Maud Hevink Moving Forward with Dementia An exploration of dementia diagnosis and support
Moving Forward with Dementia An Exploration of Dementia Diagnosis and Support Maud Simone Hevink
The research described in this thesis was performed at the Department of Psychiatry and Neuropsychology, Mental Health and Neuroscience Research Institute, Maastricht University, Alzheimer Center Limburg, Maastricht, the Netherlands. Printing of this thesis was kindly supported by Alzheimer Netherlands. ISBN: 978-94-6506-577-9 Printing: Ridderprint. www.ridderprint.nl Cover design: Yente ter Woerds Lay-out & design: Ridderprint. www.ridderprint.nl © Maud Hevink, Maastricht 2024 All rights reserved. No part of this book may be reproduced in any form or by any means without the prior written permission of the first author, or where appropriate from the publishers of the publication.
Moving Forward with Dementia An Exploration of Dementia Diagnosis and Support PROEFSCHRIFT voor het behalen van de graad van Doctor aan de Universiteit Maastricht, onder gezag van Rector Magnificus, Prof. dr. Pamela Habibović, overeenkomstig met het besluit van het College van Decanen, te verdedigen in het openbaar op donderdag 21 november 2024 om 16:00 uur door Maud Simone Hevink
Promotores: Prof. dr. F.R.J. Verhey, Universiteit Maastricht Prof. dr. M.E. de Vugt, Universiteit Maastricht Copromotor: Dr. C.A.G. Wolfs, Universiteit Maastricht Beoordelingscommissie: Prof. dr. J.P.H. Hamers (voorzitter, Universiteit Maastricht) Prof. dr. S.P.J. van Alphen (Vrije Universiteit Brussel) Dr. ir. S.R. de Bruin (Hogeschool Windesheim) Dr. A. Duits, (Universiteit Maastricht) Prof. dr. J.W.M. Muris, (Universiteit Maastricht)
Table of Contents Chapter 1 General Introduction 7 Chapter 2 Understanding Patients’ and Significant Others’ Preferences on Starting a Diagnostic Trajectory for Dementia: An Integrative Review 15 Chapter 3 The Journey of Dementia Diagnosis to Post-Diagnostic Support: A Mixed Methods Study of Healthcare Providers 47 Chapter 4 Experiences of People with Dementia and Informal Caregivers with Post-Diagnostic Support: Data from the International COGNISANCE Study 75 Chapter 5 The Utility of Researcher-Driven Projective and Enabling Techniques to Support Engagement in Research about Dementia Diagnosis and Post-Diagnostic Support 105 Chapter 6 Moving Forward with Dementia: An Explorative Cross-country Qualitative Study into Post-diagnostic Experiences 147 Chapter 7 I Just Happened to Be Lucky: A Qualitative Study Exploring Perceptions of Carer Burden and Social Care for Caregivers of People Living with Dementia in the UK 177 Chapter 8 General Discussion 201 Addendum Summary 224 Nederlandse Samenvatting 226 Impact Paragraph 228 List of Publications 232 Thesis Defence from MHeNS 234 Dankwoord 235 Over de Auteur 240
General Introduction 1
| 8 An introduction to dementia With the ageing population worldwide, the number of people living with dementia is expected to rise to 115 million people by 2050 worldwide (1). Dementia is characterized by a decline in cognitive functioning over time, examples include memory problems and behavioural and psychological symptoms such as depression (2, 3). There are many types of dementia, with Alzheimer’s Disease being the most prevalent type of dementia (4). Receiving a dementia diagnosis is important as it is often a prerequisite for accessing support and enabling people to live well. However, there are several barriers for people with dementia-related symptoms to seek help such as stigma, fear and lack of knowledge (5, 6). In addition, the timing of seeking and receiving a diagnosis along the dementia continuum is important as it can affect the opportunities for implementing support and management for dementia-related symptoms (7). To receive a dementia diagnosis is often a long and distressing process, that may in some cases take up to four years (8). Receiving a diagnosis of dementia is often a turning point for people being diagnosed and their loved ones (9, 10). A dementia diagnosis can be beneficial as knowing a diagnosis can enable people to plan for the future and obtain support (11). However, a dementia diagnosis can also have negative emotional effects; feelings of loneliness, social isolation and perceived threats to their agency and identity have been reported (12-14). For informal caregivers, it is often the start of a ‘long road of challenges’ (15). To help people live well with dementia, they often use various types of support throughout their dementia trajectory, with their need for support increasing as time progresses (16). Much of the support people with dementia need is provided by informal caregivers. The intensity of informal care increases over time as dementia progresses (17, 18). Adequate informal care has the potential to delay nursing home admission and sustain the quality of life of someone with dementia. However, providing informal care can negatively impact their own quality of life and mental and physical health (19-21). Caregivers frequently experience caregiver burden (22). Supporting informal caregivers can be beneficial as it will allow them to maintain their wellbeing whilst providing optimal support for those living with dementia (23). People with dementia and their informal caregivers face complex challenges that often require additional formal support to help them live well with
| 9 1 dementia. Post-diagnostic support services can address a variety of needs for people living with dementia and informal caregivers, and they should focus on understanding dementia, supporting psychological and emotional wellbeing as well as providing practical support (24). While post-diagnostic support can be beneficial as it can facilitate independence, positively impact quality of life and reduce depression (25-27), accessing this support in a timely and tailored manner remains a significant challenge. After their diagnosis, people with dementia often experience a lack of signposting to support services and informal caregivers struggle to access post-diagnostic support as healthcare systems are complex to navigate (28, 29). Only 50% of informal caregivers of people recently diagnosed with dementia reported they received sufficient information on dementia and support options (30). It has been demonstrated that the current state of post-diagnostic support is often insufficient to address the needs of people with dementia and informal caregivers. Accessing and utilizing post-diagnostic support is a complex process that involves disease-related factors, attitudes towards support and healthcare systems (31). Healthcare professionals play a key role in offering and ensuring the quality of post-diagnostic support for people living with dementia and informal caregivers (32). For example, healthcare professionals’ ability to provide person-centred dementia care is often impacted by a lack of dementia-specific knowledge (33). The role of healthcare professionals in enhancing the experience of post-diagnostic support is widely acknowledged, underpinning their central role in this process (34, 35). Unsurprisingly, postdiagnostic support services may not be able to fulfil all the needs of people with dementia and informal caregivers as unmet needs have been reported even after they have utilized post-diagnostic support services (36-38). Aims and outline of the thesis This thesis aims to increase our understanding of experiences from prediagnosis to post-diagnostic dementia support. To this end, several research methods are used to integrate the perspectives of people living with dementia, informal caregivers and healthcare professionals. Chapters 2 to 7 in this thesis aim to answer the following research questions; What are the preferences of people with memory complaints and their significant others regarding starting a diagnostic trajectory for dementia?
