https://orcid.org/0000-0003-4685-1890
Figures
Abstract
Background
Almost 44 million people are currently living with dementia worldwide. This number is set to increase threefold by 2050, posing a serious threat to the sustainability of healthcare systems. Overuse of antipsychotic drugs for the management of the symptoms of dementia carries negative consequences for patients while also increasing the health expenditures for society. Supportive care (SC) interventions could be considered a safer and potentially cost-saving option. In this paper we provide a systematic review of the existing evidence regarding the cost-effectiveness and cost-utility of SC interventions targeted towards persons living with dementia and their caregivers.
Methods
A systematic literature review was performed between February 2019 and December 2021 through searches of the databases PubMed (MEDLINE), Cochrane Library, CENTRAL, Embase and PsycINFO. The search strategy was based on PRISMA 2020 recommendations. We considered studies published through December 2021 with no lower date limit. We distinguished between five categories of SC strategies: cognitive therapies, physical activity, indirect strategies (organisational and environmental changes), interventions primarily targeted towards family caregivers, and multicomponent interventions.
Results
Of the 5,479 articles retrieved, 39 met the inclusion criteria. These studies analysed 35 SC programmes located at different stages of the dementia care pathway. Eleven studies provided evidence of high cost-effectiveness for seven interventions: two multicomponent interventions; two indirect interventions; two interventions aimed at caregivers of community-dwelling persons with dementia; one community-based cognitive stimulation and occupational programme.
Conclusion
We find that the most promising SC strategies in terms of cost-effectiveness are multicomponent interventions (targeted towards both nursing home residents and day-care service users), indirect strategies (group living and dementia care management at home), some forms of tailored occupational therapy, together with some psychosocial interventions for caregivers of community-dwelling persons with dementia. Our results suggest that the adoption of effective SC interventions may increase the economic sustainability of dementia care.
Citation: Guzzon A, Rebba V, Paccagnella O, Rigon M, Boniolo G (2023) The value of supportive care: A systematic review of cost-effectiveness of non-pharmacological interventions for dementia. PLoS ONE 18(5): e0285305. https://doi.org/10.1371/journal.pone.0285305
Editor: Kamal Gholipour, Tabriz University of Medical Sciences, IR Iran, ISLAMIC REPUBLIC OF IRAN
Received: February 28, 2021; Accepted: April 20, 2023; Published: May 12, 2023
Copyright: © 2023 Guzzon et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Data Availability: All relevant data are within the manuscript and its Supporting information files.
Funding: This study has been carried out within the research project “Supportive care for elderly people with severe cognitive impairment” sponsored by the OIC (Opera Immacolata Concezione) Foundation, Padova, Italy.
Competing interests: The authors have declared that no competing interests exist.
Introduction
Globally, about 43.8 million people were living with dementia in 2016 [1], and this number is projected to triple by 2050 [2]. The worldwide costs of Alzheimer’s disease, the most prevalent subtype of dementia, and other dementias were estimated at US$818 billion in 2015 (equivalent to 1.09% of the global gross domestic product); these costs overcame the threshold of US$1 trillion in 2018 and are projected to double by 2030 [1, 3]. The substantial increase in the prevalence of dementia, mostly driven by demographic trends, poses significant challenges to health and social care systems, especially in terms of economic sustainability [3–6]. The scale of the problem becomes even greater when we take into account the indirect costs related to caregivers of persons with dementia (PwDs). Caregiving exacts a toll on caregivers’ mental, emotional, physical, and financial health [7]; caregivers are twice as likely to suffer from depression [8], they use more medication and make more doctor visits [9, 10], they save less money, and up to 9% of caregivers need to quit their jobs [11].
Current drug therapies for dementia (cholinesterase inhibitors and memantine) has been shown to have a small effect on cognition. However, these medications do not significantly change the course of illness and may provoke side effects [2, 12, 13], while only a limited number of ongoing clinical trials are currently investigating the viability of drugs directed at diverse therapeutic targets [13–15]. Available medications for treating PwDs have been shown to enhance the quality of life (QoL) for both the patient and caregiver when prescribed at the appropriate time during illness. In particular, cholinesterase inhibitors are more cost-effective than placebo and probably also cost-saving (by delaying the onset of institutionalisation), while the evidence in support of the cost-effectiveness of combination therapy (a cholinesterase inhibitor plus memantine) is less clear [16, 17]. A new drug, aducanumab, has been recently approved by the US Food and Drug Administration for treatment of Alzheimer disease, even though it is uncertain whether it works at all or provides sufficient benefits to outweigh its harms [18]. While there are uncertainties relating to the cost-effectiveness of some of the drugs for dementia, there are potentially serious risks associated with using antipsychotic medications to treat the behavioural and psychological symptoms of dementia (BPSD) [19–21]; moreover, there is no clinical or economic case for using antidepressant drugs to treat people with Alzheimer’s disease who have comorbid depression [17]. A problem with antipsychotic medications is that these drugs do not offer a sufficient benefit relative to the risks they pose [22]. For example, a UK study shows that patients who received an antipsychotic treatment for 12 months were significantly more likely to have died by the 24-month and 36-month follow-up periods compared to patients who had received a placebo [23], while other studies have found a link between the use of antipsychotic drugs in dementia patients and an increase in the risk of acute pulmonary diseases, hip fracture, thromboembolism, and stroke [24–27]. Despite this evidence in conjunction with the warnings and best practice guidelines that have followed [19–21], the use of antipsychotic drugs is still widespread.
Considering the setbacks suffered in the research on viable pharmacological treatments to counteract the progression of different types of dementia [12–15], the serious risks associated with using antipsychotic medications [19–21], and the high costs of overprescribing anti-dementia drugs [28], the development of effective non-pharmacological interventions to integrate or substitute the use of medications is of particular importance to increase both the effectiveness and the economic sustainability of dementia care.
Given this background, resource allocation could be enhanced by shifting from a standard approach—focused on containing the impact of distressing symptoms on patients through medications—to a more comprehensive approach based on the notion of person-centred care. This new approach would also follow the patient throughout the whole course of the disease by providing personalised care as well as support to patients and families. Defined as supportive care [29] and representing “a full mixture of biomedical dementia care, with good quality, person-centred, psychosocial, and spiritual care,” this approach must be extended throughout the course of the illness to guarantee the overall wellbeing of PwDs and their caregivers [30]. The term supportive care (SC) refers to a wide array of non-pharmacological interventions that encompass a broad and growing range of services that are delivered either to the patient, the caregiver, or the patient-caregiver dyad [31, 32]. This approach has been previously experimented in cancer care for addressing the clinical and psychosocial needs of patients in order to provide optimal quality of life [33] and in end-of-life care for non-cancer patients [34]. One of its key aspects is the decreasing reliance on medications that do not offer a sufficient benefit relative to the risks they pose in favour of novel non-pharmaceutical interventions [35]. In the case of PwDs, SC is characterised by the continuous assistance of patients and their relatives from diagnosis until death, a holistic and interdisciplinary approach to care, and a high level of flexibility in choosing the right care practices for each case [36]. It is therefore evident that this definition of SC should not be confused with the one sometimes adopted in the cost-effectiveness literature, where the term “best supportive care” is used to denote care as usual or non-intervention.
