In health care, patients’ decisions about treatment and screening are often described as ‘preference sensitive’, where the relative importance a patient attaches to various outcomes and processes has a large, if not determining, influence on what is decided [17]. Quality decision-making is therefore said to be the extent to which the chosen option matches the informed decision-maker’s values for benefits, harms, and uncertainties [18]. The effectiveness of DAs on improving decisions (making them ‘good’ decisions) can be viewed as influencing two constructs: i) the quality of the decision-making process and ii) the quality of the choice that is made (i.e. decision quality) [19]. There is growing evidence of the effectiveness of DAs in improving both the quality of the decision-making process and decision quality for healthcare decisions [19]. A recent Cochrane review established that decision aids can improve knowledge, reduce decisional conflict, clarify expectations of possible benefits and harms, lead to choices consistent with informed values, and result in greater participation in decision making [11].
A small number of interventions have been developed to promote informed decision-making about participation in clinical trials [20–22]. Of the small number of DAs that aim to improve decision-making for trial participation (rather than aiming to improve the presentation of information or mode of delivery) most focus on specific oncology trials [23]. These DAs show some potential promise in improving key decision outcomes such as knowledge, values clarification, and decision conflict, while not negatively impacting recruitment or intention to participate [20, 21, 24]. A recent systematic review of decision aids for trials only identified one study that evaluated the effectiveness of decision aids compared to standard information in the informed consent process for clinical trials [16]. The reviewers concluded that more high quality randomised controlled trials of decision aids to support the informed consent process for clinical trials are needed [16]. In non-oncology conditions, a qualitative exploration of stakeholders’ perceptions of decision aids for randomised controlled trials that are currently in development suggested that decision aids have the potential to better engage potential participants in the decision-making process and allow them to make more personally relevant decisions about their participation [25].
Legal and ethical frameworks require proxy decisions about research participation to be based on what the wishes and feelings of the person lacking capacity to consent would be about taking part in the study [3] or, for a clinical trial, representing their ‘presumed will’ [4]. This requires the proxy to first determine what the wishes and preferences of the person they are representing would be about participating, and then make a decision based on this determination. Proxy decisions can therefore also be viewed as preference sensitive decisions, but they are based on the patient’s preferences and values rather than their own. Whilst some patients may have discussed their preferences about participating in any research in the event of a loss of capacity to consent, in practice very few will have done so [8], and so making a decision that represents their wishes may be challenging for proxies.
Development of the decision support intervention
As the interest in DAs for trial participation grows, so does the importance that their development is both rigorous and evidence-based [23]. The development of this DA for proxy decision-making was informed by a recently proposed framework for the development and evaluation of decision aids for people considering taking part in a clinical trial [23]. The framework proposed by Gillies and Campbell consists of five steps: selecting an underpinning theoretical approach to the development process; 1) developing the decision aid; 2) assessing and testing feasibility; 3) evaluating the decision aid; and 4) implementing the decision aid in practice [23]. The first two steps of the framework are reported in this paper and an overview of the process is outlined in the figure below (Figure 1. Intervention development process flow diagram)..
<Figure 1. Intervention development process flow diagram >
Intervention development frameworks
The Medical Research Council (MRC) framework for developing and evaluating complex interventions is widely used [26]. It describes an iterative process of development, feasibility/piloting, evaluation, and implementation of interventions [26], however, it has limited detailed guidance for the development phase itself. Recently, a model which focuses on the intervention development phase (6Squid) has been developed which describes how the process can be broken down into six steps, although in practice the process is non-linear and collaborative [27]. Researchers have proposed combining the elements of the development phase of the MRC Framework with elements of the 6Squid model to enhance the intervention development through a more comprehensive approach. This ‘enriching’ approach to the development phase [28] has been used for the development this decision support intervention. The methods used in the 6Squid steps are shown in Table 1. Six steps in development of the decision aid (DA) intervention below.