| 10 Chapter 2 undertakes an integrative literature review to explore preferences around starting a diagnostic trajectory for dementia. How can healthcare providers contribute to bridging the gap between the diagnostic phase and the post-diagnostic support phase? Chapter 3 utilizes a mixed approach that utilizes survey and interview data to gain a comprehensive understanding of how healthcare providers can support people from diagnosis to support. How do people with dementia and informal caregivers experience support and what factors are important in support satisfaction? Chapter 4 explores satisfaction with support through survey data of people with dementia and informal caregivers. How can qualitative methods support the engagement and participation of people with dementia? Chapter 5 describes and evaluates the development of a qualitative method called ‘projective techniques’. How do people with dementia and informal caregivers experience receiving a dementia diagnosis and subsequent support in Australia, Canada, the Netherlands and Poland. Chapter 6 compares and contrast experiences of a diagnosis and support across country through qualitative data. What is the relationship between informal caregiver burden and the use of social care services for a person with dementia? Chapter 7 explores the role of social care in caregiver burden through interviews with informal caregivers and social care providers.
| 11 1 References 1. Guerchet M, Prince M, Prina M. Numbers of people with dementia worldwide: An update to the estimates in the World Alzheimer Report 2015. 2020. 2. WHO. Factsheet Dementia 2023 [updated 15-03-2023. Available from: https://www.who.int/ news-room/fact-sheets/detail/dementia. 3. Cerejeira J, Lagarto L, Mukaetova-Ladinska E. Behavioral and Psychological Symptoms of Dementia.10.3389/fneur.2012.00073 Frontiers in Neurology. 2012;3. 4. Cao Q, Tan C-C, Xu W, Hu H, Cao X-P, Dong Q, et al. The Prevalence of Dementia: A Systematic Review and Meta-Analysis.10.3233/JAD-191092 Journal of Alzheimer’s Disease. 2020;73:1157-66. 5. Parker M, Barlow S, Hoe J, Aitken L. Persistent barriers and facilitators to seeking help for a dementia diagnosis: a systematic review of 30 years of the perspectives of carers and people with dementia.10.1017/S1041610219002229 International Psychogeriatrics. 2020;32(5):611-34. 6. Ng CKM, Leung DKY, Cai X, Wong GHY. Perceived Help-Seeking Difficulty, Barriers, Delay, and Burden in Carers of People with Suspected DementiaInternational Journal of Environmental Research and Public Health. 2021;18(6):2956. 7. Dubois B, Padovani A, Scheltens P, Rossi A, Dell’Agnello G. Timely Diagnosis for Alzheimer’s Disease: A Literature Review on Benefits and Challenges.10.3233/JAD-150692 Journal of Alzheimer’s Disease. 2016;49:617-31. 8. Bakker C, Koopmans RTCM, Pijnenburg YAL, Verhey FRJ, Vernooij-Dassen MJFJ, de Vugt ME, et al. Time to diagnosis in young-onset dementia as compared with late-onset dementia.10.1017/ S0033291712001122 Psychological Medicine. 2013;43(2):423-32. 9. Robinson L, Clare L, Evans K. Making sense of dementia and adjusting to loss: Psychological reactions to a diagnosis of dementia in couples.10.1080/13607860500114555 Aging & Mental Health. 2005;9(4):337-47. 10. Pesonen H-M, Remes AM, Isola A. Diagnosis of dementia as a turning point among Finnish families: A qualitative study.https://doi.org/10.1111/nhs.12059 Nursing & Health Sciences. 2013;15(4):489-96. 11. Moll van Charante E, van Hout H, van Kuijk L, van Marwijk H, van den Dungen P, van der Horst H, et al. Preferences regarding disclosure of a diagnosis of dementia: a systematic review.10.1017/ S1041610214000969 International Psychogeriatrics. 2014;26(10):1603-18. 12. Xanthopoulou P, McCabe R. Subjective experiences of cognitive decline and receiving a diagnosis of dementia: qualitative interviews with people recently diagnosed in memory clinics in the UK.10.1136/bmjopen-2018-026071 BMJ open. 2019;9(8):e026071-e. 13. Campbell S, Manthorpe J, Samsi K, Abley C, Robinson L, Watts S, et al. Living with uncertainty: Mapping the transition from pre-diagnosis to a diagnosis of dementia.https://doi.org/10.1016/j. jaging.2016.03.001 Journal of Aging Studies. 2016;37:40-7. 14. Aminzadeh F, Byszewski A, Molnar FJ, Eisner M. Emotional impact of dementia diagnosis: Exploring persons with dementia and caregivers’ perspectives.10.1080/13607860600963695 Aging & Mental Health. 2007;11(3):281-90. 15. Chen M-C, Lin H-R. The experiences of family caregivers in response to a dementia diagnosis disclosure.10.1186/s12888-022-04126-4 BMC Psychiatry. 2022;22(1):475. 16. Bökberg C, Ahlström G, Karlsson S. Utilisation of formal and informal care and services at home among persons with dementia: a cross-sectional study.https://doi.org/10.1111/scs.12515 Scandinavian Journal of Caring Sciences. 2018;32(2):843-51. 17. Engel L, Ajdukovic M, Bucholc J, McCaffrey N. Valuation of Informal Care Provided to People Living With Dementia: A Systematic Literature Review.https://doi.org/10.1016/j.jval.2021.04.1283 Value in Health. 2021;24(12):1863-70. 18. Wimo A, Gauthier S, Prince M. Global estimates of informal care. London: Alzheimer’s Disease International (ADI),; 2018 July 2018. 19. Bauer JM, Sousa-Poza A. Impacts of Informal Caregiving on Caregiver Employment, Health, and Family.10.1007/s12062-015-9116-0 Journal of Population Ageing. 2015;8(3):113-45.
