Patient, caregiver, health professional and researcher views and experiences of participating in research at the end of life: a critical interpretive synthesis of the literature
BMC Medical Research Methodology volume 12, Article number: 123 (2012)
The development of the evidence-base informing end of life (EoL) care is hampered by the assumption that patients at the EoL are too vulnerable to participate in research. This study aims to systematically and critically review the evidence regarding the experiences and views of patients, caregivers, professionals and researchers about participation in EoL care research, and to identify best practices in research participation.
We searched seven electronic databases, and hand searched three journals and the bibliographies of relevant papers. Inclusion criteria were original research papers on involvement in EoL care research or its impact on participants. Critical interpretive synthesis was used to integrate the whole body of empirical evidence on this topic and generate theoretical categories from the evidence.
Of a total of 239 identified studies, 20 studies met the inclusion criteria, from: the US (11), the UK (6) and Australia (3). Most focused on patients with cancer (12) and were conducted in hospices (9) or hospitals (7). Studies enquired about issues related to: EoL care research in general (5), specific research methods (13), and trial research (2). The studies evaluating willingness to participate in EoL care research showed positive outcomes across the different parties involved in research. Factors influencing willingness were mainly physical and cognitive impairment. Participating in research was a positive experience for most patients and carers but a minority experienced distress. This was related to: characteristics of the participants; the type of research; or the way it was conducted. Participatory study designs were found particularly suitable for enabling the inclusion of a wide range of participants.
The evidence explored within this study demonstrates that the ethical concerns regarding patient participation in EoL care research are often unjustified. However, research studies in EoL care require careful design and execution that incorporates sensitivity to participants’ needs and concerns to enable their participation. An innovative conceptual model for research participation relevant for potentially vulnerable people was developed.
According to projections, the number of people aged 65 and above will increase dramatically by 2030 (one in eight of the global population) . A demographic shift is occurring, with people generally living longer and a smaller number dying from infectious diseases . This brings a different and currently not well-understood scenario - involving older people suffering from chronic illnesses with complex and clinically challenging trajectories. This rapidly changing situation will create substantially greater end of life (EoL) care needs .
Within this paper, EoL care is understood in its broadest sense, referring to the care provided to people with advanced, progressing and incurable disease and their families. It covers the therapeutic approaches from diagnosis on, as well as the care provided at the very end of life, during death and the bereavement process.
Deficiencies in EoL care have been reported, such as in the area of pain and symptom management . There is a need for research involving patients at the end of their lives in order to develop and test new treatments and services and to ensure the provision of high-quality and cost-effective care [4–7]. Numerous practical and ethical obstacles to research during the EoL are well documented [8, 9]. Trial research is often hampered by problems of recruitment, high attrition rates, missing data and a lack of outcome measures validated with palliative care patients .
The ethical arguments between the value of research and the needs of patients are heavily debated and complex .
The debate reflects a community of stakeholders that is polarised between those who argue that research within this population is unjustified and those who argue that the ethical principles that govern medical research are also suitable to safeguard individuals’ interests to enable their participation in EoL care research. While it is necessary to consider the concerns of this debate there is little prospect of resolving the existing tensions by argumentation alone. Systematic reviews have appraised the evidence regarding advanced cancer patients’ attitudes towards research  and on palliative care patients’ and carers’ views about research in palliative care . This study broadens the scope by undertaking a critical interpretive synthesis (CIS)  across this field in order to synthesise existing empirical data on the attitudes and values regarding participation held by all stakeholders involved in palliative care research. Attention has been given to the methodological issues that emerge from these studies.
The study aims to appraise the evidence regarding all stakeholders’ views and experiences regarding involvement in palliative care research and to explore whether there are best practices regarding participation in research.
· Which stakeholders’ views on participation in research are represented in the evidence and what are the views held?
· Are patients and other stakeholders willing to participate in EoL care research?
· How is participation in EoL care research experienced?
· Are there differences in attitude according to group (patient or other participants’ groups)?
· What are the best practices to enable patients’ participation in research on EoL care?
We used CIS  to synthesise a diverse and complex body of evidence consisting of both quantitative and qualitative studies. This methodology incorporates procedures of conventional systematic review methodology by applying explicit searching strategies to ensure replicable, specified inclusion criteria, and data extraction procedures to enhance clarity and comparability among studies. This was combined with recent methods for interpretive synthesis which drew on traditions of qualitative enquiry. This combined approach enabled both aggregation of the literature and the generation of theoretical categories that critically demonstrate the assumptions situated within the literature.
The search was conducted in four electronic databases: MEDLINE (1950 – January week 2 2010); EMBASE (1980 – January week 2 2010); PsycINFO (1806 – January week 2 2010); and CINAHL (1982 – January). Additionally the following databases were searched for relevant articles: Cochrane Database of Systematic Reviews, The Cochrane Central Register of Controlled Trials, Cochrane Pain, Palliative and Supportive Care Trials Register.
