Patient and carer involvement in palliative care research: An integrative qualitative evidence synthesis review

Definitions and roles

Different terminology was used for those involved, for example, ‘lay stakeholders’, ⁴⁶ ‘research partners’ ⁴⁷ and ‘service users’. ⁴⁸ No one definition was applicable across all evidence and it was unclear whether interpretations of involvement were uniform.^49,50 Similarly, the term palliative care was inconsistently used, with variations in health condition, including symptoms and stages^25,51,52; service use, for example, specialist or generalist services^12,50,53,54; and inconsistencies concerning how end-of-life was defined, boundaries between different types of care and the length of time a person was considered to be in need of palliative care.^12,21,55,56 The discrepancies made it hard to conceptualise both involvement and palliative care.

The need for greater clarity of involvement roles was highlighted, particularly at the outset of studies when patients were unclear what was expected from them.^6,28,57 Evidence related to groups emphasised the need for a clear purpose.^5,58 Initial expectations of what a group could achieve were vague, roles uncertain and relationships with external bodies such as research networks confused.^5,49 Researchers needed to take time to facilitate a safe space, explain the research and clarify roles and expectations.

It could take groups a year before they started to engage meaningfully with researchers – time was needed to learn the system and how to affect change. ⁵⁹ This slowness was frustrating for some but valuable to develop aims and working practices.^50,60,61 The need for agreement by everyone was stressed and the importance of negotiating this throughout the study was described to enable ownership of the group. It was recommended that possible emotional impacts of involvement were discussed from the outset, including stressing that people need only share aspects of their personal experiences that they feel comfortable with. ⁶²

Boundaries between patients/carers and professionals were sometimes challenged as many people were in fact juggling several different roles.^63,64 Researchers had to decide how much of their own personal experiences to reveal. ⁶⁵ One described a merging between his professional and personal roles; he found himself becoming friendlier with members of the patient group and felt his professional identity was being challenged, as he shifted from being paternalistic to a more equal position. ⁶⁶

Values and principles

The values implicit within involvement were described as similar to those in palliative care,^49,67 and included transparency, accountability and honesty, for example, the importance of congruency between real and stated aims.^61,62 Respect, trust and listening were necessary to develop collaborative relationships between patients and researchers.^7,61,67 Flexibility enabled people to decide when and how to be involved – important for carers who had responsibilities and patients, whose health condition may fluctuate or progress. ⁶⁸

Particular approaches, notably Participatory Action Research, Community Based Participatory Research and emancipatory research, were suggested as being effective in promoting involvement.^69–72 These approaches used principles of equity, power-sharing and reciprocal transfer of knowledge and skills.^49,55,73 However, the evidence was limited because they had not been widely or comprehensively used.^55,69

Power was a recurrent theme, significantly the need to avoid tokenism or tick-box involvement. Sometimes roles were limited, patients had a voice but little authority for decision-making,^50,51,74 or involvement was used to meet the organisation’s agenda rather than that of patients.^71,75 Furthermore, involvement had the potential to be exploitative – thought to be important due to the potential vulnerability of patients/carers.^12,21,67,75 Even when partic-ipatory methods were used, inequalities of power and influence were reported, the experiential knowledge of patients/carers could be overshadowed by academics’ research knowledge, and the drive for research usually came from academics.^63,70,71,76 The importance of bridging different world views was highlighted. ⁷² Sometimes patients felt obliged to take part in involvement or were concerned about possible repercussions to their care if they expressed negative opinions. The ‘grateful patient syndrome’ was mentioned, where people at a difficult time in their life may be overly positive ⁷⁷ and older people were less likely to be negative. ²⁵

Patients/carers were often experiencing massive change and had fears for the future. Some had little time, non-essential commitments may be rejected or people may be pre-occupied with other things. Patients may be feeling unwell or experiencing fatigue, some had highly symptomatic conditions, a short disease duration, poor prognosis or may be facing death. Carers were facing bereavement, coping with loss or the prospect of loneliness.^{12,21,66,74,78,79} Therefore, many professionals were concerned about overburdening people or felt it inappropriate to contact patients as they may die before seeing any changes resulting from their involvement.^21,49,51,80 Gatekeeping by clinicians could also be a barrier,^{50,56,69,71,81} for example, a hospice refused to allow patients to be involved until their own Ethics Committee had cleared the study. ⁷⁷ The ethical concerns of involvement needed to be balanced with not making assumptions or removing choices from people.^21,56,66,67