| 12 20. Di Lorito C, Bosco A, Godfrey M, Dunlop M, Lock J, Pollock K, et al. Mixed-Methods Study on Caregiver Strain, Quality of Life, and Perceived Health.10.3233/JAD-201257 Journal of Alzheimer’s Disease. 2021;80:799-811. 21. Frias CE, Cabrera E, Zabalegui A. Informal Caregivers’ Roles in Dementia: The Impact on Their Quality of LifeLife. 2020;10(11):251. 22. Connors MH, Seeher K, Teixeira-Pinto A, Woodward M, Ames D, Brodaty H. Dementia and caregiver burden: A three-year longitudinal study.10.1002/gps.5244 International Journal of Geriatric Psychiatry. 2020;35(2):250-8. 23. Walter E, Pinquart M. How Effective Are Dementia Caregiver Interventions? An Updated Comprehensive Meta-Analysis.10.1093/geront/gnz118 The Gerontologist. 2019;60(8):e609-e19. 24. Bamford C, Wheatley A, Brunskill G, Booi L, Allan L, Banerjee S, et al. Key components of postdiagnostic support for people with dementia and their carers: A qualitative study.10.1371/journal. pone.0260506 PLoS One. 2021;16(12):e0260506. 25. Kelly F, Innes A. Facilitating independence: The benefits of a post-diagnostic support project for people with dementia.10.1177/1471301214520780 Dementia. 2016;15(2):162-80. 26. Ames D, Blazer D, Brodaty H, Cooper C, Engedal K, Katona C, et al. Systematic review of the effectiveness of non-pharmacological interventions to improve quality of life of people with dementia.10.1017/S1041610211002614 International Psychogeriatrics. 2012;24(6):856-70. 27. Leung P, Orrell M, Orgeta V. Social support group interventions in people with dementia and mild cognitive impairment: a systematic review of the literature.https://doi.org/10.1002/gps.4166 International Journal of Geriatric Psychiatry. 2015;30(1):1-9. 28. Peel E, Harding R. ‘It’s a huge maze, the system, it’s a terrible maze’: Dementia carers’ constructions of navigating health and social care services.10.1177/1471301213480514 Dementia. 2014;13(5):642-61. 29. Hagan RJ. What next? Experiences of social support and signposting after a diagnosis of dementia.10.1111/hsc.12949 Health Soc Care Community. 2020. 30. Nickel F, Kolominsky-Rabas PL. PMH37 - Pathways to Dementia Diagnosis and Post-Diagnostic Support: the Bavarian Dementia Survey.https://doi.org/10.1016/j.jval.2017.08.1902 Value in Health. 2017;20(9):A715. 31. Kerpershoek L, Wolfs C, Verhey F, Jelley H, Woods B, Bieber A, et al. Optimizing access to and use of formal dementia care: Qualitative findings from the European Actifcare study.10.1111/hsc.12804 Health & Social Care in the Community. 2019;27(5):e814-e23. 32. Frank C, Forbes RF. A patient’s experience in dementia care: Using the “lived experience” to improve careCan Fam Physician. 2017;63(1):22-6. 33. Monthaisong D. Nurses’ experiences providing care for people with dementia: an integrative literature reviewJournal of Nursing and Care. 2018;7(2):1-6. 34. Bailey C, Dooley J, McCabe R. Communication in healthcare interactions in dementia: a systematic review of observational studies.10.1017/S1041610214002890 International Psychogeriatrics. 2015;27(8):1277-300. 35. Shafir A, Ritchie CS, Garrett SB, Bernstein Sideman A, Naasan G, Merrilees J, et al. “Captive by the Uncertainty”—Experiences with Anticipatory Guidance for People Living with Dementia and Their Caregivers at a Specialty Dementia Clinic.10.3233/JAD-215203 Journal of Alzheimer’s Disease. 2022;86:787-800. 36. Jennings LA, Reuben DB, Evertson LC, Serrano KS, Ercoli L, Grill J, et al. Unmet Needs of Caregivers of Individuals Referred to a Dementia Care Program.https://doi.org/10.1111/jgs.13251 Journal of the American Geriatrics Society. 2015;63(2):282-9. 37. Black BS, Johnston D, Rabins PV, Morrison A, Lyketsos C, Samus QM. Unmet Needs of CommunityResiding Persons with Dementia and Their Informal Caregivers: Findings from the Maximizing Independence at Home Study.https://doi.org/10.1111/jgs.12549 Journal of the American Geriatrics Society. 2013;61(12):2087-95. 38. Amjad H, Black BS, Davis K, Johnston D, Kelly J, Leoutsakos J, et al. Unmet needs in community-living persons with dementia are common, often non-medical and related to patient and caregiver characteristics.10.1017/S1041610218002296 International Psychogeriatrics. 2019;31(11):1643-54.