The combination of search terms was piloted with a number of trial searches to achieve a balance between recall and specificity. A pre-defined search strategy was decided upon, consisting of the following search terms, representing three groups which were combined with AND.
attitude$, OR view$, OR experience$, involv$, partic$, perspect$, concern$, challeng$
palliative care, OR palliat$, OR terminal, OR terminally ill, OR supportive care, OR dying, OR death, OR end of life, OR end-of-life, OR hospice, OR bereavement.
We searched the reference lists of all relevant papers for further references. This was important as this topic concerns a body of evidence, which is not well delineated and is spread over the disparate research areas that address palliative care.
Three journals, in which the debate regarding whether or not to involve palliative care patients in research was conducted, were hand searched. These were:
The Journal of Pain and Symptom Management (from 1999 to February 2010)
Palliative Medicine (from 1989 to February 2010)
Journal of Palliative Care (from 1985 to June 2010)
Inclusion and exclusion criteria
We included original research papers that enquired specifically about involvement in EoL care research or its impact on participants. The participants included a wide variety of stakeholders who come into contact with palliative care research, including: patients, informal caregivers, health professionals, managers, and researchers. We included all research designs and searched across the spectrum of palliative care from disease diagnosis to death.
We excluded studies on oncology patients participating in trials testing curative treatments. We excluded papers on children or adults with learning disabilities as these areas have their own specific issues; and studies on recruitment, informed consent, user involvement or bereavement. Non-English language papers were excluded. We did not include papers that were already included in previous reviews. However, we investigated the individual studies in these reviews and included them if they contributed to our review questions and responded to our quality criteria. Fatally flawed studies were excluded. To identify such low-quality studies we used the following criteria (as specified in ):
Are the aims and objectives of the research clearly stated?
Is the research design clearly specified and appropriate for the aims and objectives?
Do researchers provide a clear account of the process by which their findings were produced?
Do the researchers display enough data to support their interpretations and conclusions?
Is the method of analysis appropriate and adequately explicated?
First, the identified studies were mapped according to country of origin, setting, participants, patient group, illness stage, and approach. The resultant studies were then counted. Three major categories were identified: studies that addressed issues relating to research in general; research with specific methods; and trials. Within these categories, studies were examined in more detail and the results compared. This revealed areas that were adequately covered and any remaining gaps.
In the second stage the research questions were applied to the literature across the categories. Results and emerging queries were listed. Recommendations from the studies were tabulated and organised inductively, thereby generating information on best practices.
A third stage focused on the normative assumptions underlying the studies and related these to the empirical evidence they generated. The characteristics attributed to ‘research’ and ‘care’ – and how these determined their conceptualization – were critically assessed. Whether the interpretation changed with each included study was continually tested. The findings are synthesised as a conceptual model for research participation for potentially vulnerable people.
Flow chart of search results
Please include Figure 1
The nature of the evidence
The characteristics of the studies are summarized in Table 1 and Table 2. The evidence generated was of three different types. Most studies (14) based their findings on individuals’ directly reported experiences of participating themselves in a research study. The other studies approached the topic indirectly by enquiring about attitudes [15, 16], by enquiring about participants’ settled opinions or ways of thinking about research; or asking hypothetical questions [17–20] to respondents where they are asked about a supposed situation where people who receive palliative care are invited to participate or are participating in research. These categories are denoted in Table 2 as D (direct), A (attitudinal), and H (hypothetical).
All the studies that enquired about attitudes to research participation, or hypothetical research participation used prospective designs (6). The studies that examined experiences of research participation used a retrospective design (14). They either employed descriptions of interventions or studies; analysed data recording the research process (6) [21, 22, 25, 32–34]; or a study was nested within the primary study to examine participants’ experiences of the research process (8) [23, 24, 26–31].
Types of studies and their findings
Five studies considered participation in palliative care research in general. Eleven studies enquired about issues related to specific research methods that included questionnaires, qualitative interviews, focus groups, or observational and participatory methods. One study reported on feedback to an observational design from epidemiology or the social sciences . Two studies assessed the views of those involved in clinical trial research [17, 34].