A key feature of SC in all its stages is the central role of both formal and informal caregivers: the former are meant to have in-depth knowledge and competencies to deal with dementia patients, while the latter need to be recognised as indispensable players in dementia care, and both need to form and maintain collaborative relationships to guarantee high-quality care to patients [37]. In particular, support to caregivers could be considered a win-win solution, as it is beneficial for carers, patients, and the sustainability of healthcare systems [22]. On the opposite side of the spectrum, the unregulated use—and sometimes abuse—of antipsychotic drugs is a no-win situation, as it is detrimental for the health of the patient, and it puts a strain on the budget of healthcare systems [35]. In particular, regarding the management of behavioural problems in Alzheimer’s disease, Gauthier et al. [38] suggest that non-pharmacological interventions (including psychosocial/psychological counselling as well as interpersonal and environmental management) should be attempted first, followed by the least harmful medication for the shortest time possible [38].
A unified classification of SC interventions for dementia does not exist yet, but several taxonomies can be found in the literature. For instance, Cammisuli et al. distinguish between holistic techniques, brief psychotherapy, cognitive methods, and alternative strategies [39]; in contrast, D’Onofrio et al. distinguish between cognitive and emotion-oriented interventions, sensory and multi-sensory stimulation interventions, and other interventions [40]. The World Alzheimer Report 2011 [41] and Nickel et al. [42] classified non-pharmacological interventions into four categories: physical exercise, interventions to support and enhance cognitive abilities, psychological and behavioural therapies, and occupational therapy. Our analysis will consider five categories of SC interventions for dementia, as we will explain in next section.
Over the last few years, the evidence base on the effectiveness of non-pharmacological interventions (in terms of cognitive functioning and the reduction of behavioural symptoms of PwDs) has grown considerably [31, 32]. Conversely, evidence on the value for money of non-pharmacological and SC interventions for PwDs and their caregivers is still scant, despite the growing need for healthcare systems to base resource allocation decisions on cost-effective intervention strategies.
In this paper, we provide a systematic review of the main evidence on the cost-effectiveness of five different categories of non-pharmacological and supportive practices for dementia. Our analysis aims at identifying which types of SC intervention have shown the strongest evidence of cost-effectiveness in order to provide useful information for the design of policies which may increase the economic sustainability of dementia care. Previous systematic reviews have highlighted the scarcity of economic evidence on non-pharmacological interventions for PwDs [43, 44] and their caregivers [45]. These reviews were focused mainly on interventions for community-dwelling persons with mild to moderate dementia [42] and home support interventions [46]. In contrast, we try to offer an all-encompassing review of cost-effectiveness studies on non-pharmacological and psychosocial interventions that target PwDs, their caregivers (either formal or informal), or the patient-caregiver dyad, and which are located at different stages of the care pathway for dementia and in different settings. Moreover, our systematic review is more complete since it provides evidence on the cost-effectiveness of indirect interventions such as organisational changes and innovations in the delivery of care and support.
Materials and methods
Categories of supportive care interventions
In this paper, we distinguish between five categories of non-pharmacological/SC strategies: 1) cognitive therapies; 2) physical activity interventions; 3) indirect strategies; 4) interventions primarily targeted towards caregivers; 5) and multicomponent interventions.
The main characteristics of the interventions considered are described in Table 1.
Search strategy and criteria for inclusion
A systematic literature review was performed between February 2019 and December 2021 on the healthcare electronic databases MEDLINE (PubMed), CDSR (Cochrane Database of Systematic Reviews), CENTRAL (Cochrane Central Register of Controlled Trials), Embase and PsycINFO. The search terms used to identify the articles to include in the review were as follows: (dementia OR alzheimer* OR cognitive) AND (’cost-effectiveness’/exp OR ’cost-analysis’/exp OR ’cost-utility’/exp) AND (’non-pharmacological’/exp OR psychosocial* OR ’drug-free’/exp). We considered studies published through December 2021 with no lower date limit. Additional details on the electronic search strategy can be found in the S1 File.
Study eligibility was based on the following criteria:
- Studies evaluating non-pharmacological dementia interventions;
- Interventions aimed at either the patient or the caregiver (or the dyad patient-caregiver);
- The participants in the study had a diagnosis of dementia or were caregivers of a person with diagnosed dementia;
- Research conducted as randomised controlled trial (RCT) or prospective cohort study;
- Studies with a complete economic analysis including an economic evaluation (a cost-effectiveness analysis and/or cost-utility analysis) or at least comprehensive information on outcomes and costs [85];
- Studies with an abstract in English.
We also considered the following exclusion criteria:
- Studies on the ageing population with no explicit focus on dementia;
- Research conducted as retrospective study;
- Studies which did not show a complete economic analysis upon full text screening;
- Studies with no available abstract in English.
The search strategy and the following review are both based on PRISMA 2020 recommendations [86]. The outcomes of the study selection process are described in the Results section. No prespecified protocol was followed for this systematic review.
Data collection and analysis
Data extraction was performed according to the guidelines of the Centre for Reviews and Dissemination for reviews of economic evaluations [87]. After the removal of duplicate citations using Endnote X9, the titles and abstracts of the remaining articles were initially screened by the principal reviewer (AG). After the initial screening, the principal reviewer (AG) evaluated the abstracts of the remaining publications and applied the eligibility criteria with another member of the research team (VR). Two members of the research team (AG and VR) performed the full-text screening on the publications that met the eligibility criteria. Information was collected on the type of economic evaluation, study objective, study design, description of the intervention, comparators, measures of benefit and cost, and outcome and cost results. Summary results were independently presented to the other members of the research team (OP, MR, and GB) to solve any disagreement through discussion or consultation.
The high heterogeneity in terms of interventions and outcome measures of the studies evaluated made it impossible to perform a meta-analysis, so we proceeded with a qualitative analysis.
Quality appraisal of included studies
The methodological quality of the studies included in the review was assessed using the Consensus on Health Economic Criteria (CHEC) checklist [88], which consists of 19 yes-or-no questions. To each study, we assigned a score from 0 to 19 based on the number of questions that the assessor answered with a “yes”. Studies were classified as being high-quality if the score was equal to or higher than 17, medium-quality studies were those with a score between 14 and 16, and low-quality studies were those which scored 13 or lower. The score also reflects the information contained in additional analyses for those that actively pointed to other articles for additional information on the study design and/or protocol. The principal reviewer (AG) assessed the quality of all the articles, and the other four members of the research team (VR, OP, MR, and GB) checked for accuracy within their subsets. Any disagreement was resolved through discussion or consultation. The quality appraisal was undertaken to aid in interpreting the findings and determining the strength of the conclusions drawn; no study was excluded based on the results of the quality assessment.
Main outcomes of supportive care interventions
As mentioned above, the outcome indicators considered in the studies included in the review were highly heterogeneous even though it is possible to broadly distinguish between patient and caregiver outcome measures.
