Economic evaluations of technology-based interventions used to provide care support for people with mild dementia or mild cognitive impairment and their caregivers: a systematic review

1 Economic evaluations of technology-based interventions used to 1 provide care support for people with mild dementia or mild cognitive 2 impairment (MCI) and their caregivers: a systematic review 3 4 Running title: “Efficiency of tech interventions for mild dementia” 5 6 Ana Magdalena Vargas-Martínez1* 7 8 1 Department of Nursing, Faculty of Nursing, Physiotherapy and Podiatry, University of Seville, Seville, 9 41009, Spain. 10 11 Correspondence author 12 13 Vargas-Martínez AM, Department of Nursing, Faculty of Nursing, Physiotherapy and 14 Podiatry, University of Seville – St. Avenzoar 6, Seville, 41009, Spain. Telephone 15 number: +34 670709328 16 Email: [email protected] 17 2 Economic evaluations of care support and caregivers in technology18 based interventions for people with mild dementia or mild cognitive 19 impairment (MCI): a systematic review 20 21 ABSTRACT 22 23 BACKGROUND The cost-effectiveness of interventions is a key issue owing to the 24 limited resources of healthcare services. 25 OBJECTIVE To conduct a systematic review of economic evaluations of technology26 based healthcare interventions in care support for people with dementia or mild 27 cognitive impairment (MCI) and their caregivers, and of the tools used to assess 28 effectiveness and costs. 29 METHODS The following databases were used: PubMed, National Health Service 30 Economic Evaluation Database (NHS EED) and Health Technology Assessment 31 (HTA). A total of 207 articles from 2012 to 2024 were identified and then screened. 32 RESULTS Seventeen studies were included, of which nine were study protocols. 33 Almost half (n= 8) the interventions were multicomponent. The most common 34 components used in the interventions were cognitive stimulation, physical functioning 35 and continuing support. Regarding the efficiency results of these interventions, only 36 three studies provided a full economic evaluation. The most frequent tools in the 37 economic evaluations used to measure effectiveness (measured in QALYs) and costs 38 were the European Quality of Life-5 Dimensions (EQ-5D) and Resource Utilization in 39 Dementia (RUD) instruments, respectively. 40 CONCLUSIONS Most of the interventions evaluated were cost-effective. However, 41 these results should be interpreted with caution, given the scarcity of the literature, 42 3 and further economic evaluations of technology-based healthcare interventions for 43 people with mild dementia or MCI care support and their caregivers are therefore 44 needed. Additionally, a meta-analysis could not be performed due to the heterogeneity 45 of the data. 46 47 48 KEYWORDS 49 Alzheimer’s disease, Dementia, Mild Cognitive Impairment, Technology, Wearable 50 Electronic Devices, Cost-Effectiveness Analysis, Efficiency, Quality of Life 51 52 4 INTRODUCTION 53 With an aging population, dementia represents a significant public health and care 54 challenge which has been recognized and prioritized by the World Health Organization 55 (WHO).1 In 2015, it was estimated that there were nearly 10 million new cases of 56 dementia each year worldwide, with one new case coming to light every three 57 seconds. This figure is set to rise to 82 million in 2030, and to 152 million in 2050. It is 58 also noteworthy that, according to the forecasts, the majority of people who will 59 develop dementia will be in lowand middle-income countries.1 60 Dementia has significant social and economic implications in terms of direct costs of 61 medical and social care, and the costs of informal care. In 2019, informal carers (most 62 commonly family members and friends) spent on average 5 hours per day providing 63 care for people living with dementia.2 In the same year, the estimated total global 64 societal cost of dementia was US$ 1.3 trillion, and these costs are expected to top 65 US$ 2.8 trillion by 2030 as both the number of people living with dementia and care 66 costs increase.2 The economic impact on those who look after people with dementia 67 in their homes is especially notable, which corresponds mainly to informal caregivers. 68 In Spain, a study published in 2015 estimated the value of the informal care provided 69 for disabled people, quoting figures equivalent to 1.73–4.90% of the gross domestic 70 product for the year of study.3 In particular, the impact of informal care in dementia 71 represents around 50% of global costs, and in some cases even exceeds the direct 72 costs.4 For this reason, it is crucial to involve caregivers in care planning, while paying 73 attention to the wishes and preferences of people with dementia and their families, 74 given the need to care for these people and the caregiver’s burden.2 75 Another crucial factor is the continuity of care between different care providers, 76 multiple sectors and levels of the system, and the active collaboration between paid 77 5 and unpaid caregivers, from the first symptoms of dementia until the end of life. To 78 achieve this, integrated, evidence-based, person-centered care is required in all 79 settings where people with dementia live, mainly in their homes and in the community. 80 Furthermore, the systematic monitoring and evaluation of the use of health and social 81 care systems can provide the best available evidence for the development of policies 82 and service delivery, and can improve prevention, accessibility, and coordination of 83 care for people with dementia throughout the journey from risk reduction to end of life.2 84 As regards integrated care, older adults require a comprehensive set of services to 85 prevent, slow down, or reverse the deterioration of their physical and mental 86 capacities.5 These services need to be tailored to meet the person’s needs (person87 centered) and coordinated between different health and social care providers, and 88 they must avoid causing the user financial hardship.6-8 Within this field of action, the 89 WHO supports its Member States to understand, design, and implement a person90 centered, integrated model of care by producing evidence, guidance, and resource 91 packages. 