Participation in research in general
The studies that assessed participation in research in general reported willingness towards participation mostly from hospice environments. Cassarett et al. reported on a nation-wide telephone survey to define the research that was conducted in hospices across the US, to assess hospice organisations’ perceptions regarding obstacles to research, and to identify their research priorities. They found that participation in research was related to hospice characteristics: urban location, academic affiliation, larger census and having an inpatient unit. This raises concerns about the generalisability of these results . Kirsch et al. conducted a cross-sectional anonymous survey with hospice staff to gain insight into staff’s attitudes, beliefs and values about research with the patients they cared for and their families. They found that staff were largely supportive of research, but they acknowledged a mix of protectionism towards patients and not having enough time for research as barriers to participation . Williams et al. focused on hospice patients and carers and compared their interest in research with ambulatory senior citizens. The patients and carers showed slightly lower interest in research compared to senior citizens: in survey research, (45% and 57%) and in therapeutic research (46% and 60%) respectively .
Two other studies focused on a hospital setting. Daly et al. showed the willingness of newly diagnosed cancer patients (70%) to participate in psychosocial cancer research through the evaluation of a data registry to advance research . Christ et al. described the establishment of a designated social work research unit in a major academic cancer research institution. They described the process of developing understanding and collaboration among clinicians and researchers, which required time and exposure to each other’s goals, values, skills and limitations .
Participation in research using surveys, interviews, observational or participatory methods
All except two studies [19, 20] were evaluations of actual research experiences and were retrospectively addressing participation in research on EoL issues, which could involve sensitive issues. Most studies made use of questionnaires and qualitative interviews or both, and two of focus group discussions [19, 32], of which one applied a participatory approach . One study reported on feedback to an observational design .
These studies were carried out in a variety of settings and were all targeted at patients or their informal carers’ participation. In Kendall et al. a sample of researchers was included .
The studies all reported a greater number of positive than negative results. Where the two studies using a hypothetical approach enquired about preferences [19, 20], the other studies either focused on patterns of recruitment to uncover participants’ views on research [24, 27, 30], or on the experiences of patients and carers taking part in research that used social science methodology, or both [24–27].
Barnett showed that the majority of patients found a one-hour long interview a positive, therapeutic experience . In Davies et al. most patients (88%) and carers (96%) agreed to be interviewed, while 63% reported comfort from being interviewed and only 5% disliked the experience. But they reported that there was a 95% follow-up rate of the interviews . In Emmanuel and Emmanuel, 87% of approached patients and 97% of carers were interviewed about death, dying and bereavement. 88% of patients and of 89% carers reported little or no stress from participating in an interview. The results on helpfulness were more equivocal with 16% of patients saying it was very helpful, 29% somewhat, 49% little or not, and 19% of carers saying it was very helpful, 34% somewhat, and 44% little or not .
Gysels et al. found differential patterns in decline and acceptance of interviews by patients with different conditions and across settings. Patients and carers were capable of deciding whether to participate in interviews and negotiate how they wanted this to happen . Takesaka et al. reported that 22% experienced distress due to being interviewed about EoL issues . Pessin et al. showed that 75% of patients did not experience psychosocial research as a burden, and 68% found it moderately to highly beneficial . Wright et al. assessed the involvement of patients as co-researchers which did not cause them distress, however some logistical and methodological problems appeared . A study from thanatology assessed risks and benefits associated with in-depth interviewing. Patients found discussing sensitive issues painful, but they said they benefitted by talking with a researcher and thought they could do this more openly than with a health professional. The researcher reflected on the impact of this research on herself, and recognized sadness but also meaningfulness . In Kendall et al., people affected by cancer and researchers found that people nearing the EoL want to be offered the chance to participate in research, provided it is conducted sensitively . Phipps et al. compared consenting participants and refusers in observational research and found that those participating thought they had more to gain from research compared with refusers (=0.04), and felt that pain was more of a problem (p < 0.001) . Dobratz et al. explored issues and dilemmas related to non-participation, attrition, and needs for assistance in research with home hospice participants. They found that 28.8% required assistance because of physical impairments, and 14.1% were unable to participate or lost to the study because they suffered from cognitive disturbance, and physical distress. Those who completed testing expressed gratitude at being able to contribute information that they believed would benefit others .
One study that applied a hypothetical design explored cancer patients’ views and preferences on taking part in longitudinal questionnaire-based research studies. This study shows the importance of flexibility in planning interviews as fluctuating symptoms may affect the ability and willingness to participate in this population . In a study using hypothetical questions, patients very near to death expressed a positive wish to participate in research .
Participation in clinical trials
Two studies were identified that considered research participation in clinical trials. One was targeted at patients with cancer and used a hypothetical approach  and one at hospice staff obtained empirical data .
White et al. found that patients were interested in participating in randomised controlled trials (RCTs) that concerned symptom control and relatives supported their participation in such trials. Over 75% of patients expressed altruistic views as a reason for participation. Many were prepared to complete short questionnaires, accept extra medications, investigations, hospital visits or admissions within a trial context. However deterrents were: concepts of ‘randomisation’, ‘placebo-control’ and ‘blinding’, and the possibility of side-effects .