The main patient outcomes considered in the analysed studies were the following:
- Quality of life–the cognitive and functional decline brought about by dementia has a huge impact on the patient’s QoL, and most studies include both generic health-related QoL (HRQoL) and dementia-related QoL as outcomes [89];
- Cognitive impairment–dementia impacts short- and long-term memory but also other cognitive functions such as language, abstract thinking, and judgement [90];
- Dementia severity–the gradual progression of the disease is measured with staging instruments that monitor the clinical and cognitive deterioration caused by dementia [91];
- Behavioural and psychological symptoms of dementia (BPSD)–these neuropsychiatric disturbances, such as apathy or hallucinations (or other non-cognitive symptoms), constitute a major component of dementia and have an impact on the QoL [92];
- General health–this variable is gauged by looking at comorbidities, adverse events (i.e., untoward medical occurrences in a patient, including falls and fractures), nutritional status, etc. [93, 94];
- Mental health–this variable can be measured by looking at an individual’s depression levels, anxiety levels, schizophrenic or psychotic episodes, etc. [95];
- Agitation–as one of the most commonly observed neuropsychiatric symptoms in patients suffering from dementia, this condition is described as restless behaviour or improper physical and/or verbal action that can be a source of trouble for others [96];
- ADLs and IADLs–the number of (instrumental) activities of daily living an individual is able to carry out in an accepted way is a measure of functional capacity, which is an important indicator of health in the elderly [97];
- Prescription drug use–the use of antipsychotic medications to treat the BPSD;
- Service utilisation (and related costs)–the extent to which dementia patients use medical and/or social services and resources [98], including institutionalisation [99].
For each patient outcome, Table 2 reports the correlate measures considered in the selected studies.
The main caregiver outcomes considered in the reviewed studies were the following (see Table 3 for details on the different measures for each outcome):
- Quality of life–dementia severely impacts the QoL of caregivers because caring for someone who suffers from dementia is extremely burdensome and contributes to physical and psychiatric illnesses [100];
- Burnout and burden–caregiver burden is the perceived negative effect of caring for a family member [101], while caregiver burnout is more specifically a state of physical, emotional, and mental exhaustion [102];
- Sense of competence and mastery–competence is the extent to which a caregiver feels he or she can effectively do what is needed for a patient, whereas mastery is the extent to which a caregiver feels in control of the situation; both have been linked to positive outcomes for the caregiver [103, 104];
- General health–caregivers are more likely to report poor health because they have less time to take care of themselves and face substantial stress (as indicated by the increased levels of cortisol) [105, 106];
- Mental health–depression is very common among dementia caregivers, as are sleep disturbances, loneliness, and social isolation [107, 108];
- Quality of interaction with the patient–low-quality interactions can undermine both the caregiver’s QoL and quality of care [109]; the quality of the relationship that occurs between the caregiver and the patient has been found to be predictive of outcomes like the patient’s institutionalisation and functional decline [110, 111];
- Coping strategies–coping strategies employed by caregivers, such as avoidance or wishful thinking, are linked to physical and mental health outcomes [112];
- Time spent caregiving–caring for a PwD is not only a burdensome task, but it is also time consuming, as it prevents informal caregivers from having a regular work-life balance [113];
- Service utilisation–the additional medical and social service use by caregivers themselves helps us better understand the impact dementia has on societal costs [114];
- Absenteeism–formal and informal dementia caregivers are more likely to have higher absenteeism rates [115].
Results
Study selection
The systematic search identified 5,479 publications. Duplicate citations were removed using Endnote X9, resulting in a total of 1,362 publications. After an initial screening of the titles and abstracts, 229 publications remained. After applying the eligibility criteria, 55 publications remained for full-text screening. Of these, 16 articles were excluded due to specific issues (e.g., 7 articles were excluded since they did not report a complete economic analysis upon a full-text screening). A final sample of 39 studies remained for inclusion in the review, including a study in the Dutch language [79]. The search strategy, based on PRISMA recommendations [86], is shown in the flow chart in Fig 1.
Characteristics of the included studies
We reviewed 39 studies that analysed 35 interventions: nine cognitive stimulation and occupational programmes primarily targeted at PwDs; three physical activity interventions; ten indirect interventions (organisational and environmental changes); seven interventions primarily targeted towards family carers; and six structured multicomponent interventions. The number of reviewed study (39) is higher than the number of analysed interventions (35) since some studies considered the same intervention but with a different timeframe or a different set of outcome measures: two studies [63, 116] focused the same intervention of dementia care management (Delphi-MV trial); three studies [71, 117, 118] considered the same individual therapy program for caregivers “Strategies for Relatives” (START); two studies [77, 78] considered the multicomponent WHELD (“Improving Wellbeing and Health for People with Dementia”) intervention. The main characteristics of the studies are summarised in Tables 4–8.
The interventions analysed in the studies were located at different stages of the care pathway for dementia: 14 studies focused on patients with dementia in its mild to moderate stages and/or their caregivers [63, 66, 72, 80, 83, 116, 119–126]; 19 studies addressed the moderate-to-severe stages [65, 67, 69–71, 73, 77–79, 84, 117, 118, 127–133]; and six studies focused on PwDs at different stages [62, 68, 76, 81, 134, 135]. Eight studies considered SC programmes in nursing homes and assisted living settings [65, 77, 78, 84, 128–130, 133], and two studies analysed both residential and community settings [122, 134], while the rest of the health economic analyses concerned community-based interventions. Most studies analysed SC interventions directed at patient-caregiver dyads, while nine studies focused on specific programmes supporting informal caregivers of community-dwelling PwDs [68, 69, 71–73, 76, 117, 131, 132].
Most studies (31 out of 39, and 28 out of 35 interventions) were conducted in European countries with comparable underlying health and social care systems (16 were based in the UK), while only seven studies were developed in other OECD countries, including four in the United States, one in Japan, one in Australia, and one in Canada.
The studies were quite heterogeneous in terms of their design, the cost items included, and the choice of outcome measures. Most studies (n = 34) used a randomised controlled trial (RCT) design, while five were non-randomised comparisons through prospective matched controlled trials [62, 67, 70, 128, 131].
Regarding the cost components considered, most of the studies–with few exceptions [76, 81, 127, 131]–identified all relevant costs for each alternative on the basis of a complete analysis of health and social care resource utilisation (medical outpatient and inpatient treatments, medications, medical aids, home care, day-care and nursing home care services, etc.). 18 of the analysed studies adopted a narrow perspective when measuring costs, looking only at health and social care, while the other 21 studies considered a broader societal perspective, including the opportunity costs of caregivers’ inputs and the impacts of caring on their own health and wellbeing. In the latter studies, informal care time of family caregivers has generally been evaluated considering the average opportunity cost for lost production or leisure time and average gross wage plus non-wage labour cost (proxy good approach). Many studies have considered the variation in the use of health and social services not only as components for the calculation of costs but also as an outcome element in order to assess whether the analysed intervention was effective (compared to the comparator) in reducing the use of social and health services or in limiting the consumption of drugs. However, only five studies, focused on nursing home PwDs, have investigated the ability of SC interventions to reduce the use of psychotropic drugs [129, 130], and in particular antipsychotics [65, 77, 78].