| 13 1
Understanding Patients’ and Significant Others’ Preferences on Starting a Diagnostic Trajectory for Dementia: An Integrative Review Iris Linden, Maud Hevink, Claire Wolfs, Marieke Perry, Carmen Dirksen, Rudolf Ponds Aging & Mental health, 2022 2
| 16 Abstract Objective To explore the preferences of people with memory complaints (PwMC) and their significant others regarding starting a diagnostic trajectory for dementia. Methods A systematic literature search was conducted in PubMed, PsycINFO, CINAHL, Web of Science, and Embase. Selection of abstracts and papers was performed independently by two researchers. Methodological quality was assessed with the Mixed Method Appraisal Tool. Result sections of the selected papers were thematically synthesized. Results From 2497 citations, seven qualitative studies and two mixed methods studies published between 2010 and 2020 were included. Overall quality of the studies was high to moderate. A thematic synthesis showed that preferences for starting a diagnostic trajectory arose from the feeling of needing to do something about the symptoms, beliefs on the necessity and expected outcomes of starting a diagnostic trajectory. These views were influenced by normalization or validation of symptoms, the support or wishes of the social network, interactions with health care professionals, the health status of the PwMC, and societal factors such as stigma and socioeconomic status. Conclusion A variety of considerations with regard to decision-making on starting a diagnostic trajectory for dementia were identified. This emphasizes the need to explore individual preferences to facilitate a timely dementia diagnosis.
2 | 17 Introduction The number of people with dementia in western countries is expected to increase dramatically (1). As a consequence of increased public awareness around dementia, more older people become worried about their memory or the possibility of having dementia and ask for cognitive assessment by a specialist (2, 3). At the same time, dementia is still underdiagnosed in a lot of countries and when people seek help it often occurs in a late stage of the disease when activities in daily living are already heavily impacted (4). Although the high rates of under diagnoses can partly be explained by difficulties in accessing care and the complexity of healthcare systems (5, 6), this paradox reflects the difficulty of the decision to start a diagnostic process for patients and significant others (SO). The decision to start diagnostic testing for dementia is considered preferencebased (7, 8). In the absence of curative medicine, the advantages and disadvantages of diagnostic process are, presumably, valued differently by each individual. Advantages of (early) diagnosis of dementia include: enabling patients and their SOs to plan their future and care (9), delaying the disease progression with future effective interventions (10, 11), and providing time for the person with dementia to decide on future financial, legal and medical issues while they still have mental capacity (11, 12). Disadvantages of (early) diagnosis of dementia include: fear or worries about the future due to an absence of curative treatment, possible discrimination or stigmatization, and the risk of misdiagnosis in an early stage of dementia (13, 14). Discussing advantages and disadvantages of dementia diagnosis with people with memory complaints (PwMC) and SOs facilitates a timely diagnosis. Timely diagnosis means that a diagnostic process is initiated at the right moment in time for the PwMC and their SO (i.e. the moment in time that they perceive they can benefit most from a diagnosis) (15). To explore the ‘timeliness’ of a diagnostic trajectory, preferences of PwMCs and their SOs should be considered by healthcare professionals (HCP) before the onset of the diagnostic process (15-17) in a process of shared decision making (SDM). SDM assumes that decisions should be influenced by exploring and respecting “what matters most” to patients and that this exploration in turn depends on patients developing informed preferences (18). The general practitioner (GP) is often the first HCP a PwMC visits to seek medical help (9). Therefore, GPs are
| 18 the most obvious HCPs to explore these preferences as recommended in the Dementia Guideline of the Dutch College of General Practitioners (19). Although GPs tend to value the “rightness” of time for starting a dementia diagnostic trajectory, they also consider this a complex issue (17). They experience barriers such as lack of time, confidence and knowledge or are held back due to their own attitudes towards early diagnosis, stigma or therapeutic nihilism (20-22). Insights in patient preferences and considerations concerning starting a diagnostic trajectory for dementia could support GPs to initiate the SDM process about starting a diagnostic trajectory, and it could assist in optimizing clinical guidelines (23). Previous reviews have touched upon patient preferences around dementia diagnoses, specifically on systematic population screening (24), barriers towards help seeking (25) and diagnostic disclosure (12). All have concluded that preferences around diagnosing dementia are complex and multi-factorial. They, however, do not address considerations and preferences for starting a diagnostic trajectory in case of actual memory problems. Therefore, an integrative review is conducted to explore and map the preferences and considerations of PwMCs and their SOs regarding starting a diagnostic trajectory for dementia. Method This review was registered in the International Prospective Register of Systematic Reviews (PROSPERO) (registration number: CRD42020190580). The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines were followed for article selection (26). Design An integrative review was conducted in line with the methodology described by Whittemore and Knafl (2005) (27). This methodology allows for the inclusion of quantitative, qualitative, and mixed-method studies. By including studies with different methodologies, all aspects of patients’ preferences could be integrated to get an overall understanding of preferences in the context of a timely dementia diagnosis.
2 | 19 Search strategies Papers were searched for in the PubMed, PsychInfo, Web of Science, Embase, and CINAHL databases. The search strategy included synonyms of the following concepts: ‘timely diagnosis’, ‘dementia’, ‘preferences,’ and ‘population’. MeSH terms, free text words, and equivalent index terms were used. The search was limited to English and Dutch language peer-reviewed journals published from January 2010 onwards. The time restriction was set to capture the most relevant considerations and preferences given the rapid developments in dementia diagnostics such as the possibility for biomarker-based diagnosis (28). Additionally, references of included studies were hand-searched. To optimize the search sensitivity and in line with previous studies that examine healthrelated preferences (29) and systematic search strategies for the construct preferences (30, 31), a broad definition of ‘preferences’ was used to determine eligibility for inclusion: “Patient perspectives, beliefs, expectations, goals, and the processes that individuals use in considering the potential benefits, harms, costs, and inconveniences of options in relation to each other” (32). We aimed at data triangulation on patient preferences by including GPs views on their patients’ preferences in our search strategy. The decision to start a diagnostic trajectory is usually made in general practice and GPs have longstanding relationships with their patients and knowledge of their patient’s personal life and their preferences (33). The full electronic search strategy for PubMed can be found in the appendix. The search strategies in the other databases were similar with equivalent index terms. The literature search was conducted in May 2020 and was updated in January and October 2021. Study selection Papers were considered for inclusion if they provided data regarding patients’ and SOs’(i.e. people that are directly involved in the decision-making process, such as spouses, children, other family members or friends) preferences for a dementia diagnosis and (shared) decision-making in that regard (Table 1). After removing duplicate papers, two researchers (IL and MH) independently excluded papers based on title and abstract. Next, the remaining papers were read full text. After each step, the researchers compared results and discussed differences. In cases of disagreement, a third researcher (CW) was consulted.