Grbich et al. documented the impact of a trial on hospice staff which initially produced high levels of stress, but diminished over time. The stress was replaced by enthusiasm for the achievements of the study and sadness at the loss of benefits post-trial .
Characteristics of participants and the study designs
The included studies revealed various characteristics that were either related to the individuals or groups investigated or were associated with the specific area or type of research.
Dobratz et al. provided insight into the problems commonly experienced by palliative care patients involved in research. Cognitive disturbance, and physical distress often played a role in attrition and in needing assistance to enable participation . This same finding was made by [Shipman et al. 20](see above) . Phipps et al. compared ‘consenters’ with ‘refusers’ (see above), showing that consenters thought they had more to gain from research and felt that aches or pain were more of a problem for them .
Williams showed that young people were more interested in taking part in research than older participants, but there was no difference in attitudes between older hospice and non-hospice patients . Increasing age also appeared in White et al. as the only negative factor indicative of less involvement in trial research .
Gysels et al. showed differences in motivations to research participation by patient groups, and between patients and informal carers . Takesake et al. showed higher distress for women, younger carers and family members of patients with cancer. They caution however that although these associations were strong, the reasons for them are uncertain .
Emmanuel and Emmanuel identified individual characteristics of patients associated with the impact of a research interview. People with pain, those who are able to find more personal meaning in dying, and being less at ease with talking about dying, were significantly more likely to report more stress. Patients from ethnic minorities, those who were anxious about the end of life, and those who were more spiritual or serene were significantly more likely to find an interview helpful .
Terry et al. showed that patients very close to death provide unique understandings as death nears .
Study design characteristics
Cassarett et al. showed that hospice organisations were more supportive of research that they considered to be concerned with issues directly relevant to hospice care: pain management and referral to a hospice . Shipman et al. enquired about the best format with which to conduct longitudinal studies using questionnaires. Patients preferred health professionals known to them to contact them about a study; they preferred face-to-face interviews at home but they were prepared to take part in them in hospital . Face-to face interviews were also the preferred method by patients in the studies by [Daly et al. and Terry et al. 19, 22].
There is also evidence on the feasibility of conducting interviews with carers and patients jointly or separately. The study by Davies et al. outlined the disadvantages of joint interviews , and Takesake et al. showed that joint interviewing did not cause distress in the study’s sample . In three papers the benefit was shown of a researcher who was not involved in the daily care of a patient to whom patients could open up and give their genuine view on events [24, 27, 33]. However, patients experienced distress when asked to discuss death [26, 29].
Grbich et al. showed the potential to improve the conduct of a large RCT by carrying out a parallel qualitative study .
Willingness to participate in EoL research
All of the studies that evaluated willingness to participate in EoL care research showed positive outcomes across the different parties involved [15–20, 22, 25, 28]. The majority of patients were willing to take part in research [15–20, 22, 25, 28] and their informal carers were supportive of this [17, 18]. Also hospice staff and hospice organizations were found to have positive attitudes towards participating in research [15, 16]. The studies that reported on health professionals showed the most equivocal results [15, 34].
The studies illuminate the factors that influence willingness to participate in research. Disability, physical distress and cognitive impairment formed barriers to individuals’ participation in research studies. However, some studies showed ways to enable patients to participate by providing assistance to complete questionnaires and to use flexible and responsive approaches to data collection to accommodate patients’ physical and cognitive abilities . Participation for patients was often experienced as empowering and this was expressed through gratitude towards those who made it possible to contribute .
Experiences of participating in EoL research
All 10 studies providing quantitative results showed that participating in research at the EoL was a positive experience for the majority of patients and their carers [20, 23–27, 29–32]. Benefits of participation were identified as being therapeutic or giving comfort, especially in interviews [23, 24, 26, 27] which provided the opportunity to reciprocate for good care, and to contribute to service improvement. However, there was a minority who experienced involvement in research as distressing and this was related to either, characteristics of the participants, the type of research, or the way it was conducted.
The studies that evaluated actual experiences of research participation provided particularly useful insights on the benefits and risks of working with patients and their carers at the EoL and suggest future directions for research. The studies focusing on attitudes were helpful in identifying participants’ beliefs and values about research. Future attitude studies could widen their focus and explore how these values are informed and used. Studies that used hypothetical questions are important in difficult-to-research areas but one needs to be aware that they provide a normative view on reality. Most studies have approached patients with a set of hypothetical questions, particularly those involving trial research. Examining actual experiences as patients go through these trials – what motivated them, and the challenges encountered – is likely to shed a different light on this.