92 Over recent years, the use of health interventions delivered through digital 93 technologies, such as smartphones, web-based resources and text messages has 94 increased. Its use has been aimed at facilitating remote access to effective treatments, 95 as well as improving the approach to chronic pathologies and promoting healthy 96 lifestyles, with the aim of increasing or replacing traditional health interventions.9 As 97 regards dementia, a recent systematic review and meta-analysis of internet-based 98 support interventions for family caregivers of people with dementia concluded that they 99 were generally effective in improving symptoms of depression, perceived stress, 100 anxiety, and self-efficacy in caregivers, and had potential benefits for the final 101 beneficiaries.10 102 6 Additionally, as the prevalence of dementia diagnoses rises and the expenses 103 associated with dementia care continue to escalate, it has become imperative to 104 conduct economic assessments. These evaluations are essential to ascertain the 105 cost-effectiveness of the non-pharmacological interventions provided, particularly for 106 technology-based interventions. Nevertheless, the available economic evidence for 107 these interventions is currently limited:11 for instance, the findings of the updated 108 review cited above10 only provide results of cost-effectiveness of psychological and 109 behavioral therapies in general, and not of strategies that incorporate technology such 110 as wearable electronic devices or ambulatory monitoring, among others. 111 Therefore, this systematic review aims to assess the economic impact of technology112 based healthcare interventions in the care support provided for people with dementia 113 or mild cognitive impairment (MCI) and their caregivers, as well as the tools used to 114 assess effectiveness and costs, as part of a larger research program which assesses 115 the evidence and conducts primary research to look for an integrated solution for the 116 sustainable care of elderly multimorbid patients with dementia (CAREPATH)12-13 in 117 Europe, funded by research grants from the European Commission. 118 119 MATERIALS AND METHODS 120 Sources and Search strategy 121 We conducted a systematic review of literature by searching the following databases: 122 PubMed, National Health Service Economic Evaluation Database (NHS EED) and 123 Health Technology Assessment (HTA). In addition, a secondary review was carried 124 out by searching for citations. The search was restricted to the literature published in 125 the last 12 years (2012-2024). 126 7 The search strategy was developed and tested before deciding on the final search 127 terms (Document 1, Supplementary material). 128 Eligibility 129 Table 1 presents the inclusion and exclusion criteria, using the Population, 130 Interventions, Comparators, Outcomes, and Study Designs framework.14 Only English 131 language sources were consulted. 132 As regards study designs, the economic analysis should be partial or full. Economic 133 Evaluation (EE) is recognized as the most pertinent instrument for health care 134 decision-makers,15 enabling us to compare alternative courses of action in terms of 135 both costs and health outcomes. There are distinct types of EEs, differentiated 136 primarily by the method in which the outcomes are measured, valued, and 137 incorporated into the analysis.16 In Cost-Effectiveness Analysis (CEA), alternative 138 costs and outcomes are compared, measured and evaluated in natural units derived 139 from clinical trials or observational studies. In Cost-Utility Analysis (CUA), the expected 140 costs and outcomes for each intervention are calculated, with outcomes predominantly 141 expressed as quality-adjusted life years (QALYs), thus combining quality and length 142 of life into a single metric.16 The results of an EE are expressed in terms of an 143 incremental cost-effectiveness ratio (ICER), which is defined as the difference in mean 144 expected costs divided by the difference in mean expected outcomes.16 The ICER 145 provides a metric for the expected cost required to achieve a unit of effect. When a 146 program or intervention enhances outcomes while reducing costs, it is considered to 147 exhibit dominance.16 The study protocols are included, given that the main objective 148 of this review was to compile the most frequently-used tools in the literature to assess 149 effectiveness and costs, in order to design methods to carry out the economic 150 evaluation of CAREPATH.12-13 151 8 Study selection and data extraction 152 The study selection and data extraction were completed by the study author alone. 153 First, the researcher screened the titles and abstracts of all the citations retrieved 154 against the inclusion criteria. Next, the full texts of these studies were retrieved and 155 again screened against the inclusion criteria. After screening, the data from these 156 studies were extracted and set out in a table similar to the standardized form for the 157 CRD’s Guidance for Undertaking Reviews in Health Care.14 The fields included author, 158 year, country, study design, study objective, sample characteristics, intervention and 159 comparator(s), as well as the analysis, including the decision analytic approach, time 160 horizon, perspective, outcome measurements, and effectiveness and cost data. 161 This systematic review follows the recommendations of the Preferred Reporting Items 162 for Systematic Review and Meta-Analysis Protocols (PRISMA-P) on reporting 163 systematic reviews.17 164 165 RESULTS 166 Study selection 167 Overall, 207 studies were identified, 17 of which were included in the review. The 168 selection process is shown in Figure 1. Two systematic economic reviews for 169 dementia care were identified,18-19 which were consulted to identify other potentially 170 relevant studies not available on the PubMed, HTA and NHS EED databases, as well 171 as instruments/tools to obtain data on health and non-health resource use for people 172 with dementia and/or their carers. 