Table 1. Six steps in development of the decision aid intervention
Step in intervention development
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Method used
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1) Define and understand the problem and its causes
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Systematic review, content analysis, qualitative data
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2) Clarify which causal or contextual factors are malleable
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Logic model
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3) Identify the change mechanism
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Qualitative data
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4) Identify how to deliver the change mechanism
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Qualitative data, review of current DAs
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5) Test and refine on small scale
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Review by PPI and stakeholder groups
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6) Collect sufficient evidence of effectiveness to justify rigorous evaluation/implementation
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Feasibility study*
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*Feasibility study to be conducted as part of a follow-on project
Decision aid development frameworks
The Ottawa Decisional Support Framework (ODSF), which is an evidence-based, practical, mid-range theory for guiding patients making health or social decisions, was used to develop the DA [18]. This theoretical framework consists of three components: decisional needs; decisional support; and decisional quality. The framework for the development and evaluation of decision aids for people considering taking part in a clinical trial outlined by Gillies and Campbell identified the ODSF as being a highly relevant theoretical framework to use when developing decision aids for trial participation [23]. This is because the ODSF asserts that decisional needs affect decisional quality, such as making informed and values-based choices, which in turn affect action, behaviour, and emotions such as regret [18]. These concepts are considered by Gillies and Campbell to map well onto the known issues within trial participation decisions [23] which are known to be preference-sensitive decisions [29] as is also the case for proxy decisions.
In addition to ODSF, the International Patient Decision Aids Standards (IPDAS) framework of quality criteria for patient decision aids was used to guide the development of the decision aid [30]. This evidence-informed framework provides a set of criteria which aims to improve decision aid content, development, implementation, and evaluation [30]. Alongside the IPDAS framework, a 44-item minimum standards ‘checklist’ version has been developed that is designed to rate the quality of the development process and decision-making design elements [31]. Relevant items from the IPDAS Minimal criteria v4.0 were used to inform the development of this decision support intervention. Checklist items considered not relevant, and hence excluded from the development process, included: information relating to the condition or problem and associated probabilities as this would be included in the Participant Information Sheet provided about the study (and so would be used alongside the DA) and information related to screening or tests as this was not applicable.
IPDAS guidelines for the development of patient DAs recommend that they include a process for helping people clarify their values [30]. These processes, usually termed values clarification methods (VCM), are defined as strategies intended to help individuals to evaluate the desirability of options or attributes of options within a specific decision context, in order to identify which option they prefer [32]. VCMs can be implicit and non-interactive (e.g., the individual thinks about what is important to their decision), or explicit and interactive (e.g., using a rating scale for each attribute to reflect the importance of each to their decision) which is much more widely studied [32]. Whilst DAs have been found to be effective in reducing decisional conflict and increasing knowledge, the effect of specific strategies such as VCMs is less clear [33]. As outlined earlier in the discussion of selected decision-making theories, deliberation may not always be beneficial for decision making as it may overshadow important intuitive feelings that are more difficult to formulate but may be just as important in decision making [34]. However, the use of VCMs in DAs decision aids is now widespread, with 57.1% of DAs in a recent systematic review including explicit methods to clarify values [11].
Review of existing decision aids for proxies
Although the search strategy for the systematic review of decision aids for trials specifically sought to include studies that included guardians of, or proxy decision makers for, potential trial participants, no existing decision aids for use by proxies have been identified [16]. However, a number of tools have been developed for proxies who are making other decisions on behalf of a person who lacks capacity, such as around the use of antipsychotic medication, place of care, retiring from driving due to dementia, and receiving mechanical ventilation [15, 35–37]. The DAs intended for use by proxies invariably make the assumption that the evidence that decision tools are effective in increasing knowledge and reducing decisional conflict is transferable to proxy decision-making, and there is some limited evidence of this. A small study examining the impact of a DA on proxy decision-makers’ perceptions of feeding options for people with dementia found it improved knowledge scores and reduced conflict [38]. A feasibility study of an intervention to support family carers make decisions about place of care for a person living with dementia also found that it reduced decisional conflict, although it did not remove all barriers to decision-making and some unresolved conflict remained [35].
Prior research which informed the intervention development
Clarifying the problem using the existing research evidence and in consultation with stakeholders is considered to be the first step in intervention development [27]. Phase 2 built on a systematic review which was conducted to synthesise the existing empirical evidence [8] which, together with a content analysis of information already provided to proxies [39], indicated that proxies are generally well provided with information about the study itself, but are not well informed about their role as proxy decision-maker. Whilst there is a paucity of literature regarding decision support in this context, the relationship between the parties involved appears to determine the kind of decision-making process used and impacts on the way decisions are achieved [40]. This decision support intervention therefore focuses on close family members acting as proxies, rather than others such as members of the healthcare team.
We conducted a qualitative study with family members acting as proxies which showed that they use the patient’s expressed wishes about research where these are available and, where these were not known, use their in-depth knowledge of the person’s values and preferences to facilitate decision-making on their behalf [9]. However, other proxies described it as being a difficult and challenging decision. Improving the decision-making process was recognised as being much more than just ensuring the proxy had received adequate information. Proxies thought that greater decision support when considering research decisions would help in the future, which included orientating them towards considering the person’s own views and preferences [9]. Proxies suggested that this support could take the form of a different sort of information sheet which covered their role as proxy decision-maker, and that the DA should include items that they considered would support proxies when making decisions about research (Table 2. Items for inclusion in decision aid from the qualitative interview findings)..
