Prioritising key motivators and challenges influencing informal carers’ decisions for participating in randomised trials: a Study Within A Trial (SWAT-55)

Background: Family members, or others, often assume the role of informal (unpaid) carers of people with chronic illnesses. Care-giving, however, can impact profoundly on the quality of life of caregivers and can cause carer worry, stress and guilt. Implementing interventions that positively affect the lives of carers is important, however, carers as a group are often difficult to reach. We embedded a study within a pilot-feasibility study of a mindfulness based intervention for carers of people with chronic illness to determine the key motivators and challenges influencing informal carers’ decisions for participating in a trial, and to prioritise these to inform future trial design and conduct. Methods: We conducted a Study Within A Trial using a multi-method approach involving interviews with participants from the ‘host trial’ and data from systematic reviews to develop a survey that was distributed to informal carers across Ireland. The survey consisted of 28 motivator and 17 challenge statements. Participants rated how important they thought each statement was when deciding to take part in a trial on a 5-point Likert Scale (1-very unimportant to 5-very important). Mean scores and standard deviations were calculated for each statement and arranged in descending order to provide the final priority lists. Results: Thirty-six carers responded to the survey. Helping to create awareness about carers was the top ranked motivator, followed by four study design statements related to the time at which the study occurs, the study location, format of delivery and venue. The least important motivator related to how carers were invited to take part in a study. Difficulties in planning due to the caring role emerged as the most important challenge for carers, followed by being unable to leave the care recipient on his/her own. Conclusions: Insight into decision-making for research participation will assist trial developers tailor trial processes for informal caregiver populations. We recommend that

trialists consider these motivators and challenges when designing future trials involving informal caregivers so as to enhance trial feasibility and success.

Background In 2016 the Health Research Board-Trials Methodology Research Network (HRB-TMRN)
Ireland, in collaboration with the James Lind Alliance United Kingdom, participated in a priority setting partnership (PSP) to identify and prioritise unanswered questions around trial recruitment (the PRioRiTy study) [1]. The PSP culminated in a face-to-face meeting, attended by key stakeholders (members of the public, recruiting clinicians and researchers), where a top-10 list of unanswered priority questions on trial recruitment was agreed and ranked in order of importance. Ranked highly was a question on key motivators influencing members of the public decisions for participating in randomised trials (PRioRiTy question 6) [1].
Family members, or others, often assume the role of informal (unpaid) carers of people with chronic illnesses. Care-giving, however, can impact profoundly on the quality of life of caregivers and can cause carer worry, stress and guilt. Family members providing unpaid care have been described "…as a hidden patient group…" [ 2]. Mindfulness based interventions have the potential to positively impact on the lives of carers by reducing caregiver depression, anxiety and stress, and by improving carer quality of life [3]. A randomised pilot-feasibility trial was planned by a research team in Trinity College Dublin to test a mindfulness based stress reduction (MBSR) intervention, compared to no intervention, for informal carers of people with chronic illnesses in one region in Ireland.
As informal carers represent a geographically disperse discrete group within the general public who might face specific challenges when deciding to take part in a trial, the MBSR trial, hereafter known as the 'host trial', presented an excellent opportunity to embed a Study Within A Trial (SWAT). The SWAT was designed to ascertain and prioritise key motivators and challenges influencing informal carers' decisions for participating in a trial, thus helping to advance the design and conduct of future trials in this, and other, similarly discrete populations. The SWAT protocol was prospectively registered with the SWAT repository as SWAT-55: http://www.qub.ac.uk/sites/TheNorthernIrelandNetworkforTrialsMethodologyResearch/FileStore/File