173 174 Study characteristics 175 9 Table 2 summarizes the characteristics of the economic analyses included 176 (Secondary outcome measures are reported in Table 1, Supplementary Material). 177 Of the 17 studies found in the last 12 years (2012-2024) which met the inclusion 178 criteria, 9 are study protocols. A total of 12 carried out or were going to carry out a 179 complete economic evaluation (cost-effectiveness, cost-utility or cost-benefit 180 analysis), of which only one19 used a decision analytic model, with the rest (n=5) of 181 the studies carrying out an analysis only of effects and costs. Ten studies used a 182 randomized controlled trial design,20-29 one a quasi-experimental design,30 two a 183 prospective observational design,31-32 and one a cross-sectional study,33 while one did 184 not report the study design,34 one used a mixed design (randomized controlled trial 185 and qualitative study)35 and one used a sequential multiple assignment randomized 186 trial (SMART).36 187 There were 11 studies directed towards supporting dyads of people with dementia 188 (PwD) and their caregivers20-23,25-26,28,31-32,34,36, 3 for only PwD27,30,35 and 3 specifically 189 for carers24,29,33. 190 Data Synthesis Results 191 Tools used to assess effectiveness and costs. 192 The studies differed in terms of choice of outcome measures, although some 193 similarities were identified. Nine studies used the quality-adjusted life-year (QALY) to 194 measure health benefits.20,22,24-26,28-30,36 Table 3 shows the instruments/tools (scales 195 and questionnaires) used in the literature to measure the outcome. Mainly, health 196 outcomes are measured by: severity of dementia through instruments such as 197 Neuropsychiatric Inventory Questionnaire (NPI-Q)23,26, the Revised Memory and 198 Behavior Problem Checklist (RMBPC)29,34, the Mini-Mental State Examination 199 (MMSE)20,22,25-28,32,34, quality of life through EQ-5D-5L20,22,24,26,35 or in its Proxy version 200 16 aimed to report on the psychometric evidence of the EQ-5D-5L in people with 350 dementia, found that the available evidence regarding the responsiveness of the EQ351 5D-5L was inconclusive, as the ability to self-complete this questionnaire decreases 352 as the disease progresses. Given the nature of the interventions considered in this 353 review and the target population, it would be of interest to measure not only the results 354 (Patient-Reported Outcome Measures - PROMs) but also the experiences (Patient355 Reported Experience Measures - PREMs) of patients in the provision of this type of 356 health or social health care, which is currently recognized as a valuable mechanism 357 to evaluate the quality of care from the patient's perspective.45-46 Furthermore, these 358 measures are considered of special interest in the evaluation of care for people with 359 long-term illnesses and comorbidities.47-49 360 Regarding the use of resources and costs, unlike the review carried out by Yang et 361 al.50 aimed at reporting the trials carried out using these instruments in order to guide 362 the design of future trial-based cost-effectiveness studies, the specific tool used most 363 commonly among the studies included in this review to measure both the use of 364 resources by patients with dementia and their caregivers was the RUD. Along these 365 lines, Angeles et al.18 concluded that the use of this instrument in studies encouraged 366 more harmonious analyses, although further studies are required. 367 Limitations 368 The main limitations of the studies included in this review were the scarcity of results 369 on cost-effectiveness, given the high number of study protocols that are yet to produce 370 results and studies based on both effects and cost analysis, the disparity in the type 371 of technology used in the studies, and the short time horizon used in the analyses, 372 which could affect the reliability of the conclusive results regarding the efficiency of 373 these interventions. It is also worth highlighting the single authorship as a limitation of 374 17 this review, as it may influence the selection and interpretation of the studies included. 375 To mitigate its effects, a critical reading of each of the studies included was carried out 376 through the use of standardized and internationally-used checklists depending on the 377 type of study: CONSORT51 for clinical trials, STROBE52 for observational studies and 378 CHEERS53 for economic evaluations. Another limitation is the search limit of the last 379 10 years set for this review, mainly because the number of health interventions carried 380 out through digital technologies has increased exponentially in recent years.9 381 Furthermore, specifically, the economic evaluation of this type of digital health 382 intervention is currently plagued with methodological issues.9 The evaluation of 383 medical devices has seen a recent expansion in its area of definition, which is now 384 understood as "any instrument, apparatus, appliance, software, material or other 385 article, whether used alone or in combination....to be used for human beings for the 386 purpose of diagnosis, prevention, treatment, monitoring or alleviation of disease", thus 387 employing a broader concept than merely digital technology, through the European 388 MedtecHTA project that began in 2013.54 389 In conclusion, this paper encompasses seventeen studies that analyze both the effects 390 and costs derived from technology-based interventions for PwD and their caregivers. 