Organisations and culture

Involvement was influenced by wide contextual factors including international perspectives and local politics^46,51; however, organisational culture was also highly significant.^4,50,67,82 Involvement needed to be considered a core activity, integrated throughout organisations at different levels^5,49,56,83; however, systems often were not perceived as being involvement-friendly. Competing considerations existed; pressures on staff, heavy workloads and cutbacks in the context of austerity all made involvement difficult.^4,51,60,84 Practical matters of administration, finances and travel needed to be addressed to avoid complicated bureaucratic practices.^65,84

Despite this, there was much commitment and support shown from professionals,^{4,51,57,59,64,77,84,85} including, importantly, those in senior positions.^4,50,86 Conversely, professionals were also reported as being disinterested, dismissive or displaying a lack of understanding of involvement.^5,58,60,67 Some were described as fearful or uncomfortable; they felt involvement was too impractical or unrealistic.^51,74 The representativeness of patients was questioned; some patients were thought to have their own personal agendas. ⁸² People were labelled as ‘the usual suspects’, causing annoyance and offence.^5,57 Sometimes patients were considered to be too close to their diagnosis; at other times, professionals felt their treatment occurred too long ago. ⁸⁷ There were concerns that people were not competent, that they would not consider legal, funding or other organisational constraints. ⁶⁵ These issues were poorly addressed, preventing patients from expressing their views. ⁴⁹

Nevertheless, there were encouraging progressions over time.^59,70,88 In evaluations, clinicians reported more positive views about involvement activities, which were previously small scale but now more sophisticated, focussing on larger service development issues.^50,51,84 Researchers increased their understanding of and commitment to involvement, and there was a shift in ideology away from paternalistic or superficial approaches. Professionals were surprised that patients raised issues that were small and achievable rather than large issues which required resources. They realised patients’ views were not as challenging as anticipated and involvement not as daunting as first believed, thereby also increasing their confidence.^6,51,74

Training and support

Ongoing training and support were recommended for all, including professionals.^{7,50,56,58,67} Some organisations had developed specific programmes, providing induction, training and scientific mentoring.^6,49 Training on involvement, funding, organisational structures, team building and research methods was provided.^60,86 Patients/carers appreciated being able to keep up to date with current knowledge and research. ⁸⁹ Involvement from the outset was important, to enable appropriate training and support to be developed in partnership with patients/carers.^7,54,76 The different motivations for wanting to be involved needed to be addressed, including poor or excellent treatment, wanting to give back, training opportunities and notably a desire to influence and improve both research and service provision.^5,87

However, numerous practical constraints were reported, including meeting times or the use of venues such as hospitals which may hold unpleasant memories. ⁵¹ Involvement needed to allow people to engage with ease, therefore meetings needed to accommodate those who may be ill, experiencing pain or discomfort, have limited energy or a limited attention span.^8,90,91 Informality was important, so people could move around or take medication.^62,66 Meetings/training should not last too long, breaks were useful and presentations should be kept short.^49,89,92

Flexibility and accessibility were required as people sometimes could not commit regularly or long term.^28,50,73,89 Sometimes one involvement method was used initially, for example, an advisory group, but then, if people became unwell, email communication was preferred. ²⁸ Initiatives such as keeping reflexive diaries and using de-brief time after events were recommended, if people did not want or were unable to participate in groups.^65,68,74,77 Other support mechanisms included peer mentoring or attending events with family/friends.^49,51,58,93 Advocacy may be helpful, so patients could speak on behalf of others who may be too unwell. ⁵⁰

Not every involvement method was suitable for everyone, each brought advantages and disadvantages. The use of several methods, adapted to suit different communities, was useful to enable a range of people to be involved.^{6,28,56,65,67} Methods included newsletters, website stories, talks at organisations and other outreach methods, telephone and email. ⁶⁸ Developments in IT had made virtual interaction more feasible, therefore not requiring physical presence at meetings – online training, video conferencing and online communities were all suggested.^8,47,49 One study had developed an online involvement forum.^6,94

Networking and groups

A centralised networking resource for professionals and patients/carers was suggested, to provide co-ordination, reduce duplication, and share information and learning. Professionals highlighted the importance of building alliances and linking to local organisations, for example, patient forums.^28,50,51,82 Patients wanted assistance with developing new groups, particularly patient-led organisations, and gaining mutual support.^5,25,28,84