| 20 Table 1. Inclusion and exclusion criteria. Inclusion Exclusion Written in English or Dutch Written in languages other than English or Dutch Studies reporting on preferences of people with memory complaints and their significant others regarding starting a diagnostic trajectory for dementia Studies reporting on the development or cost/benefits of the tools (e.g. biomarkers) used to make an early/timely diagnosis Studies reporting on patients’ or significant others’ views on (shared) decision making regarding starting a diagnostic trajectory for dementia Studies reporting on whether a dementia diagnosis should be disclosed or not Studies reporting on GP’s views on their patients/significant others’ preferences for starting a diagnostic trajectory or (shared) decision making Studies reporting on general public or health care professionals’ preferences for diagnostic testing on dementia Studies published after 2010 Studies reporting on preferences for dementia screening in healthy individuals Quality assessment The Mixed Method Appraisal Tool (MMAT) was used to evaluate the methodological quality of the included studies. The MMAT is a tool designed for quality appraisal in systematic reviews that include quantitative, qualitative, and mixed methods studies (34). The MMAT consists of two screening questions and five quality criteria for each study type (qualitative research, randomized controlled quantitative research, non-randomized controlled quantitative research, observational descriptive quantitative research, and mixed methods research). For mixed methods studies 15 quality criteria are evaluated (those for qualitative research, quantitative research and mixed methods research). The screening questions assess if the study is an empirical study and focuses on the clarity of research questions and whether the data collected are sufficient to answer the research questions. Of the corresponding quality criteria, it is evaluated whether they were met or not met. Ratings vary between 0% (no quality criteria met) and 100% (all five quality criteria met) (34, 35), and are recommended to be completed with a description of the quality of the studies. IL and MH independently assessed the included studies, discussed their individual ratings, and agreed on a final rating.
2 | 21 Data synthesis A thematic synthesis of the included studies was performed (36, 37). Thereto, ATLAS.ti version 8.4. was used to analyze the results sections of each paper. IL and MH independently performed line-by-line coding, conceptualized the data, and identified concepts. This process was deductively led by the conceptual definition of preferences (32) and completed with the addition of inductive codes. Using this theoretical framework allowed us to integrate single study results on an overarching level. Together, the researchers grouped concepts into themes and subthemes. Conceptual links between the themes were identified and visually displayed in Figure 2 (IL and MH). In several group sessions, researchers (IL, MH, CW, MP) discussed identified concepts, themes, and conceptual links to finalize the analyses. Results Nine papers met the inclusion criteria (Figure 1), seven qualitative studies and two mixed-methods studies (see Table 2 for their characteristics). Most papers were from the UK (N=3), followed by the Netherlands (N=2), Canada (N=2), Singapore (N=1), and Germany (N=1). Study populations consisted of PwMCs and SOs. The sample of PwMCs consisted of people that chose to pursue a diagnostic trajectory for their memory complaints as well as people who chose not to do so. They either visited their GP (38, 39) or a memory clinic (38, 40-44). In two studies (45, 46) it was unclear where the diagnostic trajectory had taken place. In all papers, participants were asked to retrospectively reflect on their decision-making process to start a diagnostic trajectory for dementia.
| 22 Figure 1. Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) flow diagram.
2 | 23 Table 2. Study designs, methods, findings, and themes related to patient preferences from included studies. Authors (Year), Country Study design Timing of data collection Participants (N = sample size), settings and information on diagnosis* Aims Themes (based on data synthesis) MMAT Birt, Poland, Charlesworth, Leung & Higgs (2019), United Kingdom A qualitative design using individual interviews After deciding on seeking help for memory complaints N = 25 patients with dementia diagnosis N= 9 patients with MCI diagnosis N= 7 patients with subjective cognitive concerns To understand the experiences of people who seek help for subjective cognitive concern and memory loss, including people not referred for further assessment. Feeling of needing to do something Beliefs on the necessity of diagnostic testing Expected outcomes of the decision on diagnostic testing Perception of symptoms The support or wishes of the social network Interaction with HCP 80% of quality criteria met Chrisp, Tabberer, Thomas & Goddard (2012), United Kingdom A qualitative design using a thematic analysis of case studies After memory clinic visit N = 20 SOs of PwMCs, information on diagnosis not provided To identify influences on the decision to consult a HCP for symptoms of dementia. Beliefs on the necessity of diagnostic testing Expected outcomes of the decision on diagnostic testing Perception of symptoms The support and wishes of the social network Interaction with HCP Health status of the PwMC 60% of quality criteria met Koehn, Badger, Cohen, McCleary & Drummond (2016), Canada A qualitative design using individual interviews After receiving a diagnosis (pre-diagnostic experience maximum 4 years ago) N = 29 people diagnosed with dementia, N = 34 SOs To provide a comprehensive understanding of the challenges to securing a diagnosis of dementia, especially among ethnic minority groups. Beliefs on the necessity of diagnostic testing Expected outcomes of the decision on diagnostic testing Perception of symptoms The support or wishes of the social network Interaction with HCP Societal factors 40% quality criteria met