In all the analysed studies, the SC intervention under investigation was explicitly compared—with regard to costs and outcome measures—with one or more alternatives (in most cases, the “usual care” alternative). Most studies, except for three [81, 128, 130], reported the incremental cost-effectiveness ratio (ICER) of SC interventions. When the outcome is measured in terms of utility values to account for the patient’s and/or carer’s QoL (e.g., using the Quality Adjusted Life Years (QALYs) gained), the cost-effectiveness analysis takes the form of a cost-utility analysis. Some studies [122, 125, 126, 128, 134, 135] calculated the net-benefits of supportive care interventions using a series of hypothetical values for the decision maker’s willingness-to-pay (WTP) for an additional unit of outcome (e.g., a one-point difference in the Neuropsychiatric Inventory (NPI) score).
Twelve studies were pure cost-effectiveness analyses [70, 73, 76, 83, 118–120, 124, 127, 129, 131, 134], eleven were cost-utility analyses [63, 65–67, 69, 72, 79, 80, 116, 125, 132], twelve developed both a cost-effectiveness and a cost-utility analysis [62, 68, 71, 78, 84, 117, 121–123, 126, 133, 135], three were cost-consequence analyses [77, 81, 130], and one was a cost-benefit analysis [128].
Quality assessment of the included studies
As indicated above, we also assessed the methodological quality of the studies included in the review using the CHEC checklist [88]. Based on the scores assigned, studies were classified as being high-, medium-, or low-quality. The quality level of the study is reported in the last column of Tables 4–8. Overall, only three studies [63, 132, 133] met all 19 criteria defined in the checklist. Applying the CHEC criteria described in the Materials and methods section, we found 15 high-quality studies, 16 medium-quality studies, and 8 low-quality ones. Details on the ratings of the studies can be found in S1–S5 Tables.
Evidence of cost-effectiveness of supportive care interventions from reviewed studies
In this section, we describe the results of the qualitative analysis of the studies considered in the systematic review by distinguishing between the five categories of SC strategies described in the Materials and methods section. As already mentioned, Tables 4–8 report the main characteristics of the analysed studies for each category of intervention, including: description of the intervention under evaluation and of comparator; country where the intervention was implemented; type of study, type of economic evaluation, and time horizon; sample size (i.e., number of PwDs and caregivers considered in the study as well as the size of intervention and control groups); perspective of the economic evaluation; patient outcome measures; caregiver outcome measures; mean ICER or other cost-effectiveness measures (e.g., the intervention’s net benefit); assessed level of cost-effectiveness; cost-effectiveness rationale; and assessed quality of the study according to CHEC.
The analysed SC interventions are rated according to three levels of cost-effectiveness:
- high cost-effectiveness when the intervention is found to be dominant or when the incremental cost per QALY gained is below the target threshold considered by the study (often this corresponds with that currently used by the British National Institute for Health and Care Excellence-NICE [136]) with a limited amount of statistical uncertainty;
- moderate cost-effectiveness when the incremental cost per QALY gained is above but reasonably close to the target threshold, the analysis leads to mixed results (e.g., there is statistical uncertainty in the ICER value or there are no established willingness-to-pay thresholds for gains in relevant outcomes) or it is partial (e.g., there is no evidence of cost-effectiveness in terms of gains for relevant outcomes);
- low or absent cost-effectiveness when the ICER is well above the target threshold or the intervention is found to be dominated by the comparator.
Cognitive therapy interventions.
Table 4 summarizes the main characteristics of nine cognitive therapy interventions. One is classified as having high cost-effectiveness, six as having moderate cost-effectiveness, and two as having low or no cost-effectiveness.
Occupational therapy seems to be the most cost-effective form of cognitive therapy, as it is linked to cost savings and in most instances an improvement in patient and caregiver outcomes [119, 127]. In particular, Graff et al. [119] studied a community-based occupational therapy intervention that included both behavioural and cognitive interventions and which was directed at community-dwelling patient-caregiver dyads. The authors found average savings of approximately €1,748 per couple who had been successfully treated with the considered occupational therapy compared to usual care. Successful outcome was defined as a clinically relevant improvement in patients and caregivers for three primary outcome measures (process scale and performance scale for PwDs; competence scale for carers). The probability of occupational therapy being the dominant intervention (i.e., more effective and less costly) was estimated to be 95%. Another form of occupational therapy–the Tailored Activity Program (TAP) analysed by Gitlin et al. [127]–showed instead moderate cost-effectiveness with regards to caregiver-side outcomes (no primary outcomes for PwDs were considered).
Four cognitive stimulation therapy programmes in different settings [121–123, 134] and one learning therapy intervention for nursing home patients [128] show moderate levels of cost-effectiveness. These interventions generally highlighted potential cost-effectiveness in terms of quite heterogeneous outcome measures such as patient’s self-assessed goal attainment, cognitive function (MMSE score), quality of life (QoL-AD) and needed quantity of long-term care, while they provided low or no evidence of cost-effectiveness in terms of QALY gains for PwDs or their carers (only the analysis by Orgeta et al. [123] found some evidence of cost-effectiveness in terms of caregiver’s QALY gained). This makes their comparability rather complicated in terms of cost-effectiveness, even if useful considerations can still be drawn from their comparative analysis. For example, comparing the point estimates of the incremental cost for 1-point improvement in MMSE and Qol-AD scores of the maintenance cognitive stimulation therapy analysed by D’Amico et al. [122] with the CST intervention examined by Knapp et al. [134] (both were targeted towards patients with mild-to-moderate dementia in different settings), the latter would seem to be relatively more cost-effective even after correcting for the different duration of the interventions and inflation.
Finally, one reminiscence group therapy programme directed at community-dwelling patient-caregiver dyads [124] and one robotic plushie therapy intervention for institutionalised dementia patients [129] show no cost-effectiveness due to a lack of impact on outcomes, and a significant cost increase with respect to comparators.
Physical activity interventions.
Table 5 summarizes the main characteristics of three physical therapy interventions for community-dwelling PwDs for which the evidence of cost-effectiveness is mixed: two are classified as having moderate cost-effectiveness, and one as having low or no cost-effectiveness.
Two studies on individually-tailored exercise programs report evidence of moderate cost-effectiveness compared to usual care: the interventions may be cost-effective in terms of improvements in behavioural and psychological symptoms measured with the NPI score [135] or in terms of improved physical performance [126]. However, in both cases they do not appear to be cost-effective when considering QALY gains compared to control condition. In particular, D’Amico et al. [135] observed that there was no established cost-effectiveness benchmark for the NPI with which to compare their estimates, while the estimated mean cost per QALY was rather high relative to the upper threshold (£30,000) generally associated with cost-effectiveness judgements by NICE in the UK.
On the contrary, a much larger trial, with a more extended observation period [125] provides strong evidence that a tailored, structured, moderate-to-high intensity exercise programme for PwDs in addition to usual care is unlikely to be cost-effective when compared with usual care alone: the intervention is associated with a higher cost and a lower effect (in either improvement in cognitive outcomes or QALYs) and was dominated by the comparator.