391 Most of the interventions (12 out of 17) were carried out in European countries with 392 comparable underlying health and social care systems. It could be concluded that 393 most of the interventions evaluated were cost-effective when addressed to the PwD 394 or to the caregiver separately, but not to the PwD-caregiver dyad. However, there is a 395 lack of full economic evaluations, that is, those that include analyses of cost396 effectiveness, cost-utility, cost-benefit or cost minimization, as only three studies 397 reported these data. It is important to note, too, that this review includes 9 study 398 protocols that report on the methodology used to carry out an economic evaluation of 399 18 technology-based interventions for supportive care for patients with dementia and their 400 caregivers. 401 The main tools used to assess effectiveness and costs to carry out the economic 402 evaluation, were QALYs, measured through the EQ-5D as a measure of effectiveness, 403 and the instrument known as RUD, to measure the use of health and social resources. 404 405 AUTHOR CONTRIBUTIONS 406 A.M.V.M: Conceptualization; Methodology; Formal analysis; Research; Writing – 407 Original Draft; Writing & Editing. 408 409 ACKNOWLEDGEMENTS 410 I am grateful to Dr Juan Oliva, Dr Luz María Peña-Longobardo and Pedro Abizanda 411 Soler, who provided useful comments on the draft. This paper is part of the 412 CAREPATH project, which is aimed at assessing evidence and conducting primary 413 research into an integrated solution for the sustainable care of elderly multimorbid 414 patients with dementia in Europe and is funded by research grants from the European 415 Commission (Reference: 945169 — CAREPATH — H2020-SC1-BHC-2018-2020). 416 417 FUNDING 418 The author has no other source of funding to report. 419 420 DATA AVAILABILITY 421 The data supporting the findings of this study are available within the article and/or its 422 supplementary material. 423 424 19 CONFLICT OF INTEREST 425 The author reports no conflicts of interest. 426 20 REFERENCES 1. GBD 2019 Dementia Forecasting Collaborators. Estimation of the global prevalence of dementia in 2019 and forecasted prevalence in 2050: an analysis for the Global Burden of Disease Study 2019. Lancet Public Health 2022: 7: e105-e125. 2. Global action plan on the public health response to dementia 2017–2025. Geneva: World Health Organization; 2017. 3. Oliva-Moreno J, Peña-Longobardo LM and Vilaplana-Prieto C. An estimation of the value of informal care provided to dependent people in Spain. Appl Health Econ Health Policy 2015; 13: 223-231. 4. Wimo A, Seeher K, Cataldi R, et al. The worldwide costs of dementia in 2019. Alzheimers Dement 2023; 19: 2865-2873. 5. World Health Organization (WHO). UN Decade of Healthy Ageing: Plan of Action 2021-2030. Available from: https://www.who.int/initiatives/decade-of-healthy-ageing 6. Fabbricotti IN, Janse B, Looman WM, et al. Integrated care for frail elderly compared to usual care: a study protocol of a quasi-experiment on the effects on the frail elderly, their caregivers, health professionals and health care costs. BMC Geriatr 2013; 13: 31 7. Dixon-Woods M, Kirk MD, Agarwal MS, et al. Vulnerable groups and access to health care: a critical interpretive review. National Coordinating Centre NHS Service Delivery Organ RD (NCCSDO), 2005. 8. Rich E, Lipson D, Libersky J, et al. Coordinating care for adults with complex care needs in the patient-centered medical home: challenges and solutions. In: White paper prepared by Mathematica Policy Research under Contract No. HHSA290200900019I/HHSA29032005T. AHRQ Publication No. 12–0010-EF. Rockville: Agency for Healthcare Research and Quality, 2012. 21 9. Gomes M, Murray E and Raftery J. Economic Evaluation of Digital Health Interventions: Methodological Issues and Recommendations for Practice. Pharmacoeconomics 2022; 40: 367378. 10. Leng M, Zhao Y, Xiao H, et al. Internet-based supportive interventions for family caregivers of people with dementia: systematic review and meta-analysis. J Med Internet Res 2020; 22: e19468. 11. Nickel F, Barth J and Kolominsky-Rabas PL. Health economic evaluations of nonpharmacological interventions for persons with dementia and their informal caregivers: a systematic review. BMC Geriatr 2018; 18: 69. 12. Pournik O, Ahmad B, Lim Choi Keung SN, et al. CAREPATH: Developing Digital Integrated Care Solutions for Multimorbid Patients with Dementia. Stud Health Technol Inform 2022; 295, 487-490. 13. Pournik O, Ahmad B, Lim Choi Keung SN, et al. Interoperable E-Health System Using Structural and Semantic Interoperability Approaches in CAREPATH. Stud Health Technol Inform 2023; 305, 608-611. 14. United Kingdom National Health Service Centre for Reviews and Dissemination. Systematic Reviews: CRD’s Guidance for Undertaking Reviews in Health Care. York: Centre for Reviews and Dissemination, University of York, 2009. 15. Goeree R and Diaby, V. Introduction to health economics and decision-making: Is economics relevant for the frontline clinician? Best Practice and Research: Clinical Gastroenterology 2013; 27, 831–844. 16. Drummond M, Sculpher M, Claxton K, et al. Methods for the economic evaluation of health care programmes (4th ed.). United Kingdom: Oxford University Press, 2015. 17. Moher, D., Shamseer, L., Clarke, M., et al. Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) 2015 statement. Systematic Reviews 2015; 4, 1. 22 18. Angeles RC, Berge LI, Gedde MH, et al. Which factors increase informal care hours and societal costs among caregivers of people with dementia? A systematic review of Resource Utilization in Dementia (RUD). Health Econ Rev 2021; 11, 37. 19. Clarkson P, Davies L, Jasper R, et al. Home Support in Dementia (HoSt-D) Programme Management Group. A Systematic Review of the Economic Evidence for Home Support Interventions in Dementia. Value Health 2017; 20, 1198-1209. 20. Goodman-Casanova J M, Guzmán-Parra J, Guerrero G, et al. TV-based assistive integrated service to support European adults living with mild dementia or mild cognitive impairment (TVAssistDem): study protocol for a multicentre randomized controlled trial. BMC geriatrics 2019; 19, 247. 21. Shaw C A, Williams K N, Lee R H, et al. Cost-effectiveness of a telehealth intervention for inhome dementia care support: Findings from the FamTechCare clinical trial. Research in nursing & health 2021; 44, 60–70. 