Context of SWAT-55
The SWAT-55 host trial was a planned pilot-feasibility randomised trial (ClinicalTrials.gov identifier NCT03048565) based on the following PICO (population, intervention, comparator, outcomes); Population: Informal carers, defined as a person (relative, neighbour, friend or significant other) providing personal help, support or care for an individual (adult or child) with a chronic illness and who were not a paid health care provider. A person with a chronic illness was defined as an adult or child with a diagnosed condition of six months duration or longer. Access to carers was through Family Carers Ireland, a registered charity representing carers in Ireland (https://familycarers.ie/about-us/) who agreed to distribute letters of invite via email to carers in the Dublin region.
Intervention: A MBSR programme delivered over 8 weeks (two hours/week) by a trained mindfulness teacher, with participants encouraged to practice mindfulness exercises between sessions.
Outcomes: Clinical outcomes were baseline and post intervention (up to 2 weeks from end of programme and 6 months follow-up) stress, mindfulness and quality of life data collected using selfreport questionnaires. Pilot measures included recruitment processes, data collection methods and intervention delivery. Feasibility outcomes were recruitment success, time to recruit, attendance at classes, dropouts and participant satisfaction.
Host trial sample size, randomisation and recruitment The planned sample size for the host trial, based on a recommended sample size for pilot studies of 30 per group [4] was 80 informal carers, or 40 per group allowing for a 25% attrition rate (10 participants per group) at 6-months follow-up, randomised on a ratio of 1:1 using a computer-randomised number generator. Ethical approval to conduct the study was granted by the Research Ethics Committee of the lead researcher's university.
Recruitment to the host trial commenced in March 2017 with intervention delivery planned for April-June 2017. SWAT-55 was planned to commence in June 2017.
An invitation to participate in the host trial was emailed to 538 Dublin based registered Family Carers Ireland members. Intervention delivery was initially planned as face-to-face, however, by the end of August, despite efforts, two carers only were recruited and randomised to the intervention group, one of whom had to withdraw subsequently because he/she was unable to attend the intervention sessions. Following a Trial Steering Group (TSG) meeting a decision was made to deliver the intervention in an online format and open the study to a wider national base. Ethical approval was granted, and an updated study invitation letter was circulated in September 2017. A social media invitation was also posted to the Family Carers Ireland Facebook page. By October 2017, 11 carers only were recruited to the study; five in the intervention group and six in the control. Following a further TSG meeting a decision was made to change the trial design to a before and after study, thus converting the host trial to a non-randomised pilot-feasibility study [5], on the basis that a randomised trial was not feasible at recruitment level. Participants recruited to the control arm of the original trial were subsequently offered the intervention, and the revised design was further advertised. Between mid-October and end of December 2017, 17 carers were recruited to the study, 15 returned baseline data, of which seven returned end of intervention data (quality of life, mindfulness and stress outcomes). These challenges further emphasised the need for SWAT-55 so as to explore the reasons why informal carers, as a discrete group within the general population, may, or may not decide to take part in a randomised trial.

Design
A multi-method two-phase study was conducted. Phase 1 involved a series of face-to-face interviews with a sub-sample of participants from the host trial, and a review of systematic reviews that assessed motivators or barriers for taking part in trials [6][7][8][9][10][11][12].
Resultant data from both the interviews and systematic reviews were used to collate a list of key motivators and challenges. Phase 2 was a national survey of how important these key motivators and challenges were to informal carers in making a decision on trial participation.

Phase 1
The host trial consent form provided participants with an option to agree to future contact for follow-up studies arising from the trial. Of the 17 carers recruited to the host trial, 11 agreed to future contact. These 11 carers were purposively contacted and invited to take part in an interview designed to ascertain their views of and reasons (motivators and challenges) for participating in a trial. We aimed to recruit 10 participants for interview, however only two came forward. The interviews, which were semi-structured using an interview guide were held at a mutually agreed time and venue. The interviews were recorded, transcribed verbatim, and analyzed, based on a thematic analytical approach, to determine common categories of i) motivators and ii) challenges, for use in phase 2 of the study.
Seven systematic reviews [6][7][8][9][10][11][12] of studies of reasons for participating in trials were identified in an evidence mapping exercise conducted as part of the PRioRiTy study [1] based on a search of MEDLINE, EMBASE, Cochrane Database of Systematic Reviews, Social Sciences Citation Index and ERIC using a combination of search terms from the Cochrane systematic review of strategies to improve recruitment to trials [13] and from the ORRCA project [14]. Data related to motivators and challenges for participating in trials were extracted from these reviews and combined with the data from the interviews to develop categories, inclusive of aligned motivator/challenge statements associated with these categories, for use in the phase 2 survey. An 'audit trail' of developing categories from initial codes was maintained to ensure confirmability of the process. Survey development occurred between January and June 2018.