Table 2. Items for inclusion in decision aid from the qualitative interview findings
1. Why people with cognitive impairment are included in research
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2. That the proxy’s decision or advice should be based on what the person’s wishes and feelings about taking part would be if they had capacity to decide
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3. That the proxy should consider if there is any reason why the person would not have wanted to participate
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4. The relevant advantages and disadvantages and how they relate to the person themselves
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5. That the person should be involved in the decision as much as possible
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6. That the proxy can take time to decide and they can always change their mind
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As the majority of the participants were proxies of someone living with dementia, the intervention was developed with this population as its focus, although there is no reason to suggest it would not be appropriate for proxies of people with other conditions associated with cognitive impairment.
Collaborative development with stakeholders
Consultation with relevant stakeholders is an important step in the intervention development process [27]. This intervention was part of a larger project which benefitted from having the support of a lay advisory panel from the initial conception of the project through to the development of the intervention itself. The lay advisory group, also known as a Public and Patient Involvement (PPI) group attended a discussion meeting to review the first draft of the decision aid, broadly following a cognitive debriefing approach to instrument development which can identify difficult or confusing areas of the item being reviewed and propose a better version [41]. The aim is also to identify whether the interpretation of an item differed between those reviewing it [41].
Additionally, a larger group of stakeholders was convened and consulted who had experience as either a researcher who involve people who lack capacity in their research, family member who may be approached to act as a proxy, and those familiar with supporting family members of people with dementia. The importance of the involvement of practitioners and other stakeholders in developing and prototyping interventions, to ensure that they can be adopted, implemented and maintained in the contexts for which they are intended, is emphasised in many of the frameworks guiding intervention development [27, 42]. Their role is important in throughout the process, but particularly when determining the content, format, and delivery of the intervention [27]. Acceptability of an intervention has been defined as ‘a multi-faceted construct that reflects the extent to which people delivering or receiving a healthcare intervention consider it to be appropriate, based on anticipated or experienced cognitive and emotional responses to the intervention’ [43], and can be considered to have both cognitive and affective components [44]. This stage forms the ‘alpha’ testing of the intervention which is described as an iterative process of testing by people directly involved in the development process, and considered to be a necessary part of the development of decision aids [45].
Development of a socio-ecological model of proxy decision-making
The term ‘complex interventions’ are primarily used to refer to interventions as system changes that are focused on health promotion, social interventions, and public health more broadly. Complexity is increasingly being conceived in terms of how interventions interact with their contexts, where contexts are broadly defined as any feature of the circumstances in which an intervention is conceived, developed, implemented and evaluated [42]. There is a call for intervention researchers to move away from viewing interventions as discrete bundles of components which can be described in isolation from their contexts, and better understand the systems into which change is being introduced [46]. Thus there is a focus on interventions viewed as disruptions to complex systems, rather than on the intrinsic properties of interventions [47]. Decision aids, particularly this one to be used by proxies, can be considered to have properties of complexity. It is a complex intervention with several interacting components [48], entailing complex behaviours and a range of effects [49], and also as an event occurring within inevitably complex systems such as within a family, legal and ethical frameworks, and a healthcare system.
Proxy decision-making for research, whilst not traditionally thought of as a behaviour, is similarly situated within complex systems which can be perceived as having a number of levels which have contextually dependent dynamic interactions between them. Developing a socio-ecological model (SEM) as phase 3 of the process was found to be a useful organising construct for proxy decision-making. It was adapted from the model most commonly used for health promotion and behaviour change interventions [50]. An ecological perspective recognizes that individuals are located within a broader social context [51, 52]. In this model, behaviour is conceived as being determined by five levels of analysis: intrapersonal factors, interpersonal processes, community factors, institutional/organisational factors, and public policy or socio-cultural factors [53]. Developing the SEM enabled an exploration of the many different factors or levels involved, including the legal frameworks at both a policy and socio-legal level, the ethical governance processes involved, the knowledge and attitudes of the researcher and practitioner community, the interpersonal relationship between the proxy and the person they represent, which ultimately centre around the person who lacks capacity to consent. The main influences on the issue being examined were classified according to the socio-ecological model distinguishing between the different levels of the model (Figure 2. Socio-ecological model of proxy decision-making for research)..
< Figure 2. Socio-ecological model of proxy decision-making for research >