| 24 Authors (Year), Country Study design Timing of data collection Participants (N = sample size), settings and information on diagnosis* Aims Themes (based on data synthesis) MMAT Kunneman et al. (2017), The Netherlands A qualitative design using focus groups complemented with surveys After undergoing testing at a memory clinic N = 10 people with a selfreported diagnosis (FTD, MCI, early AD or memory complaints) and their SOs N=1 person who was told not be concerned, no dementia To assess patients’ and caregivers’ views on and experiences with decisions about diagnostic testing for Alzheimer’s disease and receiving test results. Feeling of needing to do something Beliefs on the necessity of diagnostic testing Expected outcomes of the decision on diagnostic testing Interaction with HCP Health status of the PwMC Societal factors The support or wishes of the social network 100% of quality criteria met Lee, Barlas, Thompson & Dong (2018), Singapore A qualitative design using semistructured interviews After decisionmaking process on further assessment resulting from a positive screening result for cognitive impairment N = 5 SOs of people with moderate to severe risk of cognitive impairment, information on further diagnosis not provided To explore caregivers’ experience in decision-making regarding patients’ participation in further diagnostic assessment to confirm cognitive screening results for dementia. Expected outcomes of the decision on diagnostic testing Perception of symptoms The support or wishes of the social network Health status of the PwMC Societal factors 60% of quality criteria met Lohmeyer, AlpinarSencan & Schicktanz (2011), Germany A qualitative design using individual interviews and focus groups After undergoing testing at a memory clinic N = 12 people with selfreported MCI or early dementia diagnosis, 32 SOs of people with (early) dementia To explore caregivers’ experience in decision-making regarding patients’ participation in further diagnostic assessment to confirm cognitive screening results for dementia. Feeling of needing to do something Expected outcomes of the decision on diagnostic testing Perception of symptoms Interaction with HCP Societal factors 80% of quality criteria met
2 | 25 Authors (Year), Country Study design Timing of data collection Participants (N = sample size), settings and information on diagnosis* Aims Themes (based on data synthesis) MMAT Morgan et al. (2014), Canada A qualitative longitudinal design using individual interviews After the decision on testing Two timepoints: Day of the appointment at the memory clinic Six months after the memory clinic appointment N = 46 SOs of PwMCs, 33 patients were diagnosed with AD, MCI, FTD or VCI, 5 patients had no cognitive impairment, 2 patients had inconclusive results after diagnostic testing To explore rural family caregivers’ experiences in the period leading up to diagnostic assessment at a memory clinic, their hopes, and expectations of the assessment, and their experiences six months following the diagnosis. Feeling of needing to do something Beliefs on the necessity of diagnostic testing Expected outcomes of the decision on diagnostic testing Perception of symptoms The support or wishes of the social network Interaction with HCP Health status of the PwMC 100% of quality criteria met Mukadam, Cooper, Basit & Livingston (2011), United Kingdom A qualitative design using semistructured interviews After dementia diagnosis N = 18 SOs of people with a dementia diagnosis To explore the link between attitudes to help-seeking for dementia and the help-seeking pathway in minority ethnic carers. Expected outcomes of the decision on diagnostic testing Perception of symptoms Interaction with HCP Health status of the PwMC Societal factors 100% of quality criteria met Visser et al. (2019), The Netherlands Observational study complemented with surveys After undergoing testing at a memory clinic N = 136 dyads (PwMC - SO) and N = 41 clinicians, N= 53 patients diagnosed with dementia N= 21 patients with an MCI diagnosis N = 34 patients had normal cognitive function after diagnostic testing N = 28 patients had another or unclear diagnosis Describe the clinician-patient communication in the diagnostic process in memory clinics. Interaction with HCP 100% of quality criteria met Note. HCP= Health Care Professional, MCI = Mild Cognitive Impairment, AD = Alzheimer’s disease, FTD = Frontotemporal dementia , VCI = vascular cognitive impairment, no dementia, MMAT = Mixed Method Appraisal Tool, PwMC = Person with memory complaints, SO = Significant Other, , * = diagnosis as reported by the authors in the original study.
| 26 Quality assessment The quality of the studies was high to moderate, with MMAT ratings of 80 -100% (6 studies), 60% (2 studies), 40% (1 study). The two qualitative studies that were complemented with surveys (41, 44) did not identify themselves as mixed method studies and mainly focused on the qualitative part of their study, therefore only the qualitative parts of these studies were assessed for quality criteria and used for data synthesis. For the qualitative studies with an 80100% score the qualitative approach was adequate for the research question, findings were (mostly) adequately derived from the data and interpretation of results was substantiated by data (for example by using quotes). For the studies with an 60% score it was unclear if all results were substantiated with data and whether the data collection methods were adequate to address the research question. In the study with a 40% score, it was impossible to tell how the findings were derived from the data and if there was coherence between data sources, collection, analysis and interpretation. Findings Six analytic themes emerged from our data synthesis. Preferences for starting a diagnostic trajectory for dementia arose from (1) views on diagnostic trajectories. These views are influenced by (2) symptom normalization or validation, (3) the support or wishes of the social network, (4) interaction with HCPs, (5) the health status of the PwMC, and (6) societal factors such as stigmatization, cultural beliefs, and socioeconomic status (see Figure 2). See the appendix for an extensive overview of the themes, categories, codes, and illustrative quotes.