Patient/carer advisory groups were common involvement methods,^{62,70,71,74,77,95} with members often also active elsewhere, for example, involved in service provision.^82,83 There were particular challenges regarding the stability of patient groups, including a lack of funding.^58,80 Clinicians could help establish and publicise groups, and provide ongoing support, for example, free meeting space. ²⁵ However, it was important for professionals to facilitate group collaboration, not discriminate between patient groups, and to step back to allow patients to implement their own agendas. ²⁵

Some people were initially anxious on joining a group; it took time for relationships to develop and members to feel comfortable. Several groups showed an element of peer support – not the intended purpose but often needed due to the nature of palliative care.^5,25 However, it was difficult to sustain the long-term survival of groups and the loss of influential members caused groups to destabilise.^25,87 There was a need for continuous recruitment and succession planning.^50,58

Tensions were reported when individuals dominated discussions. Often conflicts were self-managed, but it was important to give time to these process issues.^25,58,62,86 Facilitation was sometimes demanding because when people spoke of their personal experiences, it could be hard to move the conversation on.^7,92 The composition of groups was discussed. Mixed panels of professionals and patients provided better networking opportunities but could be harder to facilitate to enable patients to have a meaningful contribution.^60,86 It may be helpful if patients outnumbered professionals ⁸⁵ or if the chair was a patient/carer.^51,82 Some groups aimed to be more participatory in approach but this was unusual and required increased resources.^48,67,80,96

An evolutionary process of group development over time was reported, as knowledge, expertise and confidence grew.^{6,50,59,60,83} Initially group members were engaged mainly in consultations, for example, reviewing patient information. They then undertook more collaborative involvement, such as becoming peer researchers or co-applicants on funding bids.^{65,74,84,96–98} As groups gained experience, they established more influential relationships with professionals, took more pro-active roles in decision-making, and contributed to policy and development.^{4,49,59,60,82,96} Some also started thinking about their own research ideas as patient-led projects.^47,99

Perspectives and diversity

Differences in perspectives were common, mainly between patients/carers and the dominant perspective, professionals.^{28,46,51,84,92,100} There was a need to make the patient/carer voice more visible throughout, including in dissemination.^{28,50,61,71,75,83} It may be helpful to view differing perspectives as not in competition, but as different opinions that all needed to be understood to reach a solution. ⁶⁵ For example, in studies where patients undertook data analysis, different themes were identified from that of researchers alone.^76,80,93,101

Perspectives were particularly relevant for research that used Nominal Group Techniques, including Priority Setting Partnerships (PSPs) or similar,^{28,61,68,79,90,91,102–104} or Delphi studies.^105,106 PSPs were often driven by clinicians suggesting a need for greater involvement, to enable patient/carer priorities to be included rather than merely validating those identified by professionals.^75,103,106 Facilitators of groups/workshops needed to ensure that no stakeholder groups dominated or were excluded. For example, there may be conflicts between patients and carers.^12,61,77

Greater diversity in involvement increased the credibility of research findings and/or obtained more relevant priorities; however, difficulties were often reported.^7,56,103 The exclusion of some groups – people from Black, Asian and minority ethnic (BAME) backgrounds, those on a lower income or with a non-cancer condition – reflected their exclusion in palliative care service provision.^49,77 For others, older people or those in rural communities, practical considerations of transport and access were exacerbated.^6,12,107 Younger people and those with rare conditions could also be excluded,^62,96 and some people were self-conscious or reluctant to mix with others, for example, people with laryngectomies. ²⁵ The exclusion of those at the end of life was also highlighted,^12,99 although one PSP had focussed solely on this group^90,108 – other PSPs used weighting mechanisms to ensure patient priorities were reflected.^79,103 Recruitment from marginalised communities required greater resources and different approaches, for example, outreach methods or regional recruitment for those with rarer conditions.^7,25,49

Several papers commented that the ‘right type’ of patient seemed to be, for example, in better health, English speaking, had access to IT and available in the daytime!^62,87 Such patients could be recruited to the exclusion of others as they were more accessible to professionals, sometimes hand-picked with no open recruitment processes.^49,87 Often recruitment was solely from existing patient/carer groups, thereby creating further exclusion as most people are not a member of any group.^12,25,49,75 Groups served some conditions better than others – some had no or few groups, in particular, rarer conditions.^25,96,102

The word ‘representative’ caused difficulties; aiming for diversity rather than representativeness was recommended.^21,65,75,82 Demographic factors and health conditions could be used for recruitment, along with those with different experiences and attitudes, for example, ‘patients who volunteer, enthusiastic patients, hard to reach patients, alienated and sceptical patients, isolated patients, patients who are very ill, carers, family members and friends’. ²¹