Age appeared to be a factor that influenced attitudes towards participation in research [17, 18]. Differences were found in the motivation to participate by patient groups  and individual characteristics  and despite the strong associations found in quantitative studies there are no discernable explanations . The identified patterns of views and impacts of research require exploration beyond the typical demographic variables, and designs are needed to capture the complexity of concerns and interactions between those involved in research. A way forward is the development of mixed-methods designs, which offer multiple opportunities to verify data and its interpretation. However, these designs require thorough planning from the outset and reflexivity in the way they are conducted. Palliative care encompasses those patients and carers at diagnosis through to death and beyond, and as such longitudinal studies could provide insight in to how levels of willingness and experiences with research change throughout these stages, and could help determine the best ways to conduct studies at different stages.
Gaps in the evidence-base
We did not find any evidence of participation in research from beyond the US, the UK or Australia. This reflects the concentration of palliative care services and research centres in these countries . Studies from beyond the developed world will add to the findings from this review and are expected to show how different environments and values impact on attitudes and experiences with research. This geographical bias also determined the study settings, which were mainly in-patient hospices or hospitals. Other settings such as the home and the community were under-represented.
The majority of the studies focused on patients with cancer, while there were only a few studies that included other advanced diseases. It is important to include the range of diagnoses that have palliative care needs as different patient groups can have distinct concerns regarding research. The gap in knowledge about cancer patients in the advanced stages of illness seems to be addressed since the review undertaken by [Todd et al. 12]. Most of the studies identified in this review targeted the later stages of illness as concerns regarding research were expected to become more pronounced nearer to death. There were studies that did not precisely describe the stage of illness which is necessary to compare experiences within the heterogeneous population who require palliative care. Given the findings regarding age as a significant influencing factor, future studies could include older people.
As the ethical discussion about research participation revolves for the most part around patient vulnerability, the studies included here have mostly investigated patient views. A variety of other stakeholders were represented, but more evidence is needed about the roles they fulfil and the particular contribution of each of these parties. The views of members of ethics committees or funders of EoL research were absent. Also studies that focus on interactions between different stakeholders could reveal perceived impacts of research, areas of conflict or opportunities for mutual understanding.
Research participation as teamwork
Several studies showed that research at the EoL is best realised as a collaborative effort [21, 24]. Division of tasks between researchers and clinicians can lead to frequent and extensive discussion that provides opportunities to develop a realistic appreciation for each other’s capabilities and limits . Patients and other participants in research need to be engaged as early as possible [15, 35]. This requires a campaign of public education  regarding the importance of research . Health professionals concerns also need to be addressed [15, 21, 34, 37, 38]. Conditions conducive to research are required, including: the sharing of skills, supportive institutions [28, 34, 39], formal partnerships between care providers and academic entities and extra resources [32, 34, 38]. Flexibility is required in the policies, protocols  and evaluations made by ethics committees in order to counter administrative constraints and protective attitudes that often hinder research [40, 41]. Research in palliative care often requires mixed methods or a diversion from standard procedures (for example when dealing with informed consent), which are circumstances that ethics committees are generally not used to working with [40, 41]. A ‘relational ethics’, where researchers and ethics committees work in partnership, has therefore been promoted as a suitable model to enable valid and sensitive research in palliative care [40, 41]. Participatory designs were found particularly suitable to actively include a diversity of participants who can determine the design and conduct of research [28, 30, 34, 39].
Research with care
This review shows that the ethical concern about patients at the EoL as too vulnerable to participate in research is often unjustified. However, the studies providing evidence about participation in research should not simply dismiss these concerns that have grown through debate and argumentation in ethics. Rather, they need to put these side by side and critically assess how the findings can inform the best way forward. Therefore, we analysed the rationale that underlies the reasoning about research in EoL care. We investigated the normative framework that organises the problematic issues pertaining to EoL care research from the entire body of evidence. The resulting conceptual model frames our understanding about what research is and does in EoL care settings. The model shows the ‘taken for granted’ knowledge on which respondents base their actions and develop their attitudes.
The ethical arguments frame research and care as if they have oppositional goals and values (see Figure 2). This is what is called the deontological-utilitarian conflict where the goal of enhancing outcomes for all people needs to be balanced against the goal of ensuring optimal comfort and benefit . People should in other words not be subjected to greater risk than what they would endure in routine assessment or treatment. Research is understood as instrumental, using the patient and others taking part in it, as a means to an end. Care, on the other hand, has the best interests of the patient and others in mind. Research is for the benefit of future patients, as it is argued that patients will already have died by the time that the findings will have materialised in interventions, while care can be provided immediately and directly. Within this discourse, research is perceived as active, and those who participate in it as passive, as the latter are presented as being subjected to research. It is objective, and removed, while care is personal and close. Stakeholders representing different territories should stay on their own sides to avoid misunderstandings. Researchers who transgress these boundaries of research and care – for example, those who collect data through qualitative methods, which inevitably involves close communication and empathy, are told to take all precautions to protect patients from deception. In the same way, the clinician-scientist is challenged as inappropriate, as this could lead to coercion based on the trust developed between the health professional and the person being cared for. The opposition is reflected in the metaphors attached to these domains where positions are crystallised in terms of ‘warm’ care and ‘cold’ research.