2 | 27 Figure 2. Visual representation of identified themes. Note. HCP = Health Care Professional, PwMC = Person with memory complaints.
| 28 Theme 1: views on starting diagnostic trajectories PwMCs and SOs form a view on starting a diagnostic trajectory based on needs, beliefs on the necessity and expected outcomes of starting a diagnostic trajectory. Feeling of needing to do something. PwMCs and SOs frequently describe the feeling of ‘needing to do something’. PwMCs and SOs describe this as ‘the ability to do something for your own health’ or ‘the ability to take control of the situation’ (42, 43). ‘Mrs. Weber: I think that everyone should do something for his health or illness. No? And not simply sit it out and put the blame on other things’. (42). PwMCs specifically want to reduce uncertainty about the cause of the symptoms (38, 41, 43). Furthermore, the feeling that something is off and the hope to be reassured are motives for PwMCs and SOs to pursue a diagnostic trajectory (38). Beliefs on the necessity of a diagnostic trajectory. PwMCs and SOs held beliefs that determined their perceived necessity of starting a diagnostic trajectory. PwMCs did not perceive a diagnostic trajectory as necessary if they believed an HCP would not be able to help them, believed they did not need help, or prioritized their physical problems (39, 40, 45). ‘Things that are important to her–her corns, she has very painful corns that sometimes leads her to not being able to move, and sometimes I can’t even get an appointment at the polyclinics. And then it gets worse to walk. . . To her, these two things matter more than her mind. In her mind, she’s fine’ (39). These beliefs were often fueled by PwMCs’ fear of developing dementia (38, 40). On the contrary, PwMCs and SOs who believed a diagnosis would help them move forward psychologically or that there was no harm in finding out, pursued a diagnostic trajectory (41, 43). Expected outcomes of starting a diagnostic trajectory. SOs specifically had expectations on the benefits of a diagnosis, which resulted in pursuing a diagnostic trajectory. In five studies SOs expected to start
2 | 29 (medical) treatment as soon as possible or to at least have “a foot in the door” for future therapy as a result of a diagnosis (39, 41-43, 46). ‘Well I’d like to think there’s a medication that would help her. It helps everything else. It certainly is not going to help her a year or two down the road, it’s not going to, it’s too late. I’m hopeful that maybe there will be yet. (husband)’ (43). In addition, SOs were more inclined to start a diagnostic trajectory when they expected that a diagnosis would ease caregiver burden, would enable access to support and specialist care (39, 42), or provide information on the prognosis of the disease (43). In addition, SOs expected that a diagnosis would stimulate the PwMC to start living healthier or aid advance care planning (38, 42) Contrarily, some SOs expected a diagnostic trajectory would do more harm than good. This expectation came from respecting the PwMCs’ wish to not get tested or protecting the PwMC from the distress of diagnostic testing (39, 40, 46). Furthermore, they expected that a diagnosis would prevent the PwMC from living happy while still healthy which made them less inclined to pursue diagnostic testing (39), as did the expectation that a diagnosis would not contribute to future planning or to slowing down dementia (39, 42). In one study, a SO explained the PwMC’s reluctance towards diagnostic testing through the expectation that a diagnosis would result in placement in an institution (45). Theme 2: Normalization or validation of symptoms Normalizing symptoms by PwMCs and SOs weakens beliefs on the necessity of a diagnostic trajectory and the drive to do something because symptoms are not interpreted as problematic. SOs indicated that they did not believe a diagnostic trajectory was necessary if they interpreted memory complaints as part of normal aging, thought they were due to another illness, did not associate them with a disease, or perceived a low level of deterioration (39, 40, 45, 46). Conflicting interpretations of symptoms between the PwMC and the SO delayed starting a diagnostic trajectory (40, 42, 45, 46). The belief that diagnostic testing was not necessary could be strengthened when others normalized the symptoms too (40, 42). Contrary, when symptoms are interpreted as problematic by either the PwMC, SO or others such as friends and family members, worries about them increase, which contributes to the feeling of needing to do something about the symptoms and a heightened
| 30 belief on the necessity of a diagnostic trajectory. Symptoms were interpreted as problematic when changes in the PwMC’ behavior were unmanageable, abnormal, or could not be explained by alternative explanations (38, 39, 43, 45, 46). Moreover, the necessity of a diagnostic trajectory was acknowledged by PwMC and SO when the symptoms were validated by others (38, 45, 46). ‘Then some a-an old friend of his noticed he was doing that. So err I thought it was about time that we went to see the doctor, our GP.’ (46) Theme 3: Support or wishes of the social network Support of the social network (i.e. close family and friends other than the SO) could be vital in taking the step to start a diagnostic trajectory. The wishes of close family and friends can influence expected outcomes or can weaken beliefs on the necessity of a diagnostic trajectory. PwMCs who were afraid to take the step to start a diagnostic trajectory alone, but were supported by their family and friends (regardless of their perception of symptoms) decided to pursue diagnostic testing (38), whereas a limited social network of the PwMC is often perceived as a barrier for starting diagnostic testing (40, 43, 45). ‘I was getting a little bit worried because I knew I was repeating things, but I wasn’t brave enough to take the first step myself so when my daughters asked if I would go to the doctor I said I would.’ (38). On top of that, PwMCs pursued a diagnostic trajectory just for the sake of accommodating family wishes (38, 41). On the contrary, the PwMCs’ decision to start a diagnostic trajectory was delayed when caregiving children refused to deal with the symptoms (43). Moreover, one PwMC decided to not pursue a diagnostic trajectory because a relative perceived help seeking as unhelpful (38). Theme 4: Interaction with HCPs HCPs’ reactions to symptoms and communication style could heighten and weaken PwMCs’ and SOs’ beliefs on the necessity of a diagnostic trajectory. PwMCs and SOs described interactions with HCPs whereby the HCP normalized or was dismissive of their symptoms, which in most cases delayed starting a diagnostic trajectory (38, 40, 43, 45, 46).