22. Husebo B S, Allore H, Achterberg W, et al. [email protected] the clinical pathway for home-dwelling people with dementia and their caregivers: study protocol for a mixed-method, stepped-wedge, randomized controlled trial. Trials 2020; 21, 510. 23. Reuben D B, Gill T M, Stevens A, et al. D-CARE: The Dementia Care Study: Design of a Pragmatic Trial of the Effectiveness and Cost Effectiveness of Health System-Based Versus Community-Based Dementia Care Versus Usual Dementia Care. Journal of the American Geriatrics Society 2020; 68, 2492–2499. 24. Klein O A, Boekholt M, Afrin D, et al. Effectiveness of a digitally supported care management programme to reduce unmet needs of family caregivers of people with dementia: study protocol for a cluster randomised controlled trial (GAIN). Trials 2021; 22, 401. 23 25. Malmgren Fänge A, Schmidt S M, Nilsson M H, et al. The TECH@HOME study, a technological intervention to reduce caregiver burden for informal caregivers of people with dementia: study protocol for a randomized controlled trial. Trials 2017; 18, 63. 26. van Santen J, Dröes R M, Bosmans J E, et al. The (cost-) effectiveness of exergaming in people living with dementia and their informal caregivers: protocol for a randomized controlled trial. BMC geriatrics 2019; 19, 50. 27. Ponvel P, Shahar S, Singh D K A, et al. Multidomain Intervention for Reversal of Cognitive Frailty, Towards a Personalized Approach (AGELESS Trial): Study Design. Journal of Alzheimer's disease: JAD 2021; 82, 673–687. 28. Ghani Z, Saha S, Jarl J, et al. Short Term Economic Evaluation of the Digital Platform "Support, Monitoring and Reminder Technology for Mild Dementia" (SMART4MD) for People with Mild Cognitive Impairment and their Informal Caregivers. Journal of Alzheimer's disease: JAD 2022; 86, 1629–1641. 29. Blom M M, Bosmans J E, Cuijpers P, et al. Effectiveness and cost-effectiveness of an internet intervention for family caregivers of people with dementia: design of a randomized controlled trial. BMC psychiatry 2013; 13, 17. 30. Piera-Jiménez J, Daugbjerg S, Stafylas P, et al. BeyondSilos, a Telehealth-Enhanced Integrated Care Model in the Domiciliary Setting for Older Patients: Observational Prospective Cohort Study for Effectiveness and Cost-Effectiveness Assessments. JMIR medical informatics 2020; 8, e20938. 31. Garzón-Maldonado FJ, Gutiérrez-Bedmar M, Serrano-Castro V, et al. An assessment of telephone assistance systems for caregivers of patients with Alzheimer’s disease. Neurologia 2017; 32: 595-601. 24 32. Guisado-Fernandez E, Blake C, Mackey L, et al. A Smart Health Platform for Measuring Health and Well-Being Improvement in People with Dementia and their Informal Caregivers: Usability Study. JMIR aging 2020; 3, e15600. 33. Stephen C, Sultan H and Frew E. Valuing telecare using willingness to pay from the perspective of carers for people with dementia: a pilot study from the West Midlands. Journal of telemedicine and telecare 2014; 20, 141–146. 34. Chodosh J, Colaiaco BA, Connor KI, et al. Dementia Care Management in an Underserved Community: The Comparative Effectiveness of Two Different Approaches. J Aging Health 2015; 27, 864-893. 35. Milne H, van der Pol M, McCloughan L, et al. The use of global positional satellite location in dementia: a feasibility study for a randomised controlled trial. BMC psychiatry 2014; 14, 160. 36. Wan Y, Cai Y, Liao S, et al. Smartphone-based versus traditional face-to-face collaborative care for community-dwelling older adults living with dementia in China: protocol for an implementation science-based sequential multiple assignment randomised trial. BMJ Open 2023; 13, e067406. 37. Marsh K, Phillips CJ, Fordham R, et al. Estimating cost-effectiveness in public health: a summary of modelling and valuation methods. Health Economics Review 2012; 2, 17. 38. Caro JJ, Briggs AH, Siebert U, et al. Modeling good research practices—overview. Med Decis Mak 2012; 32, 667–677. 39 Philips Z, Bojke L, Sculpher M, et al. Good practice guidelines for decision-analytic modelling in health technology assessment. PharmacoEconomics 2006; 24, 355–71. 40. Guzzon A, Rebba V, Paccagnella O, et al.The value of supportive care: A systematic review of cost-effectiveness of non-pharmacological interventions for dementia. PLoS One 2023; 18, e0285305. 25 41. Nguyen KH, Comans TA, Green C. Where are we at with model-based economic evaluations of interventions for dementia? a systematic review and quality assessment. Int Psychogeriatr 2018; 30, 1593-1605. 42. Knapp M, Shehaj X, Wong G. Digital interventions for people with dementia and carers: effective, cost-effective and equitable?. Neurodegener Dis Manag 2022; 12, 215-219. 43. Li L, Nguyen K-H, Comans T, et al. Utility-based instruments for people with dementia: a systematic review and meta-regression analysis. Value Health 2018; 21, 471–481. 44. Keetharuth AD, Hussain H, Rowen D, et al. Assessing the psychometric performance of EQ5D-5L in dementia: a systematic review. Health Qual Life Outcomes 2022; 20, 139. 45. Lloyd H, Jenkinson C, Hadi M, et al. Patient reports of the outcomes of treatment: a structured review of approaches. Health Qual Life Outcomes R 2014; 12, 5. 46. Wolf JA, Niederhauser V, Marshburn D, et al. Defining patient experience. Patient Exp J 2014; 1, 7-19. 47. World Health Organisation. WHO Global Strategy on Integrated People-Centred Health Services 2016-2026: Executive Summary. WHO, Geneva, Switzerland, 2015. 48. National Collaboration on Integrated Care and Support. Integrated Care and Support: Our Shared Commitment. NHS England, England, UK, 2013. 49. The King’s Fund. Implementing the NHS Five Year Forward View: Aligning Policies with the Plan. NHS England, London, UK, 2015. 50. Yang F, Dawes P, Leroi I, et al. Measurement tools of resource use and quality of life in clinical trials for dementia or cognitive impairment interventions: A systematically conducted narrative review. Int J Geriatr Psychiatry 2018; 33, e166-e176. 