonstrate that it is possible to analyse self-expressions from people with FTD. A further aspect must be considered in this discussion. Do the terms needs, demands and coping, which were used in the research question, target the primary issue? Upon initial examination, these concepts may appear to be too narrow. People with dementia do not always judge their situations in terms of problems. The expression of needs correlates with aspects of wellbeing or quality of life (van der Roest et al. 2007). The search terms (Table 2) were quite broad in this study and included concepts such as personal satisfaction, patient preference and quality of life or care, which together cover a wide spectrum. Some needs and demands were proposed that were different from those of people with late-onset dementia (Beattie et al. 2004). It is not known whether people with bvFTD have the same or similar requirements as those with other types of dementia. An examination of how these individuals address their specific disease-associated restraints during daily life could provide an important source of information on their needs and demands and possibilities for treatment options. Communication with people with FTD may be difficult, and their social behaviour may be irritating to or challenging for others in their environment. Current research suggests that behaviours that are experienced as challenging by carers may be meaningful activities from the perspective of the person with dementia or expressions of unmet needs (Penrod et al. 2007). However, there are reasons to believe that those unmet needs differ according to the type of dementia (Koehn et al. 2011). Therefore, it is necessary to determine whether this assumption is transferable to people with bvFTD. Based on the mentioned studies about the social behaviour of people with FTD (Barsuglia et al. 2014, Mendez et al. 2014), a secondary analysis could determine whether unmet needs could be the trigger for 382

this behaviour. Knowledge of the needs and demands of people with bvFTD is indispensable for developing and providing effective and tailored support. Search strategy With respect to the search strategy, the search terms may have been too precise or perhaps not sufficiently precise. To examine each possibility, the fit of the search terms and sub-terms was examined relative to the underlying theoretical concepts (defined above). The results indicated that the search terms, the strategy employed and the databases searched were precise and exhaustive enough to discover relevant research publications. The term ‘FTD’ does not appear as a keyword before 2000. For example, in MEDLINE, none of the central terms, such as frontotemporal dementia (2010), frontotemporal lobar degeneration (2010) or Picks disease (2000), were used as MeSH terms prior to 2000. Therefore, if the beginning of FTD-focused research did not occur earlier, there may not be many actual results about self-expressions or even psychosocial interventions. This review specifically examined the needs of people with bvFTD. To ensure that the search was as precise as possible, all papers with generic samples of ‘dementia’ were excluded. This procedure risks excluding research projects in which people with bvFTD were involved but not specifically named, particularly in studies with a focus on early-onset dementia. A possible step forward might be a secondary analysis of the data from those studies in which people with FTD were involved but not specifically mentioned within the research results. This work would be worthwhile if it proved possible to extract specific findings from people with FTD. Furthermore, limiting the survey to English and German literature may have led to some publications being ignored. In this context, excluding non-scientific literature, particularly autobiographical texts, such as statements posted in Internet blogs, is another limitation of this review. These decisions were made because we wanted to identify scientific research publications to point out the current state of research. The inclusion of non-scientific literature and secondary analyses of research into early-onset dementia should be considered in future research focusing on the needs of people with FTD. The final question is whether the search should have been extended to additional databases. However, most of the articles that seemed to be relevant and were assessed using the full text appeared in two databases, specifically MEDLINE and Psy© 2015 John Wiley & Sons Ltd