Indirect strategies.
Table 6 summarizes the main characteristics of ten indirect interventions in different settings, described in eleven studies (two studies [63, 116] considered the same Dementia Care Management programme). Two interventions are classified as having high cost-effectiveness, three as having moderate cost-effectiveness, and five as having low or no cost-effectiveness.
Overall, group-living for certain populations of PwDs (specifically those who are on the verge of needing institutionalisation) and community-based Dementia Care Management seem to be the more cost-effective indirect strategies.
In particular, Wimo et al. [67] analysed group living (an intermediate level of care between home and institutionalisation) for dementia patients and compare it to home living and to institutional living. They found it to be dominant over both alternatives even at low WTPs. This result indicates that the intervention should be recommended as long as there are patients suitable for group living in institutions or as long as there are patients living at home who are on the threshold of being institutionalised, even though additional evidence is needed since the study was not a RCT and was conducted several years ago.
Michalowsky et al. [63] evaluated a Dementia Care Management programme, aiming to support patients and their caregivers through coordination and management of treatment and care and consisted of a nurse-led in-depth assessment of patients’ unmet needs to optimise and individualise dementia treatments (DelpHi-MV trial). The intervention was delivered in participants’ homes by nurses with dementia-specific qualifications. The study was conducted from the public payer perspective, considering only outcomes for PwDs. In the base-case analysis, Michalowsky et al. [63] found the intervention to be dominant over usual care in patients living alone, and they report an ICER of €26,851 per QALY (below the NICE’s threshold of £30,000 per QALY) for PwDs living with a caregiver. In particular, treated patients faced higher costs for medications but had lower costs in terms of in-hospital treatments, nursing home care, and delayed institutionalisation (the time to institutionalisation was delayed on average seven months in patients who received the intervention). The study reports cost-effectiveness with a probability 88% at a WTP of € 40,000 per QALY (close to the NICE’s reference threshold of £30,000). A recent follow-up study by Rädke et al. [116] focused on subgroups of participants in the DelpHi-MV trial, and they found the intervention to be dominant over usual care for patients older than 80, females, patients living alone, and with functional impairment or a cognitive deficit; for these groups, the probability of the intervention being cost-effective at a WTP of € 40,000 per QALY was significantly higher, compared to the whole sample of patients.
Other three community-based indirect interventions [62, 70, 120] show moderate levels of cost-effectiveness. MacNeil Vroomen et al. [62] compared two forms of Dementia Care Management (Intensive Case Management Model (ICMM) and Linkage Model (LM)) with usual care, and they found for all the considered outcomes (QALYs,NPI and GHQ scores) a probability of 97% or higher for ICMM being cost-effective over LM and usual care at a WTP of €0 per incremental unit of effect. However, the interventions showed also a small negative impact on QALYs for PwDs in both case management groups compared to the control group, and so the authors observed that policy makers should decide whether this small negative effect on QALYs is acceptable based on the generated cost savings that the ICMM model appeared to provide. Moreover, they pointed out that their findings should be interpreted with caution since the study was not a randomized controlled trial. Another indirect strategy consisting in adult day-care (Wimo et al., [70]) showed moderate cost-effectiveness in a prospective study conducted several years ago: although the authors reported non-significant changes in outcomes and costs between the intervention and the control group, they noted a significant decrease in costs (while QoL measures remained at the same level) for the subgroup of patients with the highest levels of psychosocial distress. In a more recent RCT, Melis et al. [120] evaluated the cost-effectiveness of the Dutch Geriatric Intervention Programme, consisting of regular nurse visits for community-dwelling frail older people, including PwDs. The difference in the treatment effect was calculated as the difference in the proportions of successfully treated patients (prevention of functional decline together with improved wellbeing). The ICER, expressed as the total incremental cost per successful treatment, was €3,418 (-21,458 to 45,362). According to the authors, the intervention had a 95% probability of being cost-effective compared with usual care for a WTP of €34,000 for a successful treatment, but this result does not seem decisive in establishing its real value for money since there was no established WTP threshold for the outcome considered.
Lastly, three indirect interventions directed to institutionalised patients (MARQUE programme of training sessions for nursing home staff [65], two Dementia Care Mapping (DCM) protocols [130, 133]) and two interventions for community-dwelling patients (a Dementia Care Management programme [66] and an assistive technology and telecare programme for independent living [68]) did not show evidence of cost-effectiveness.
Interventions primarily aimed at supporting family caregivers.
Table 7 summarizes the main characteristics of seven community-based interventions to support informal caregivers of PwDs, described in nine studies (one intervention–START–has been analysed by three separate publications [71, 117, 118]). Two interventions are classified as having high cost-effectiveness, three as having moderate cost-effectiveness, and two as having low or no cost-effectiveness.
Some psychosocial interventions that target problem areas linked to informal caregivers’ risks and QoL provide evidence of higher cost-effectiveness. In particular, Nichols et al. [73] considered a psychosocial intervention (REACH II) for caregivers of community-dwelling dementia patients, consisting of individual sessions and telephone-administered support group sessions. In this case, the ICER represented the cost of an additional hour of non-caregiving time that could be “purchased” by the intervention. There was no significant difference in formal healthcare use between the control and intervention dyads (carers and patients), while there was a significant reduction of hours of provided care for the caregivers in the intervention group compared to those in the control group. Nichols et al. found that the six-month intervention was cost-effective if one was willing to spend $4.96 per day for one extra hour of non-caregiving time for each caregiver.
Three studies [71, 117, 118] analysed the cost-effectiveness of the “Strategies for Relatives” (START) intervention, an individual psychosocial therapy programme which employed a similar therapeutic approach of the REACH II intervention and was aimed at informal caregivers to help them cope with the illness faced by their non-institutionalised relative. This particular approach consisted of an eight-session, manual-based coping intervention delivered by supervised psychology graduates to family carers of PwDs in addition to usual treatment. A first study by Knapp et al. [71] examined the short-term (eight months) cost-effectiveness of START, finding that the intervention had a 99% chance of being cost-effective compared with usual treatment alone at a WTP threshold of £30,000 per QALY gained (the higher threshold currently used by NICE). Livingston et al. [117] considered the START intervention for a longer timeframe of 24 months, and they found it to be dominant over usual care when looking at caregiver-side outcomes (such as the caregiver’s QALYs and depression and anxiety measures) and considering carer-and-patient costs combined; moreover, the intervention had a 70% probability of being cost-effective in terms of carer QALY gain at the WTP threshold of £30,000 per QALY. A 2019 follow-up by Livingston et al. [118] found that, after six years, the positive difference in outcomes between the intervention and control groups was small but statistically significant and sustained, whereas the difference in costs was economically large, despite the fact that there was no significant difference in some patient-side outcomes such as time to institutionalisation or death. These studies also reported cost-effectiveness of START with respect to the HADS scale for depression and anxiety in caregivers (e.g., a mean ICER of £179 for 1-point reduction in the HADS total score), even though a commonly accepted reference WTP threshold for this particular outcome is unknown.