2 | 31 ‘The family doctor said ‘‘no’’ in the beginning, he did not think of it. He felt I might be too sensitive’ (45). However, a strong drive to reduce uncertainty on the cause of the symptoms led some PwMCs to seek a second opinion (38). When the HCP took concerns about memory complaints seriously, PwMCs’ and SOs’ beliefs on the necessity of starting a diagnostic trajectory were confirmed and diagnostic testing was pursued (38, 46). Moreover, when HCPs presented decisions implicitly (instead of an option for which patients’ preferences mattered) beliefs on the necessity of starting a diagnostic trajectory could be heightened or weakened (depending on the patients’ initial beliefs) (44). The ability of the PwMC and SO to communicate their concerns, could in turn, impact the HCPs reaction and therewith PwMCs’ and SOs’ views on starting a diagnostic trajectory (45). Also, the level of trust in the HCP and their perceived qualifications (e.g. age, education level) influenced whether their advice impacted views on starting a diagnostic trajectory (38, 45). Theme 5: Health status of the PwMC Beliefs on the necessity of starting a diagnostic trajectory could be affected by heightened or diminished awareness of symptoms due to the health status of the PwMC. PwMCs with a family history of dementia were more inclined to believe diagnostic testing is necessary in the presence of minor memory complaints (41). ‘It runs in the family, and that is another reason why I decided relatively quickly to do something.’ (41) Also, comorbidities often provide a distraction from the memory complaints, which can affect the belief of PwMCs, SOs, and HCPs that diagnostic testing is not yet needed (40, 45). However, the need for medical help for physical complaints can also lead to HCPs to notice memory complaints, which in turn affect the belief that a diagnostic trajectory is necessary (40, 46). In addition, a crisis (e.g. a fall or traffic accident) often strengthens the belief that diagnostic testing is necessary (40, 43). Other health-related factors can accelerate or decelerate the decision to start a diagnostic trajectory, sometimes regardless of the PwMCs and SOs’ initial views. For example, impaired mobility of the PwMC was a reason to defer a diagnostic trajectory (39).
| 32 Theme 6: Societal factors The expected outcomes of a diagnostic trajectory could be affected by cultural perceptions on the benefits or drawbacks of a diagnosis and its societal consequences. Some PwMCs and SOs perceived a diagnostic trajectory as harmful due to a fear of being stigmatized for mental health problems (42, 45, 46). Cultural beliefs on family hierarchy, the family responsibility of taking care of older family members, and ceding household duties later in life also lead to views that diagnostic testing is not beneficial (39, 46). ‘You don’t want to bring in outside agencies unless you have to . . . because it’s intrusive . . . and when you can’t deal with it we’ll go to outside agencies who will help us deal with it. That’s where we’re coming from’ (46). Moreover, a lack of knowledge due to societal factors like a minority background can impact views on the necessity and accessibility of a diagnostic trajectory as mentioned by a few participants in Koehn et al. (45). Financial motives related to a diagnosis (e.g. social security income), can be a reason to pursue a diagnostic trajectory (41). Discussion This integrative review is the first to explore and map preferences of PwMCs and SOs on starting a diagnostic trajectory for dementia. PwMCs and SOs, who decided to pursue a diagnostic trajectory, were driven by uncertainty about symptoms, believed testing was necessary to help deal with symptoms, or expected to start treatment or have access to other forms of support after a diagnostic trajectory. PwMCs and SOs who delayed or decided to refuse a diagnostic trajectory believed they did not need help, prioritized physical problems, or expected diagnostic testing to be harmful in living (mentally) healthy. These views do not exist independently but are influenced by normalization or validation of symptoms, support or wishes of their social network, interactions with HCPs, health status of the PwMC, and societal factors such as stigmatization, cultural beliefs, and socioeconomic status. Although in theory the identified feelings, beliefs and expectations are defined as parts of the concept preferences (32), in this study beliefs on the necessity and expected outcomes of starting a diagnostic trajectory appeared to be partly circular. That is, when the expected outcomes are favourable, beliefs on the necessity of starting a diagnostic trajectory are likely to be heightened and vice
2 | 33 versa. For example, PwMCs with a family history of dementia are more inclined to believe diagnostic testing is necessary when minor memory complaints are present (41). This could be explained by expected outcomes in the form of support after diagnosis based on previous experiences with family members. However, ‘the feeling of needing to do something’ can be seen as ‘the gut feeling’ often described as ‘doing something is better than doing nothing’ or the ‘more-is-better heuristic’ (47). These ‘gut feelings’ do not always lead to patient preferences that are concordant with their values, because emotions can alter perceptions of quantity and value (48). The ‘feeling of needing to do something’ might therefore be less susceptible to the identified factors because it’s a more emotion-based view. This review highlights the challenge of finding the right timing for starting a diagnostic trajectory for both the PwMC and their SO, as their preferences can be different as was suggested by others (49, 50). Some PwMCs believed nothing could be done about the symptoms, help was not needed (45, 46), or a diagnosis was not perceived as beneficial for future planning (39, 42), whereas SOs specifically perceived diagnostic testing as beneficial because of possibilities to start treatment or gain access to support (39, 41-43, 46). The expectations on the benefits of a diagnostic trajectory identified in our review are similar to a review that identified perceived benefits of screening such as access to treatment, financial benefits, and the ability to plan ahead in patients, clinicians, and the general public (24). They highlight the importance of family on the decision to undergo screening, which is similar to the role of the family in deciding on starting a diagnostic trajectory for dementia identified in our review. Our results show that PwMCs and SOs preferences are shaped by interactions with HCP, societal factors, and support or opinions of the social network, this is in line with the ecological perspective on patient preferences by Street et al. (51). Their ecological model suggests that patient preferences are shaped by the social, cultural, economic, and media context. The context of the health care system might influence patients’ preferences on top of the identified societal factors in our review. Access to health and support services has been experienced as complex by persons with dementia and their caregivers (5, 52), which may negatively affect expected outcomes of a diagnostic trajectory. The ecological perspective also describes that patient preferences can in turn be affected by clinical encounters, when patients’ initial preferences based
www.ridderprint.nlRkJQdWJsaXNoZXIy MTk4NDMw