Relationships and communication

It was important for professionals to develop collaborative relationships, have ongoing conversations and establish a two-way dialogue – not just email asking for comments.^84,93,97 Sometimes it was difficult to build relationships^6,94 or it took longer as communities became involved in their own ways, in their own time.^57,69,72 There was sometimes mutual suspicion between patients/carers and professionals with a resulting need to develop a common agenda and work towards unified goals. ²⁵ Participatory approaches were suggested, whereby different groups can come together to co-develop a vision for research. ⁷² A willingness to remain open, listen, revise expectations and try new things all helped. ⁸⁷ It was suggested the best way for professionals to learn was to interact with people with life-limiting conditions or experiencing bereavement. The ability to listen to ‘awful stories’ was thought to be valuable. ⁵⁰

Communication needed to be accessible, as complicated technical language and jargon could alienate some people.^{6,7,50,66,91,109} Researchers were able to communicate regularly with academic colleagues; however, this was not possible with patients/carers. Lack of feedback was therefore sometimes an issue, resulting in patients feeling frustrated with not knowing what had happened to their views or if any action had resulted.^{28,56,57,65,68} It was important to keep patients/carers regularly updated, for example, via newsletters.^77,80 Emerging findings could also be provided in case patients/carers were unable to receive final study results.^49,89

Relationships formed or were strengthened, teamwork was developed and barriers broken down. ⁵⁰ There was increased dialogue and communication, contributing to organisational change. Boundaries were positively blurred between patients/carers and professionals, as involvement began to be perceived as a joint process with shared responsibilities.^51,97,98 Throughout studies, patients/carers challenged assumptions, asked questions and influenced professional complacency. They brought common-sense to meetings and kept discussions grounded in reality.^{50,74,80,92,110}

Emotions and impact

Involvement in palliative care could have a profound effect and high emotional cost. Sometimes it affirmed people’s experiences of disrespectful services or brought back memories of loved ones who had since died.^5,28,62,74 The ill-health and death of fellow group members or participants caused distress.^63,66,74,109 One paper reported that ‘people shed tears at every event’. ⁶³ Sometimes patients/carers had to withdraw from involvement as a result.^21,109 Conversely, few professionals described any such emotional attachment. ⁸²

Involvement itself also caused distress, as activities could be carried out poorly, for example, it could be disempowering to sit on an intimidating committee.^25,58,75,77 Sometimes patients experienced unrealistic expectations from professionals regarding their health or available time.^49,71 People could feel overwhelmed by the pressures of involvement, issues connected with power imbalances or upsetting attitudes of professionals, for example, when health conditions were discussed insensitively.^5,57

However, many more positive impacts were reported. Patients commonly described involvement as giving them a focus, motivating them to carry on in life. ⁵ Coming together in groups was reassuring and confidence boosting; being with others with the same health condition could be beneficial and some were seen as role models.^5,50,98 Other social benefits included networking, making friends and gaining mutual support. Working together, including with professionals, brought a sense of solidarity and comradery.^57,66,80 Patients/carers increased their knowledge of research and ethical issues.^28,57 Researchers gained a greater understanding of palliative care, the experiences of the communities they were working with and what it was like living with particular health conditions.^46,72,74

Patients found that involvement improved their relationship with their illness, helping them to come to terms with it. They learnt more about themselves, their health condition and how to better live with it.^57,109 It gave them a sense that something positive had come out of the sometimes negative experiences of diagnosis and treatment and helped to dispel feelings of hopelessness. Some commented that involvement had enabled them to think more about palliative care and to take responsibility for planning their own death.^5,50,57,66 Carers too found that being involved eased difficult feelings regarding their experiences. ¹¹¹

Other psychological benefits included increased self-confidence and feelings of empowerment. Involvement was seen as a positive way to keep active and combat depression and loneliness. A sense of personal achievement was gained and people felt recognised and valued for their contribution.^5,57,83 Their expertise in their own condition was acknowledged but they were not just being defined as a patient, they felt helpful and able to do something again. Sometimes, people went on to be involved in further research, study or other activities.^66,85,110

Despite the challenges, patients/carers were still keen to be involved and reported strong emotional attachments to involvement.^22,48,82,87 Members of one group described how they had not considered giving up, despite the fact that several members had died. They described their involvement as ‘fun’. ⁵