Three interventions to support family carers [69, 76, 131] are characterised by moderate cost-effectiveness. Drummond et al. [69] focused on a caregiver support program that included nurse visits, support groups and respite care, and they found limited evidence of cost-effectiveness due to the statistically non-significant difference in outcome levels between the intervention and control groups. Gaugler et al. [131] analysed an adult day care service with the explicit aim of supporting informal caregivers. The mean ICERs of the intervention were calculated as the cost necessary to alleviate role overload (ROS score) and depression (CES-D score) by one unit both in the short and long term. The authors found that the daily costs of carers’ benefits were reduced over a 1-year period (to $4.51/day per unit of ROS score and $2.20/day per unit of CES-D score, respectively) and that the long-term utilisation of day care could help to lessen the time caregivers spent managing symptoms associated with dementia and allow them to spend more time in work-related activities. Shaw et al. [76] compared a telehealth intervention to assist caregivers to traditional telephone-based assistance, and they found that the intervention could be close to the WTP threshold for an increase in caregiver competence score (measured from the Short Sense of Competence Questionnaire–SSCQ) considered for other caregiving-related interventions. In all these three cases, the lack of commonly accepted WTP thresholds for the outcomes considered reduces the possibility of measuring the real cost-effectiveness of the interventions.
Finally, one intervention revolving around regular family meetings for informal caregivers [72] and a structured befriending service for family carers [132] were not found to be cost-effective compared to usual care in terms of QALYs gained by carers or dyads.
Multicomponent interventions.
Table 8 summarizes the main characteristics of six multicomponent interventions, described in seven studies (one intervention–WHELD–has been analysed by two separate publications [77, 78]). Two are classified as having high cost-effectiveness, two as having moderate cost-effectiveness, and two as having low or no cost-effectiveness.
A multicomponent programme for nursing home patients shows high levels of cost-effectiveness: the UK-based “Improving Wellbeing and Health for People with Dementia” (WHELD) intervention. This approach consists of a protocol to manage agitation coupled with physical exercise and psychosocial activities, all within a person-centred care framework. The protocol is focused on training care staff and promoting tailored person-centred activities and social interactions; it also involves the development of a system for triggering the appropriate review of antipsychotic medications by the prescribing physician. A preliminary study by Ballard et al. [77] found evidence of the potential high cost-effectiveness of WHELD. In particular, the intervention produced significant benefits in terms of patients’ QoL measured with a Dementia Quality of Life (DEMQOL) proxy (i.e., the DEMQOL score reported by the carer), agitation (Cohen-Mansfield Agitation Inventory-CMAI score), and overall neuropsychiatric symptoms (NPI score), especially for people with moderately severe dementia. Taking into account the health and social care costs, the authors found that the WHELD intervention reduced costs compared to usual care; therefore, the benefits achieved were associated with cost savings. In another study, Romeo et al. [78] found that the WHELD intervention was cost-effective compared to usual care alone across a wide range of WTPs on the part of a decision maker for a unit improvement in outcome (the considered outcome measures were both QALYs and CMAI scores). The cost-effectiveness was mostly attributed to the lower health and social care costs faced by the intervention group compared to the control group. The authors also found that these results were mainly relevant to residents with clinically significant agitation in dementia. These studies on WHELD [77, 78] aimed to report on the value of nursing home residents using interventions that consider the reduction of antipsychotic use, but they did not find a significant reduction in antipsychotic consumption in the treated group of PwDs compared to the control group.
Steinbeisser et al. [83] provide evidence of high cost-effectiveness for MAKS, a non-pharmacological treatment with four components (motor stimulation, activities of daily living stimulation, cognitive stimulation, and social functioning) for individuals in day care centres with mild or moderate dementia. They found that the intervention had a high probability to be dominant with better outcomes (a higher ability to perform activities of daily living measured with ETAM scores, better cognitive abilities measured with MMSE scores) and lower costs than usual care.
Other two multicomponent interventions [79, 84] show moderate levels of cost-effectiveness. Wolfs et al. [79] studied an intervention adopted in the Netherlands that consisted of an integrated approach protocol involving the use of a diagnostic research centre for psycho-geriatrics. This centre was designed to enable health professionals to first map the needs of the community-dwelling patient and their caregiver, and then deliver a personalised treatment course consisting of different kinds of activities. They found that the intervention was cost-effective in terms of QALYs gained but not in terms of improvements in clinical measures such as cognitive impairment or behavioural and psychological symptoms; however, these results were subject to relevant statistical uncertainty. El Alili et al. [84] focused on the Namaste Care Family Program for nursing home patients and they found limited evidence of cost-effectiveness owing to high statistical uncertainty surrounding the results and to the fact that for two outcome measures (QUALID for patient QoL and GAIN for caregiving) no societal WTP thresholds have been defined yet.
Lastly, one structured psychosocial intervention analysed within the Danish Alzheimer’s Intervention Study (DAISY) [80] and one multicomponent support intervention for couples [81] showed a very low probability to be cost-effective compared to usual care even though neither was found to be detrimental for either patients or caregivers.
Discussion
Main findings
This systematic review highlights the main evidence on the cost-effectiveness of SC interventions for PwDs and their caregivers. The analysed studies were quite heterogeneous in quality and included relevant costs and outcome measures. Nevertheless, the higher quality studies may provide useful findings on the value for money of specific interventions.
Eleven studies provided evidence of high cost-effectiveness for seven interventions: two multicomponent programmes (WHELD, targeted towards patients in nursing homes [77, 78], and MAKS in day care centres for community-dwelling people [83]); two indirect interventions (a group living service for PwDs [67] and a community-based Dementia Care Management programme [63, 116]); two interventions, START and REACH II, aimed at caregivers of community-dwelling PwDs [71, 73, 117, 118]; and one community-based cognitive stimulation and occupational programme for community-dwelling PwDs [119]. None of these studies showed a low level of methodological quality as regards the economic analysis: five [63, 71, 83, 116, 119] were assessed as being of high quality according to the CHEC criteria [88] while six other studies [67, 73, 77, 78, 117, 118] received a medium-quality appraisal. Undoubtedly, both the target populations and the methods adopted for measuring the cost-effectiveness of these SC interventions are quite heterogeneous, as we have shown in the previous section. However, all the interventions with evidence of high cost-effectiveness except one (the REACH II intervention for informal caregivers analysed by Nichols et al. [73]) were also found to be dominant (less costly and more effective than comparators) with a high probability according to sensitivity analysis, pointing out that they appear to be particularly promising in terms of economic sustainability.
Other sixteen SC interventions were found to be moderately cost-effective: six cognitive stimulation and rehabilitation programmes for community-dwelling PwDs [121, 123, 127], nursing home residents [128], or PwDs in different settings [122, 134]; two home-based individually tailored physical exercise programmes [126, 135]; three indirect interventions, including a Dementia Care Management programme [62] and two home care services [70, 120]; three interventions for family caregivers [69, 76, 131]; and two multicomponent interventions targeted towards community-dwelling PwDs [79] and nursing home patients [84].