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cINFO. The search began using MEDLINE and was then supplemented and enhanced with PsycINFO, retrieving many duplicates. Further searches (Figure 1) retrieved very few articles, which, interestingly, were not identical to those found through MEDLINE and PsycINFO. These findings suggest that this theme is currently very specific and marginal and that it is seldom studied and – unusually – is published in databases that do not belong to the main databases for medical fields. Disease-related aspects Because the research question and strategy were sufficiently precise and adequate, a different explanation for the research gap must be found. The most obvious reason for the remarkable results of this review may be found in the manifestations of bvFTD, including irritating and striking changes in behaviour, loss of insight in addition to alterations in the perception of the environment and social behaviour. These symptoms make it difficult to include people with bvFTD in studies. First, reduced empathy and executive problems, such as rigidity of thinking, represent core criteria and also come across readily to professionals. Therefore, researchers may conclude that people with bvFTD do not have the ability to communicate feelings and thoughts regarding their needs and demands. Furthermore, people with bvFTD mostly do not consider themselves as being affected by their diagnosis. Therefore, they might not respond to any invitation to contribute to a study with a focus on FTD and would therefore not participate. This assumption must also be examined. In addition to the often late diagnosis, there may be rapid progression of the disease, which limits research possibilities that rely on direct verbal communication, and longitudinal studies would be difficult to achieve. Furthermore, the eligible population is small, and the difficulties of precisely diagnosing the disease complicate identifying appropriate participants. Undoubtedly, access to this field is particularly difficult. Simultaneously, the burden on informal caregivers is obvious, as they not only must find ways of responding to behaviours from their relatives with FTD that they find challenging and must manage the serious problems of everyday life but they must also cope with their relatives’ changes in personality (Nunnemann et al. 2012). The perspective of the caregivers is much easier to investigate; therefore, their dominant position in current research reports is unsurprising. © 2015 John Wiley & Sons Ltd

Future perspectives The difficulties we have described cause enormous challenges for collecting and analysing data, and serious issues concerning research methods must be solved. Researchers have to consider whether established methods of data collection and data analysis must be adapted and modified, when studying everyday life from the perspective of those with changed emotions, perceptions and ways of communicating. One such approach may be using observations rather than questioning and using flexible study designs with sufficiently open approaches. Although, there appears to be no alternative to using an open and flexible research design, some considerations must be raised on the theoretical assumptions of research on the subjective experience, when applying to people with FTD. Often used premises (Flick 2014) are, for example:

• • • •

Social reality is understood as a process of collective construction and assignment that has a process-related character and is reflexive. ‘Objective’ framework conditions become relevant via subjective significance for the world in which one lives. The communicative character of social reality enables the construction and re-construction of social reality and therefore becomes the starting point for research. And the person investigated interprets his/her reality relative to and adapts it to the interpretations of his/her culture.

Within the meaning of interpretative sociology, generating ‘understanding’ and ‘meaning’ is typically essential for the applicability and validity of qualitative research methods. Whether research methods based on these fundamental theoretical postulates are applicable for people with FTD remains a key interest for discussion. An approach that could generate maximum understanding and at best a shared reality would be to use a combination of various research methods and focus on the individual interpretation of the ‘here and now’ of a person’s situation.

Conclusions This review shows that the self-reported needs and demands of people with bvFTD appear to be a marginal issue in the scientific community. This finding reveals an obvious gap in perceiving and addressing the needs of the individuals involved and their need for specific support.

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If the quality of life of people with FTD is to be improved, it is necessary to understand the influence of this disease on their everyday lives. Interventions should be oriented towards and respond to the needs and demands of the persons concerned and their caregivers. Current knowledge of this illness and its impact on everyday life is notably deficient. Therefore, currently, it is not possible to examine whether existing services and structures meet the needs and requirements of those affected or whether new services or structures must be developed. Therefore, research is required to determine the actual needs and demands of people with FTD. This research is not feasible if there is no understanding of what people with bvFTD ‘say’, regardless of how they express their needs. As an alternative to interviews based on verbal communication, observational methods might be used to draw conclusions about underlying needs. Despite the challenges, information on the subjective views of people with bvFTD needs to be gathered, and a reliable method must be ascertained for doing so. To be successful and creative, tailored approaches must be developed and/or employed to extend the portfolio of methods beyond more traditionally used practices of data collection and analysis. Developing these concepts may mean choosing methods with a wide and open approach, such as ethnography (Hirschauer 2001), and combining methods such as videography (Knoblauch & Schnettler 2009) and interviews.

Source of funding None.

Description of authors’ roles CD, SN and DH conceptualised and wrote the manuscript. CD, SN, DH and MZ conducted the literature search of the scoping review, screened the articles independently based on title and abstract and full text. MH developed the research question, study design and supervised the project. The final version was reviewed by all authors (CD, SN, DH, MZ and MH).

Conflicts of interest None.

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What is known about the subjective needs of people with behavioural variant frontotemporal dementia? A scoping review.

Behavioural variant frontotemporal dementia (bvFTD) is an early-onset and progressive neurodegenerative disease associated with strong changes in judg...
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