Outcome measures
The study outcomes were a prioritised list of i) motivators and ii) challenges, based on the level of importance that respondents assigned to each of the survey's motivator and challenge statements.

Ethical approval
Ethical approval for SWAT-55 was granted by the Research Ethics Committee of the lead researcher's University.

Data collection and analysis
Quantitative analytical techniques were used to aggregate individual's ranking of motivators and challenges. Each participant was asked to rank each motivator and challenge on a 5-point Likert scale of 1=very unimportant, 2=somewhat unimportant, 3=neutral/unsure of importance, 4=somewhat important, and 5=very important. Mean scores and standard deviations (SD) for each item were calculated. The items were subsequently arranged in descending order of importance based on the mean and SD scores attributed to them. The priority list of informal carers' motivators and challenges for participating in trials was determined.

Results
Interviews, systematic reviews and survey development The interviews, involving one male and one female of the host trial, were of 21 and 38 minutes duration, respectively. One participant provided full-time care, and the other part-time care. The care recipients were a child with cerebral palsy and an adult with multiple sclerosis. Although only two carers participated in the interviews, both provided valuable data for survey development. Table 1 presents examples of the categories, and associated codes, which emerged from the analysis of the interview data. The motivators and challenges data extracted from the systematic reviews, some of which overlapped with the emergent categories from the interview data, are presented in Table   2. Using the results of the interviews and the systematic reviews, core categories, with related motivator/challenge statements, were developed for use in the survey. Figure 1 illustrates some examples of these. The final survey consisted of five core motivator categories with 28 associated statements and two core challenge categories with 17 associated statements (Additional File 1). The motivator categories were; carer identity (3 associated statements), study design (12 associated statements), altruism/common good (4 associated statements) personal interest (6 associated statements) and study information (3 associated statements). The challenge categories were personal risk (11 associated statements) and study design (6 associated statements).

Insert Figure 1
Survey findings Thirty-six informal carers returned a completed survey. The majority of respondents were ≥ 36 years of age, female, and educated to leaving certificate level, or beyond (Table 3). When asked to indicate with whom they lived, some respondents' ticked more than one response option (e.g. lived with partner and children). Two respondents indicated living on their own, and the remainder reported living with a partner (n=22), their children (n=19), their mother (n=7), their father (n=2) and with siblings (n=2). Twenty-nine of the 36 respondents (81%) lived in the same residence as the person they were caring for. Table 4 provides the employment status of the 36 respondents, beyond their employment as an informal carer. Not answered 3% (n = 1) Figure 2 illustrates the length of time, in years, that the respondents have been informal carers, with the majority having spent more than 10 years in a caring role (61%; n=22).
Thirty-three respondents (92%) reported that they provided care on a full-time basis, with the remaining three (8%) providing care part-time. The care recipients*, in most cases, were children (n=16), followed by parent(s) (n=10), partner/spouse (n=9) and sibling(s) (n=3) (*n=38 as some respondents indicated they cared for more than one person).  and venue. The least important motivators for deciding to participate in a trial, from the carers' perspectives, related to study information issues; that is how they were informed of or invited to take part in the study, with all three associated statements averaging mean importance scores of 3.5 or less. Priority list of challenges Table 6 provides the list of challenges prioritised by the mean and SD scores attributed to each challenge statement. Personal risk, associated with difficulties in planning due to the caring role emerged as the most important challenge for carers when deciding on participating in a trial (mean 4.13, SD 1.25), followed by being unable to leave the care recipient on his/her own. Not knowing the person running the study was deemed to be the least important challenge for carers when deciding to take part in a trial (mean 2.74, SD 1.18).