51. Schulz KF, Altman DG, Moher D, et al. CONSORT 2010 statement: updated guidelines for reporting parallel group randomized trials. Ann Intern Med 2010; 152, 726-732. 32 included. During the intervention, caregivers are supported on-line by a psychologist (coach) who gives feedback on the exercises sent by the caregivers. schedule. Topics of the bulletin do not overlap with the content of ‘Mastery over Dementia’. PieraJiménez et al. 2020 30 Spain Observational Prospective Cohort and quasiexperimental Study Effectiveness (using the Model for Assessment of Telemedicine) and CostEffectiveness (using Monitoring and Assessment Framework for the European Innovation Partnership on Active and Healthy Ageing (MAFEIP)) 198 patients: Care recipients assessed for eligibility were involved in a domiciliary care program as described by BurgosDíez et al. (study condition): age ≥65 years, special health needs due to the presence of chronic diseases and the need for social care based on Barthel Index of Activities of Daily Living and Instrumental Activities of Daily Living. BeyondSilos Project: a TelehealthEnhanced Integrated Care Model in the Domiciliary Setting for Older Patients A basic health and social care coordination 3-state Markov model: (1) baseline disease stage (the patient remains in the same state or improves), (2) deteriorated disease stage (the patient worsens), and (3) death 8 months; 3% Health care and societal perspective Barthel index scale Instrumental Activities of Daily Living scales Geriatric Depression Scale (GDS) Corresponding trial ASSIST Tool GarzónMaldonado et al. 31 2017 Spain Prospective observational study To evaluate a Telephone Assistance System (TAS) for caregivers of patients with Alzheimer's disease (AD) from 2 points of 97 patients with a diagnosis of AD according to NINCDSADRDA criteria and their 97 On-site assistance Telephone assistance on demand NR 12 months; NR NR Cost/day of a Specialist Neurologist, and a liaison nurse or case manager nurse Estimated time invested in the Corresponding trial Corresponding trial 33 view: financially (Costs Analysis) and according to the level of satisfaction of the caregiver. informal caregivers. telephone consultation by the neurologist and by the liaison nurse or case manager nurse Travel costs by own means Level of satisfaction of the caregiver GuisadoFernandez et al. 32 2020 Ireland Longitudinal quantitative analysis To create a multidimensional profile for evaluating the well-being progression of the PwDinformal caregiver dyad for a year during their use of a connected health (CH) platform, designed for monitoring PwD and supporting their informal caregivers at home, and to conduct a longterm follow-up using the proposed wellbeing profile at different timeinterval evaluations. A total of 11 PwD–informal caregiver dyads were recruited Connected HEalth Sustaining homestay (CHESS): CH platform works on a tablet (Samsung Galaxy Tab A 10.1, 2016) and is connected to a series of PwD monitoring devices for home use, including a blood pressure (BP) monitor (Omron M6 by OMRON Healthcare Ltd), an electronic weighing scale (Withings, France), and an activity and sleeping tracker (Withings Go). No comparator NR 0, 3, 6, 9,12 months; NR NR PwD: Functional status: Disability Assessment Dementia (DAD) scale Cognitive status: Mini-Mental State Examination (MMSE) QoL: self-reported Dementia Quality of Life (DEMQoL) scale and DEMQoL Proxy (PwD’s caregiver-reported QoL) Informal caregiver: Mental health: Hospital Anxiety and Depression Scale (HADS) Sleep quality: Pittsburgh Sleep Quality Index (PSQI) Caregiver burden: Zarit Burden Interview (ZBI) scale Corresponding trial No cost analysis 34 Stephen et al. 2014 33 UK A crosssectional study (STUDY PROTOCOL) A cost-benefit analysis of the STAY telecare service based in the West Midlands. 47 carers from Sandwell Council’s database and 40 carers from the BUDS charity database. A total of 34 completed questionnaires were returned, from 21 carers (45%) from the council database and 13 carers (33%) from the charity database. NR NR NR NR Perspective of carers of people with dementia. Details about what telecare service they had previously Received, using tick-box options for each potential product. Willing to spend on telecare Corresponding study Corresponding study Chodosh et al. 2015 34 US NR Compare the effectiveness and costs of a telephone-only approach to inperson plus telephone for delivering an evidence-based, coordinated care management program for dementia 151 patient– caregiver dyads from an underserved predominantly Latino community Use of care management protocol (ACCESS project) implemented in different formats: in-person visits at home and/or in the community plus telephone and mail, versus telephone and mail only. Use of care management protocol (ACCESS project) implemented in different formats: telephone and mail only. NR 12 months; NR NR Caregiver Burden: Zarit Burden Interview Care-recipient memory and problem behaviors: the Revised Memory and Behavior Problem Checklist (RMBPC) Corresponding trial Literature (assumptions) Milne et al. 2014 35 Scotland A feasibility study for a randomized, controlled trial (RCT). An observational study using mixed methods, combining data from qualitative interviews and focus groups with questionnaire and resource use data. To determine the: -likely available number of eligible patients, caregivers or other appropriate participants and the willingness of participants to be recruited and of social care staff to identify and recruit participants 20 people with dementia (due to any cause) who met the local criteria for the GPS service and were willing to use it. Global Positional Satellite (GPS) devices Standard care NR 6 months; NR NR Modified Caregiver Strain Index (mCSI) Hospital Anxiety and Depression Score (HADS) for caregiver Mini Mental State Examination (MMSE) for patients Barthel Score for patients Corresponding trial Corresponding trial 35 -perceptions of participants as to what constitutes problematic wandering behavior/getting lost, the perceived utility of GPS