Twelve SC interventions showed low or no cost-effectiveness: two cognitive stimulation programmes directed at community-dwelling patient-caregiver dyads [124] and institutionalised patients [129]; one aerobic exercise and resistance training programme [125]; five indirect interventions, including training sessions on agitation for nursing home staff (MARQUE) [65], memory clinics [66], telecare [68] and Dementia Care Mapping in a nursing home setting [130, 133]; two interventions primarily targeted towards family caregivers [72, 132]; and two community-based multicomponent interventions [80, 81].
Our analysis partially confirms some results of previous systematic reviews. For example, the systematic reviews by Nickel et al. [42], Knapp et al. [43], and Clarkson et al. [46] suggested that tailored occupational therapy for community-dwelling patients and caregivers [119, 127] and cognitive and long-term psychological interventions directly delivered to PwDs [122, 134] may be either highly or moderately cost-effective with regard to specific outcomes, while joint reminiscence groups for PwDs and carers 124] were found unlikely to be cost-effective. Previous systematic reviews, including the analysis by Jones et al. [45], provided mixed evidence with regard to interventions aimed directly at informal caregivers, even though in more recent reviews [42, 46], the START intervention [71, 117, 118] emerged as potentially cost-effective. Our analysis shows that some forms of psychosocial intervention for informal caregivers are highly cost-effective (e.g., the START and REACH II programmes [71, 73, 117, 118]) or moderately cost-effective (e.g., the support programmes analysed by Drummond et al. [69] and Gaugler et al. [131], and the telehealth intervention considered by Shaw et al. [76]), while other similar interventions have little or no cost-effectiveness (e.g., family meetings and befriending [72, 132]).
In contrast to other reviews, we found a high value for money of structured multicomponent interventions targeted towards patients in nursing homes [77, 78] and individuals in day care centers with mild or moderate dementia [83], which have the potential to draw benefits from the most cost-effective one-dimensional programmes. This was the case for the UK-based Improving Wellbeing and Health for People with Dementia (WHELD) programme for patients in nursing homes, which combines person-centred care, physical exercise, psychosocial activities, training for care staff, and the development of a system for triggering the appropriate review of antipsychotic medications for PwDs [77, 78]. On the contrary, other multicomponent interventions were assessed as moderately cost-effective [79, 84] or without any significant cost-effectiveness [80, 81]. We also found evidence of moderate cost-effectiveness of specific cognitive stimulation programmes for institutionalised PwDs (in particular, the learning therapy programme analysed by Sado et al. [128]) or for community-dwelling PwDs and their carers (specifically, the tailored cognitive rehabilitation programme investigated by Clare et al. within the GREAT trial [121]). In contrast to previous reviews [42, 46], we were not able to find clear evidence of high value for money for exercise programmes. For example, two individually tailored exercise interventions significantly improved patients’ physical performance [126] or NPI scores [135] but they did not appear cost-effective when considering QALY gains; the DAPA, another exercise programme that was studied by Khan et al. [125], was dominated by usual practice in terms of cost-effectiveness. Similarly, several indirect interventions centred around organisational and environmental changes showed either moderate [62, 70, 120] or no [65, 66] cost-effectiveness.
Finally, two studies [68, 76] provided inconclusive evidence regarding the cost-effectiveness of telehealth and telecare interventions to support independence and improve QoL of both patients and informal caregivers, which could be particularly useful in situations such as the coronavirus disease pandemic we are experiencing. While the FamTechCare intervention to assist informal caregivers appeared to be cost-effective when compared to traditional telephone support intervention [76], the use of assistive technology and telecare in supporting PwDs to continue to live safely within their own homes [68] did not prove to be cost-effective compared to more basic systems mainly due to the difficulty of adapting the indirect intervention to the needs of PwD and their carers.
Methodological and operational challenges for the cost-effectiveness of supportive care interventions
From this review, we can identify a number of critical issues concerning both the methodology of economic evaluations and actual barriers to achieving better value for money of SC for dementia.
A first issue is the high methodological heterogeneity of the available studies in terms of quality, populations studied (regarding severity, comorbidity, and care settings), and the inclusion of relevant costs and outcome measures, which make it difficult to generalise their results. A frequent limitation of the analysed studies is the short time frame adopted for measuring most outcomes and costs. This is a result of the fact that most studies were trial-based evaluations. Additional research would be required to investigate the effects of SC over longer time horizons. For example, using a decision analysis modelling strategy to compare the costs and effectiveness of the interventions in the longer term could be an option to explore.
A second methodological issue concerns the instruments used to measure the outcomes of SC in terms of QoL for people with dementia and to derive QALYs in cost-utility analyses. The reviewed studies applied two types of instruments: a) generic instruments to assess HRQoL, such as the EQ-5D [62, 65, 66, 68, 79, 80, 84, 122, 124–126, 128, 133], the IWB scale [67, 70], and the SF-12 [63, 72, 116]; and b) dementia-specific instruments to measure the QoL of PwDs, such as the QoL-AD [117, 122–125, 134], the DEMQOL [121, 122], the DEMQOL-Proxy [65, 77, 78, 122, 123, 133, 135], and the QUALID [84]. The generic and dementia-specific QoL measures generally did not provide consistent cost-effectiveness findings. In particular, generic measures may not capture all relevant aspects associated with PwDs’ experiences, even though instruments such as the SF-12 or EQ-5D have been shown to be suitable for HRQoL self-reporting by PwDs in mild and moderate stages [63, 66, 68, 116, 122, 124–126].
Since the assessment of self-report HRQoL and dementia-related QoL in PwDs is often characterised by recall bias and missing values, many studies opted for using carer-proxy reports of PwD QoL [62, 65, 68, 72, 77–80, 84, 117, 122–125, 133, 135]; another option was to convert data from clinical/health measures into QoL estimates according to the views of expert external observers [67, 70]. However, there are important differences between self-report and carer-proxy (or expert-proxy) reports [42, 137, 138]. For this reason, it may be useful to use both self and proxy ratings in the economic analyses of SC. Actually, some of the analysed studies [68, 122–125, 133] provided both measures of PwDs’ QoL, thus confirming that self-rated and proxy QoL measures often have low levels of overall agreement and therefore cannot be assumed to substitute for each other. Furthermore, because the outcomes for caregivers and PwDs are typically interlinked, it is important to jointly assess the QoL of patient-caregiver dyads in order to take into account the type of caregiving relationship, which is an area of outcome assessment that has not yet been adequately developed. Four studies considered in our review assessed the QoL of patient-caregiver dyads [62, 66, 72, 80] by calculating the combined QALY scores through the simple summing of the QALYs for the PwD and the caregiver.
Another methodological issue concerns the identification and evaluation of the costs of SC interventions. In this respect, 18 out of 39 of the studies under review adopted a narrow perspective, looking only at the health and social care system and overlooking the opportunity costs of informal caregiver inputs and the impacts of caring on their own health and wellbeing. Given the pivotal roles of family and other carers in dementia care, several authors [42, 43] recommend that economic evaluations of SC programmes for PwDs take on a societal perspective, including all relevant costs irrespective of where they occur and where they are funded.