Where objections to research were expressed in the studies under review, these came from the concern that research would detrimentally affect the care that patients need. For example, the protective attitudes by hospice staff towards their patients in [Kirsch et al. 15] gave evidence of such a concern, or the apprehension patients expressed towards terms associated with trial research in [White et al. 17], where terms such as ‘randomisation’, ‘placebo-control’ and ‘blinding’ deterred patients from participating in research. This reflects similar concerns found in an earlier study of patients’ and the public’s view of the process of randomisation, where the role of chance by using computerised procedures – equivalent to tossing a coin – were used as a way of determining treatment allocation in the context of life-threatening research, and were found to be upsetting and trivialising .
Successful studies were the result of research that was conducted with care, with for example tailored data collection to accommodate patients’ disabilities and to enable their participation . The studies using a participatory design were also based on mutual recognition of each other’s strengths and weaknesses and were conducted within a teamwork-based setting . Such studies suggest that a different model of research in EoL care is needed (see Figure 3). When research is embraced by care, many of the ethical objections become invalid. The issue of being used as a means to an end, or in other words as a mere instrument for research goals, does not apply any longer when research is embraced by care. A collaborative model with equal input of all the parties involved in research, who share a concern to care will lead to sensitivity to the needs that arise during the conduct of research studies. This also guarantees the immediate benefits that involvement holds for patients, such as empowerment, self-validation or the opportunity it offers for altruism [13, 19]. A relational approach where the ideas of different stakeholders from a diversity of disciplines are exchanged, can lead to an intersubjective view on what is investigated and the way research is conducted. The concern about granting the right to participate in research shifts towards mutual responsibility among those involved and is realised through open dialogue. The attention then focuses on what support and supervision is needed in order to optimise participants’ contributions [8, 17] and this can lead to a change in culture in terms of attitudes towards research [21, 34].
This paper set out to systematically assess the evidence relating to barriers and solutions to participation in research at the EoL. However the review is limited as only original research studies were included. Papers presenting ethical debates on participation within EoL care research were excluded. For a comprehensive overview of the field, the ethical arguments need to be read alongside this review. It is also possible that the gaps identified in the evidence-base are not neutral with regard to the aim of the study, and that they represent an indication for undetected problems in EoL research, which can lead to publication bias.
This review provides an evidence-base about ethical concerns regarding research at the EoL. Although the literature focused most on patients with cancer, a range of other participant groups were also represented, such as health professionals, carers and researchers. The evidence shows that all those involved in research are in principle supportive of research at the EoL. The group most likely to have concerns about involving patients at the EoL were health professionals.
This review showed that the ethical concerns regarding patient participation in EoL care research are often not justified. Patients are generally willing to contribute to research and the majority of those who take part in research, report it as a positive experience. Patients confirm that it brings them direct benefits. However, there was a minority of people for whom taking part caused distress, requiring increased sensitivity to individuals’ changing needs and circumstances. Assistance needs to be in place for such situations. The palliative care population commonly experience fluctuating symptoms across their disease trajectory, which can cause varying levels of physical and cognitive impairment. Therefore, research needs to be flexible and equipped with methods that can enable patients and carers to participate in research on EoL care.
Denying patients the opportunity to participate in research on the basis of an a priori label of vulnerability is paternalistic. Given the diversity of this population, patients need to be approached as individuals, to assess whether it is desirable to involve them in research. This makes the model presented here not only applicable to the EoL population, but to all potentially vulnerable people.
National Institute on Aging: Why population aging matters: a global perspective. 2007, US Department of Health and Human Services, US Department of State: National Institutes of Health
World Health Organization: The global burden of disease. 2008
A Report from the Economist Intelligence Unit: The quality of death. 2010, London: Ranking end-of-life care across the world
Casarett D: Ethical considerations in end-of-life care and research. Journal of Palliative Medicine. 2005, 8: S148-S160. 10.1089/jpm.2005.8.148.
Jubb AM: Palliative care research: trading ethics for an evidence base. Journal of Medical Ethics. 2002, 28: 342-346. 10.1136/jme.28.6.342.
Kaasa S, Dale O: Pain and Palliation Research G: Building up research in palliative care: a historical perspective and a case for the future. Clinical Geriatric Medicine. 2005, 21: 81-92. 10.1016/j.cger.2004.08.009.
Riley J, Ross JR: Research into care at the end of life. Lancet. 2005, 365: 735-737.