location and which types of people might benefit most from the intervention -rates of retention, compliance, and completion of study questionnaires -availability, usefulness and limitations of routinely acquired health service and social care data to assess outcomes - standard deviation of outcome measures to inform estimates of sample size for a future trial - time needed to collect and analyze data. Index of Capability for older people for both caregiver and person with dementia (ICECAP-O) Carer Experience Scale (CES) adverse events: injuries and falls, out-of-hours calls, emergency room… health and social care resource use (i.e., general practitioners, hospital out-patient wards and in-patient episodes, emergency room and out-of-hours visits, social care visits, and admissions to care homes) Quality of life: ICECAP-O capability index and the Carer Experience Scale Wan et al. 2023 36 China A sequential multiple assignment randomized trial (SMART) (STUDY PROTOCOL) To (1) develop a Collaborative Care Model (CCM) for people living with dementia and their caregivers in urban communities in Chengdu; (2) This study will collaborate with community healthcare centers in Chengdu to recruit communitydwelling Smartphonebased collaborative care for communitydwelling older adults living with dementia Traditional facetoface collaborative care for communitydwelling older adults living with dementia NR 6, 12 and 18 months; NR Societal perspective Quality of life: Quality of Life in Alzheimer’s Disease Patient Caregiver burden: Zarit Burden Interview Corresponding trial Corresponding trial 36 identify the most effective strategy to deliver collaborative care, including delivery channel and delivery frequency, using the sequential multiple assignment randomized trial (SMART); and (3) evaluate the feasibility of implementation and costeffectiveness of the CCM under the REAIM framework. people living with dementia and their caregivers. The total sample size is 286 pairs of participants. We further assumed a dropout rate of 20% in the follow-ups. Therefore, 358 pairs of participants will be recruited. 37 TABLE 3 Health and social outcomes and costs measured in the reviewed studies Health outcomes Measures PwD Cognitive impairment, cognitive function, Dementia Neuropsychiatric Inventory, 12-item version with caregiver distress (NPI-12) [22] Neuropsychiatric Inventory Questionnaire (NPI-Q) Severity [23] [26] Montreal Cognitive Assessment (MoCA) [23] Goal Attainment Scale (GAS) [23] Global Deterioration Scale (GDS) [25] The Revised Memory and Behavior Problem Checklist (RMBPC) [29] [34] Trail Making Test (TMT) [26] Modified Neuropsychological Tests Battery (mNTB) [27] Mini-Mental State Examination (MMSE) [20] [22] [25] [26] [27] [28] [32] [34] Clinical Dementia Rating (CDR) [27] Cohen-Mansfield Agitation Inventory (CMAI) [22] Blessed Roth Dementia Scale [34] Quality of Life EQ-5D-5L [20] [22] [24] [26] [35] EQ-5D-5L-P (Proxy) EQ-5D-3L [25] [28] Quality of Life in Alzheimer’s disease scale (QoL-AD) [20] [22] [23] [25] [28] Dementia Quality of Life utility measure (DEMQOL-U or DEMQOL-P-U) [32] Caregiver quality of life (The Caregiver Quality of Life instrument (CGQOL) [34] Health Utilities Index (HUI) [34] EQ-5D+C (‘cognition’ as a sixth dimension) [29] Quality of Life in Alzheimer’s Disease Patient [36] Dependence, Functional level for instrumental and personal activities Index of Capability for older people for both caregiver and person with dementia (ICECAPO) [35] Barthel Index of Activities of Daily Living [30] [35] Lawton Instrumental Activities of Daily Living (I-ADL) Scale [20] [22] [25] [30] Brody Personal Activities of Daily Living (P-ADL) Scale [22] Functional Activities Questionnaire (FAQ) [23] Functional Assessment Staging Test (FAST) [22] [31] BRIEF-Cope [27] De Jong Gierveld Loneliness Scale [27] Resilience Scale [27] General Self-Efficacy Scale (GSES) [27] University of Rhode Island Change Assessment (URICA) Psychotherapy version [27] Disability Assessment Dementia (DAD) scale [32] Short Physical Performance Battery (SPPB) [26] Physical Activity Scale of the Elderly (PASE) [26] Intrinsic Motivation Inventory (IMI) [26] Physical fitness: Fried Frailty Criteria [27] 2-min Step Test [27] Grip Strength Test [27] Chair Stand Test [27] Chair Sit-and-Reach Test [27] Timed Up-and-Go Test [27] Back Scratch Test [27] Exercise Self-Efficacy Scale (ESES) [27] Maximum oxygen uptake (VO2max) [27] Comorbidity in dementia General Medical Health Rating Scale (GMRH) [22] 6-item Falls Efficacy Scale-International (FES-I) [25] Charlson Comorbidity Index [30] Pain in dementia Mobilization–Observation–Behavior–Intensity Dementia Pain Scale (MOBID-2) [22] Clinical Meaningful change Clinical Global Impression of Change (CGIC) [22] Depression Patient Health Questionnaire-8 [23] Geriatric Depression Scale (GDS-15) [20] [22] [30] Cornell Scale for Depression in Dementia (CSDD) [22] Number of unmet needs The Camberwell Assessment of Need for the Elderly (CANE) [24] Social functioning Adult Social Care Outcomes Toolkit (ASCOT) [26] General The Older Persons and Informal Caregivers Survey Minimum DataSet (TOPICS-MDS care recipient): contains information on the physical and mental health and wellbeing of older persons and informal caregivers [26] Caregiver Anxiety and Depression Center for Epidemiologic Studies Depression Scale (CES-D) [21] [29] The Patient Health Questionnaire-9 items (PHQ-9) [34] Hospital Anxiety and Depression Scale (HADS) [25] [29] [32] [35] Competence The Short Sense of Competence Questionnaire (SSCQ) [21] [26] [29] Distress Relative Stress Scale (RSS) [22] The Dutch version of the Revised Memory and Behavioral Problem Checklist (RMBPC) [29] [34] Severity of caregiver strain Modified Caregiver Strain Index (MCSI) [23] [35] Burden Dementia Burden Scale-Caregiver (DBS-CG) [23] Zarit-Burden Interview (ZBI-7) [24] [36] 38 Zarit Burden Interview (ZBI-12) [20] [25] [28] [32] [34] Self-Perceived Pressure from Informal Care scale (SPPIC) [29] Experience Positive Aspects of Caregiving (PAC) [23] Positive care experiences of the informal Caregiver - Scale of Positive Experiences (Positieve Ervaringen Schaal (PES)) [26] Carer Experience Scale (CES) [35] Caregiver Satisfaction [23] Wellness Pittsburgh Sleep Quality Index (PSQI) [32] Self-efficacy Caregiver Self-Efficacy scale [23] Costs Measures Social support Lubben Social Network Scale (LSNS-6) [24] Medical Outcome Social Support (MOSS) [27] Resource use in general Resource Utilization in Dementia (RUD) Lite instrument [22] [24] [25] Resource Utilization in Dementia-Formal Care (RUD-FOCA) [22] The Questionnaire for the Use of Medical and Non-Medical Services in Old Age-FIMA [24] The older persons and informal caregivers survey minimum DataSet (TOPIC-MDS) [26] The Client Service Receipt Inventory (CSRI) [20] iMTA Questionnaires on Costs Associated with Psychiatric Illness (TIC-P) [29] Polypharmacy and treatment burden, adherence and compliance The Medication Assessment Questionnaire [20] Multiple appointments Cost/day of neurologist and a liaison nurse or case manager nurse [33] Estimated time invested in the telephone consultation by neurologist and liaison nurse or case manager nurse [33] Unplanned care Adverse events: out-of-hours calls, emergency room, injuries, falls… [26] Coordination of care across services Others Measures Usability of technology System Usability Scale (SUS) [20] Implementation aspects Measurement Instrument for Determinants of Innovations (MIDI) [26] Note: most commonly-used scales and/or questionnaires in bold 39 TABLE 4A Data synthesis: components of interventions Components Studies [reference] Primarily for PwD Location (electronic location devices to support safe walking) [35] Relationships [29] Loneliness [29] Relaxation [29] Motivation [29] [30] Socialization [29] Medication use [28] Health monitoring (blood pressure, pulse, glucose, weight, etc.) [20] [32] Cognitive stimulation [20] [27] [28] [32] Reminiscences [20] Healthcare education [22] Physical functioning (falls prevention) [26-27] [32] Daily living activities [22] [30] Continuing support (collaborative consultation) [23] [34] [28] [36] Primarily for Caregiver Psychoeducation [29] Cognitive behavioral therapy [29] Problem solving therapy or care recommendations [21] [29] Assertiveness training [29] Relaxation [29] Alerting about changes and warning [33] Consultation [31] Monitoring adverse and dangerous events [25] Educational section offering advice about daily care [22] [32] Volunteer support from nonprofit organizations [22] Care support (needs assessment) [24] [36] TABLE 4B Data synthesis: devices used for interventions Devices Studies [reference] FamTechCare telehealth intervention (video-monitoring unit: iPad Mini with Behavior Capture videorecording application, Bluetooth remote, and iPad stand; plus telephone) [21] [email protected] (technical aids, cognitive intervention devices and assisted living systems/smart house systems) [22] Health systems-based dementia care (HSDC) and Community-based dementia care (CBDC) [23] CMD – Care management program (tablet PC-based care management system) [24] Telehealth-Enhanced Integrated (social and health) Care Model (BeyondSilos): security sensors (e.g., fire and water detectors, behavioral movement sensors, and a cell phone with GPS tracking and fall detection), medical devices (e.g., weight scale, blood pressure meter, glucometer, or oximeter), serious games, a personal diary, and a videoconferencing system) [30] TV-based assistive integrated service (TV-AssistDem) (Android software) [20] Telephone calls and mailings (ACCESS) [34] Telephone assistance system [31] Smart Health Platform: Connected HEalth Sustaining home-Stay (CHESS) (tablet computer, monitoring devices for home use including a blood pressure monitor, an electronic weighing scale, activity and sleeping tracker) [32] Technological home monitoring kit: control unit and set of sensors that notify caregivers via their phones (homeleaving sensors, bed-occupancy sensors, smoke and water leak sensors, automatic lights, and other interactive functions) [25] Interactive cycling [26] 40 AGELESS (telerehabilitation approach) [27] GPS location systems [35] Digital Platform “Support, Monitoring and Reminder Technology for Mild Dementia” (SMART4MD) (health app for tablets) [28] Telecare support service (STAY) (bed-leaving sensors, smoke alarms, heat sensors) [33] Internet-based [29] WeChat mini program [36] 41 TABLE 5 Types of interventions compared using a full economic evaluation (3 papers; 9 comparisons in terms of efficiency) Intervention Comparator Incremental effect Incremental cost ICER Conclusions* References FamTechCare telehealth intervention Telephone support intervention For caregivers: -2.24 (depression – CES-D change score)a $41.47 per dyad per week $18.51 per dyad per week to achieve a significant improvement in depression in the FamTechCare caregivers Cost-effective Shaw et al. 2021 [21] 1.24 (competence – SSCQ change score)b $41.47 per dyad per week $36.38 per dyad per week to achieve a significant improvement in competence in the FamTechCare caregivers Cost-effective BeyondSilos, a TelehealthEnhanced Integrated Care Model in the Domiciliary Setting Usual cared 0.731 QALYs €4755 €6505.52 per QALY gained Cost-effective Piera-Jimenez et al. 2020 [30] SMART4MD App Standard care For PwMCI: –0.00358 QALYs (IC95%: – 0.009 to 0.002) For PwMCI: €12 (IC95%: –2090.33 to 2115.28) For PwMCI: Dominated For PwMCI: Dominated Ghani et al. 2022 [28] 0.3322 (IC95%: –0.42 to 1.08) (adjusted QoL-AD) €12 (IC95%: –2090.33 to 2115.28) €36 (adjusted QoL-AD) Cost-effective 0.2100 (IC95%: –0.12 to 0.54) (MMSE adjusted) €12 (IC95%: –2090.33 to 2115.28) €57 (MMSE adjusted) Cost-effective For informal caregivers: 0.0028 QALYs (IC95%: –0.002 to 0.008) For informal caregivers: –€ 539 (-2623.78 to 1545.42) For informal caregivers: €-192500 per QALY gained For informal caregivers: Dominant (based on QALYs) 0.23 (IC95%: –0.72 to 1.18) (ZBI adjusted) –€ 539 (-2623.78 to 1545.42) €-2343.48 (ZBI adjusted) Dominant (based on ZBI adjusted)