Regarding the operational challenges surrounding the adoption of cost-effective SC, a first issue to consider is that pure cost-effectiveness analyses measure the ICER as an incremental cost per one-point difference in specific outcome measures, such as the MMSE [83, 122, 134], QoL-AD [117, 122, 124, 134], NPI [62, 135], CMAI [78, 129, 133], or carer’s HADS scores [71, 117, 118]. However, in contrast to a cost-utility analysis, where the ICER is expressed in terms of the incremental cost per QALY gained and the British NICE’s acceptability threshold range of £20,000–30,000 per QALY is frequently used, no established cost-effectiveness benchmark exists for such outcome changes. Therefore, as reported by many of the analysed studies, it is quite difficult to ascertain whether a particular SC intervention represents good value for money to the health and social care system, because we do not know the decision maker’s willingness-to-pay for a one-unit reduction in the MMSE, Qol-AD, NPI, CMAI, or HADS scores. In these cases, a useful strategy adopted by some of the studies examined [62, 63, 83] could be to measure the probability that the intervention leads to a significant outcome improvement for different levels of the societal WTP, starting from a WTP equal to zero (i.e. without entailing increased costs for the taxpayer). Another often used strategy is to measure the societal WTP by quantifying the costs avoided thanks to the improvement in the outcome. For example, Murman et al. [139] showed that a 1-point worsening of the NPI score is associated with an incremental increase of between $247 and $409 per year in total direct costs of care based upon year 2001 US dollars. However, even in the absence of a WTP defined a priori by the policymaker for improvements in some of specific outcomes (MMSE, QoL-AD, NPI, etc.), carrying out comparative analyses of cost-effectiveness such as ours can provide useful policy indications, highlighting, for homogeneous categories of interventions and PwDs, those types of SC which show lower ICERs. For example, in previous section, we have shown that, after controlling for characteristics of targeted population (patients with mild-to-moderate dementia in different settings), intervention duration and price inflation, the cognitive stimulation therapy studied by Knapp et al. [134] had much lower ICERs than the maintenance cognitive stimulation therapy analysed by D’Amico et al. [122] in terms of both MMSE points and Qol-Ad points gained.
A final operational challenge underlined also by Knapp et al. [43] is that the cost-effectiveness of SC for PwDs depends crucially on the degree of integration between health and social care services, which are often delivered by different providers and funded from different budgets. It is therefore crucial to improve coordination between these services in order to increase the efficiency and effectiveness of interventions for PwDs.
Limitations
Even though we conducted a comprehensive literature search based on extensive search terms, some papers meeting the criteria for inclusion might not have been identified. Furthermore, our systematic review may be subject to a language bias, as only publications in English or those with an abstract in English were included.
Some studies reviewed [67, 69, 70] were published years before the development of guidelines for assessing the methodological quality of health economics evaluations, such as the CHEC criteria [88]; therefore, their quality assessment may have been compromised. In any event, we decided to include these studies in the review because they evaluated forms of home support not considered by more recent studies, thereby allowing us to cover a wider range of SC interventions for PwDs.
Future research
Our systematic review has highlighted the potential cost-effectiveness of multicomponent SC interventions targeted towards patients in nursing homes (e.g., the WHELD programme [77, 78]) that combine several interventions (person-centred care, physical exercise, psychosocial activities, behaviour management and training for care staff, the development of multi-disciplinary teams) with positive effects in terms of QoL and a decrease in challenging behaviour. Other studies [79–81] provided mixed evidence with regard to the value for money of community-based structured multicomponent interventions targeted at persons with MMD. Future research should therefore examine the cost-effectiveness of structured multicomponent interventions in different care settings and consider subgroups of PwDs at different disease stages. Furthermore, it is important to assess the impact on cost-effectiveness of the different components of multi-disciplinary interventions by focusing on the assessment of the roles of care coordination and case management. Eliciting the contribution of each component to the interventions’ costs and outcomes would be important in terms of policy by highlighting how and why specific interventions may work to benefit PwDs and/or their caregivers.
Similar to other systematic reviews [42, 45, 46], we found mixed evidence with regard to SC interventions targeted towards informal caregivers. Specifically, some forms of psychosocial intervention for informal caregivers are highly cost-effective [71, 73, 117, 118] or moderately cost-effective [69, 76, 131], while others have shown little or no cost-effectiveness [72, 132]. As such, further investigation is needed to ascertain the real effects of interventions aimed directly at carers.
Finally, given that, as mentioned in the Introduction, one of the key aspects of SC should be the decreasing reliance on medications, in particular antipsychotics, that do not offer a sufficient benefit relative to the risks they pose, it appears somewhat surprising that only a few of the examined studies considered the reduction in antipsychotic drug consumption as one of the outcomes to be evaluated. In our opinion, future cost-effectiveness analyses of SC interventions should focus more on this aspect which so far appears to be rather neglected.
Conclusion
To assess the current state of research on the cost-effectiveness of SC interventions for dementia, we performed a systematic review of the economic evidence, which is still scarce despite the several calls for action that have been made in the past few years [19, 43, 45, 140]. We reviewed 39 studies that analysed 35 SC programmes located at different stages of the care pathway for dementia that were generally directed at patient-caregiver dyads. Most interventions (23 out of 29) were implemented in European countries with comparable underlying health and social care systems.
We found that the most promising SC programmes in terms of cost-effectiveness were some multicomponent interventions targeted towards both nursing home residents, such as the WHELD programme [77, 78] and day-care service users [83], together with some forms of tailored occupational therapy [119] and home care support services [63, 67, 116] for community-dwelling PwDs. Our analysis has also shown that some forms of psychosocial intervention for informal caregivers of community-dwelling PwDs, such as the REACH II and START programmes [71, 73, 117, 118], were highly cost-effective. These results suggest the importance of policies promoting the adoption of effective supportive care interventions to increase the economic sustainability of dementia care.
Further research is required to establish the cost-effectiveness of structured multicomponent interventions in different care settings by considering subgroups of PwDs at different disease stages and assessing the impact of each component of the intervention. Moreover, since the evidence on the cost-effectiveness of SC interventions targeted towards informal caregivers is mixed, further investigation is needed to ascertain the real effects of these interventions on both the PwD and his/her carer. Lastly, we think that empirical evidence on the real ability of SC interventions to reduce the use of antipsychotic medications in PwDs is still lacking and should be considered in future research.
Supporting information
S2 Checklist. PRISMA 2020 for abstract checklist.
https://doi.org/10.1371/journal.pone.0285305.s002
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S1 Table. Assessment of methodological quality of the studies on cognitive therapy interventions.
https://doi.org/10.1371/journal.pone.0285305.s004
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S2 Table. Assessment of methodological quality of the studies on physical activity interventions.
https://doi.org/10.1371/journal.pone.0285305.s005
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S3 Table. Assessment of methodological quality of the studies on indirect strategies.
https://doi.org/10.1371/journal.pone.0285305.s006
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S4 Table. Assessment of methodological quality of the studies on interventions primarily aimed at supporting family caregivers.
https://doi.org/10.1371/journal.pone.0285305.s007
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S5 Table. Assessment of methodological quality of the studies on multicomponent interventions.
https://doi.org/10.1371/journal.pone.0285305.s008
(DOCX)
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