Beaver K, Luker K, Woods S: Conducting research with the terminally ill: challenges and considerations. Int J Palliat Nurs. 1999, 5: 13-17.
Grande CE, Todd CJ: Why are trials in palliative care so difficult?. Palliative Medicine. 2000, 14: 69-74. 10.1191/026921600677940614.
Rinck GC, van den Bos GA, Kleijnen J, de Haes HJ, Schade E, Veenhof CH: Methodologic issues in effectiveness research on palliative cancer care: a systematic review. J Clin Oncol. 1997, 15: 1697-1707.
Duke S, Bennet H: A narrative review of the published ethical debates in palliative care research and an assessment of their adequacy to inform research governance. Palliative Medicine. 2010, 24: 111-126. 10.1177/0269216309352714.
Todd AM, Laird BJ, Boyle D, Boyd AC, Colvin LA, Fallon MT: A systematic review examining the literature on attitudes of patients with advanced cancer toward research. Journal of Pain and Symptom Management. 2009, 37: 1078-1085. 10.1016/j.jpainsymman.2008.07.009.
White C, Hardy J: What do palliative care patients and their relatives think about research in palliative care? - a systematic review. Supportive Care in Cancer. 2009, 18: 905-911.
Dixon-Woods M, Cavers D, Agarwal S, Annandale E, Arthur E, Harvey J, et al: Conducting a critical interpretive synthesis of the literature on access to health care by vulnerable groups. BMC Medical Research Methodology. 2006, 6: 35-10.1186/1471-2288-6-35.
Kirsh KL, Walker R, Snider S, Weisenfluh S, Brown GM, Passik SD: Hospice staff members' views on conducting end-of-life research. Palliative & Supportive Care. 2004, 2: 273-282.
Casarett DJ, Karlawish J, Hirschman KB: Are Hospices Ready to Participate in Palliative Care Research? Results of a National Survey. Journal of Palliative Medicine. 2002, 5 (3): 397-406. 10.1089/109662102320135289.
White CD, Hardy JR, Gilshenan KS, Charles MA, Pinkerton CR: Randomised controlled trials of palliative care - a survey of the views of advanced cancer patients and their relatives. European Journal of Cancer Care. 1828, 2008: 44-
Williams C, Shuster J, Clay O, Burgio K: Interest in Research Participation among Hospice Patients, Caregivers, and Ambulatory Senior Citizens: Practical Barriers or Ethical Contraints?. Journal of Palliative Medicine. 2006, 9: 968-974. 10.1089/jpm.2006.9.968.
Terry W, Olson LG, Ravenscroft P, Wilss L, Boulton-Lewis G: Hospice patients' views on research in palliative care. Internal Medicine Journal. 2006, 36: 406-413. 10.1111/j.1445-5994.2006.01078.x.
Shipman C, Hotoph M, Richardson A, Murray S, Koffman J, Harding R, et al: The views of patients with advanced cancer regarding participation in serial questionnaire studies. Palliative Medicine. 2008, 213: 913-920.
Christ GH, Siegel K, Weinstein L: Developing a research unit within a hospital social work department. Heal Soc Work. 1995, 20: 60-69.
Daly BJ, Douglas SL, Foley H, Lipson A, Liou CF, Bowman K, et al: Psychosocial registry for persons with cancer: a method of facilitating quality of life and symptom research. Psycho-Oncology. 2007, 16: 358-364. 10.1002/pon.1091.
Barnett M: Interviewing terminally ill people: Is it fair to take their time?. Palliative Medicine. 2001, 15 (2): 157-158. 10.1191/026921601666846232.
Davies EA, Hall SM, Clarke CR, Bannon MP, Hopkins AP: Do research interviews cause distress or interfere in management? Experience from a study of cancer patients. Journal of the Royal College of Physicians of London. 1998, 32: 406-411.
Dobratz MC: Issues and dilemmas in conducting research with vulnerable home hospice participants. J Nurs Scholarsh. 2003, 35: 371-376. 10.1111/j.1547-5069.2003.00371.x.
Emanuel EJ, Fairclough DL, Wolfe P, Emanuel LL: Talking with terminally ill patients and their caregivers about death, dying, and breavement: is it stressful, is it helpful?. Arch Intern Med. 2004, 164: 1999-2004. 10.1001/archinte.164.18.1999.
Gysels M, Shipman C, Higginson IJ: "I will do it if it helps others": motivations and distress among patients taking part in qualitative studies on palliative care. Palliative Medicine. 2006, 20: 255-
Kendall M, Harris F, Boyd K, Sheikh A, Murray SA, Brown D, et al: Key challenges and ways forward in researching the "good death": qualitative in-depth interview and focus group study. British Medical Journal. 2007, 334: 521-10.1136/bmj.39097.582639.55.