Discussion
This SWAT has identified and highlighted important factors from the perspectives of carers that influence decision-making on trial participation. As the focus of healthcare internationally is to increase community-based care and avoid admission to secondary healthcare facilities for as long as possible [15], compounded by a rapidly increasing older person population [16], many individuals may find themselves in a caring role for which they are ill-prepared [17,18]. Evaluating interventions to support informal carers psychologically, socially, physically, or otherwise, is important. As a discrete group within the general population informal carers can be a hard to reach population, not least of all because of regional dispersity [19] and the cost, time and inconvenience that might be associated with taking part in a trial [19,20]. Furthermore, where trials do recruit, attrition rates in studies on carers can be high, for example, from 25% to > 40% across studies [21,22].
Understanding informal carers' views of research will assist trial developers, and other researchers, accommodate their unique needs. Carer identity, specifically, research that will help raise awareness about carers was the number one collectively identified motivator for trial participation. This supports the earlier quote describing informal carers as a 'hidden patient group' [2] and raises consideration of a possible sense of isolation or loneliness that carers may experience in their caregiving roles. Trial developers and researchers, in efforts to enhance participation in trials involving informal carers, need to consider the explicit demands that the caring role places on carers. These, in particular, identified in this study, were difficulties with planning ahead (number 1 priority challenge) and being unable to leave the care recipient alone, or for any length of time, all of which have implications for the design of any future, similar, host trial.

Implications of SWAT-55 for a future host trial
The non-feasibility of the host trial at recruitment level implies that any plan for a future, similar trial needs to take cognisant of carers' motivators and challenges, and would require a major rethink as to how the trial might be designed and implemented. For example, how informal carers come to know of a study (e.g. through a family member or friend, or through a leaflet), or their awareness of those conducting the study, were ranked overall, as being neutral or unimportant when deciding to take part in a trial. Thus advertising/invitational methods will not necessarily optimise recruitment to a future trial, rather other aspects of trial design, such as the value the trial has for carers' identity, and how the intervention is delivered will play a greater role. Challenges to conducting SWAT-55 A number of challenges in conducting the SWAT were encountered which may limit the results. These included limited recruitment to the host trial resulting in a reduced purposive sampling frame for phase 1 of the SWAT. Although rich data were provided by the two interviewees, a wider pool of participants might have better ensured, or at least increased our confidence in data saturation or sufficiency. This was overcome somewhat, however, by the extraction of data from related systematic reviews, and a triangulation of these data in developing the survey.
Despite extensive efforts, 36 participants only responded to the national survey, which is likely to represent a very small proportion of the population of informal carers across Ireland. Although we cannot be sure, the inability of Family Carers Ireland to distribute the survey via their email list, and the move towards distribution via Facebook, may have impacted on the survey response rate. We need to consider that had a larger number responded, providing greater caregiver representation, the ranked priority list of motivators and challenges might ultimately be different. The difficulties encountered with recruitment, however, could relate to how individuals identify with the concept of caring as evidence suggests that many individuals do not formally identify themselves as a carer or in a caregiving role [23]. For example, in a qualitative study of 40 relatives or friends, the researchers concluded that "…self-identification with the role and label of carer is nuanced, shifting and variable" [24] and refers to other studies that have shown variation in how relatives identify with the term carer [25,26]. In one study, for example, exploring recruitment with carers of people with multiple sclerosis, participants suggested using the term 'support' or 'assistance' in place of the term 'caregiver' and highlighted that many people would not consider themselves to be carers [23]. This is an important consideration for researchers when advertising trials to an already hard to reach population, and how carers, including their associated roles, are described may need to be considered within trial participant information.

Conclusion
Insight as to the motivators and challenges that influence informal caregivers' decisions for research participation, as offered by this SWAT, will assist trial developers and researchers to better tailor trial design and associated processes for informal caregiver populations, and adds to what is a limited evidence base. Consideration of these motivators and challenges to enhance participation has the potential to increase trial feasibility and success and reduce research waste. We recommend that trial developers consider these motivators and challenges when designing future trials involving informal caregivers.

Consent for publication
Applied as part of study consent processes