Pessin H, Galietta M, Nelson CJ, Brescia R, Rosenfeld B, Breitbart W: Burden and benefit of psychosocial research at the end of life. Journal of Palliative Medicine. 2008, 11 (4): 627-632. 10.1089/jpm.2007.9923.
Phipps E, Harris D, Braitman LE, Tester W, Thompson N, True G: Who Enrolls in Observational End of Life Research? Report from the Cultural Variations in Approaches to End of Life Study. Journal of Palliative Medicine. 2005, 8: 115-120. 10.1089/jpm.2005.8.115.
Takesaka J, Crowley R, Cassarett D: What is the risk of distress in palliative care survey research?. Journal of Pain & Symptom Management. 2004, 28: 593-598. 10.1016/j.jpainsymman.2004.03.006.
Wright DNM, Hopkinson JB, Corner JL, Foster CL: How to involve cancer patients at the end of life as co-researchers. Palliative Medicine. 2006, 20: 821-827. 10.1177/0269216306073110.
Wright K, Flemons D: Dying to know: Qualitative research with terminally ill persons and their families. Death Studies. 2002, 26: 255-271. 10.1080/07481180211268.
Grbich C, Abernethy AP, Shelby-James T, Fazekas B, Currow DC: Creating a research culture in a palliative care service environment: a qualitative study of the evolution of staff attitudes to research during a large longitudinal controlled trial. Journal of Palliative Care. 2008, 24: 100-109.
Henderson M, Addington-Hall JM, Hotopf M: The willingness of palliative care patients to participate in research. Journal of Pain & Symptom Management. 2005, 29: 116-118. 10.1016/j.jpainsymman.2004.12.001.
Fallowfied L, Jenkins V, Brennan C, Sawtell M, Moynihan C, Souhami R: Attitudes of patients to randomised clinical trials of cancer therapy. European Journal of Cancer Care. 1989, 34: 1554-1559.
Fallowfield L, Ratcliffe D, Souhami R: Clinicians attitudes to clinical trials of cancer therapy. European Journal of Cancer Care. 1997, 33: 2221-2229.
Tolley DC, Payne R: Hospice Partnerships with Academic Entities: Philosophical and Historical Background and Assessment of Future Needs. Journal of Pain and Symptom Management. 2007, 33: 90-98. 10.1016/j.jpainsymman.2006.10.007.
Seymour J, Payne S, Reid D, Sargeant A, Skilbeck J, Smith P: Ethical and methodological issues in palliative care studies: The experiences of a research group. NT Res. 2005, 21: 169-188.
Koenig BA, Back AL, Crawley LM: Qualitative methods in end-of-life research: recommendations to enhance the protection of human subjects. Journal of Pain & Symptom Management. 2003, 25: S43-S52. 10.1016/S0885-3924(03)00060-5.
Larkin PJ, de Casterle BD, Schotsmans P: A relational ethical dialogue with research ethics committees. Nursing Ethics. 2008, 19: 234-242.
Fine PG: Maximising benefits and minimising risks in palliative care research that involves patients near the end of life. Journal of Pain & Symptom Management. 2003, 25: S53-S62. 10.1016/S0885-3924(03)00056-3.
Jenkins V, Leach L, Fallowfield L, Nicholls K, Newsham A: Describing randomisation: patients' and the public's preferences compared with clinicians' practice. Br J Cancer. 2002, 87: 854-858. 10.1038/sj.bjc.6600527.
The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2288/12/123/prepub
We would like to thank Mel Rumble for editing and proofing this paper.
MG and CE are funded for the MORECare project, by the UK’s National Institute of Health Research (NIHR)/Medical Research Council (MRC) as part of the Methodology Research Programme (MRP)’. Grant reference: G0802654. IJH is an NIHR senior investigator. CE holds an NIHR/Chief Nursing Officer Clinical Lectureship. This report is independent research arising from the Clinical Lectureship supported by the NIHR. The views expressed in this publication are those of the authors and not necessarily those of the National Health Service, the NIHR or the UK Department of Health.
The authors declare that they have no competing interests.
IJH identified the need to review this area of research and is PI for project MORECare, the MRC project, which this review is part of. All authors consented to study design. MG undertook the review and drafted the paper with significant input from CE and IJH. All authors approved the final version.
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Gysels, M.H., Evans, C. & Higginson, I.J. Patient, caregiver, health professional and researcher views and experiences of participating in research at the end of life: a critical interpretive synthesis of the literature. BMC Med Res Methodol 12, 123 (2012). https://doi.org/10.1186/1471-2288-12-123
- Research participation
- End of life
- Palliative care
- Critical interpretive synthesis