Background
Methods
Design
Definitions
Interventions
Expert panel
Phase I. Scoping and systematic reviews
Review 1
| To discover the experiences and views of participation in EoLC research of patients, caregivers, professionals and researchers and to identify best practices, we searched seven databases, hand searched three journals and the bibliographies of relevant papers. Inclusion criteria were: original research papers on involvement in EoLC research or its impact on participants. Critical interpretive synthesis (CIS) was used to integrate evidence regarding patient, caregiver, professional and researcher views on, and experiences with, participation in EoLC research, and identify best practices in research participation [17]. |
Review 2
| To appraise the state of the evidence of EoLC we conducted a systematic literature review of the evidence of effectiveness of palliative care teams in cancer. We searched six databases augmented by reference lists of earlier reviews. Inclusion criteria were: specialist (that is, with trained and dedicated professionals) palliative care in the home, hospital, or designated inpatient settings for patients with cancer and evaluation of the team. Outcomes were pain, symptoms, quality of life, use of hospital services and anxiety. Studies were excluded if they did not test specialist palliative care services. Meta-synthesis combined the studies according to type of team [16]. |
Review 3
| To appraise the methods used and challenges encountered in developing and evaluating palliative and EoLC services we developed the initial scoping into a systematic review specifically addressing this topic. We searched six databases and bibliographies of relevant papers. Inclusion criteria were: systematic reviews on the effectiveness of generalist and/or specialist palliative care (SPC) services for patients with advanced illness and/or their families. Narrative synthesis appraised the methods used against the MRC guidance steps, the main problems encountered and best practice solutions [18]. |
Phase II. Transparent Expert Consultation (TEC) and Stakeholder Workshops
1. | TEC planning by the MORECare project team, expert panel, and other experts identified in the literature to agree on the focus, scope the literature, and identify topic experts with appropriate multiagency and discipline mix (from health care and clinical research, not only palliative care) for the five workshops. We aimed always to include experts in the methods external to palliative and EoLC, researchers, clinicians, service developers and policy makers in palliative care, patients and consumers. |
2.. | Specific research questions for each TEC were agreed by the expert panel and included in the invitations sent two to three months in advance |
3. | TEC conduct - format: Morning – initial consideration of issues through two or three brief presentations by experts on the subject followed by equal time for discussion. Afternoon – three parallel working groups discussed and generated recommendations on ‘best practice’ to address the issues. Each individual completed a standard form asking them to list specific best practice recommendations to overcome the issues and rank these 1 to 5 (highest to lowest). Members of each group give feedback in turn on recommendations in priority order until the lists were exhausted or time exceeded. Groups discussed recommendations and where possible agreed on the ranking of the importance of the proposals. The afternoon was recorded to ensure that all aspects were captured and individual recommendation sheets and rankings were collated. |
4. | Editing of recommendations by the MORECare team to remove duplicates or merge similar proposals and remove any proposals which were strongly generic rather than EoLC specific. |
5. | Online consultation on recommendations– inviting all TEC attendees and the MORECare Project Advisory Group, that included the expert panel, to rank each proposal. Participants were asked to rate how much they agreed with each recommendation on a numerical scale from one (strongly disagree) to nine (strongly agree). They were able to make comments on each recommendation and general comments at the end of the consultation. |
6. | For each statement we report median agreement to determine the highest ranked items and interquartile (IQ) and total range to determine the degree of consensus. Narrative comments were collated. |
Phase III. Synthesis
Ethics
Results
TEC summit topic | Areas considered in TEC workshops | Number of attendees | Number of online responders | Backgrounds of individuals attending and responding |
---|---|---|---|---|
Ethics[20] | (1) Participation in research; (2) Research ethics committee approval; (3) Informed consent | 28 | 26 | ethicists, academics, researchers, members of research ethics committees, clinicians, service providers, commissioners, patients/carers |
Outcomes[22] | (1) Outcome measure properties; (2) Optimal time points; (3) Validity of proxy data | 31 | 28 | academics, researchers, clinicians, commissioners, experts in outcome measurement |
Mixed methods[23] | (1) Phase I and pre-clinical studies; (2) Phase II and III studies and trials; (3) Implementation studies | 33 | 26 | journal editors, academics, researchers, clinicians, experts in health services research/mixed methodology, patients/carers |
Statistics[21] | (1) Missing data; (2) Attrition; (3) Response shift | 20 | 19 | statisticians, researchers, academics, clinicians, patients/carers |
Health economics[24] | (1) Cost methods and relevance to EoLC; (2) Outcome assessment; (3) Equity issues. | 28 | 34 | health economists, service commissioners, researchers, academics, clinicians, patients/carers |
TOTAL | 140 | 133 |
Recommendations | |
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Introduction/background | 1. Present theoretical framework for the intervention and levels of need established |
2. Present objectives appropriate to the level of intervention development | |
Study design | 3. Indicate and justify stage in MRC guidance for development and evaluation of complex interventions, for example, feasibility, preliminary evaluation, efficacy/cost effectiveness and wider effectiveness |
4. Feasibility stages should test both feasibility of the intervention and of methods of evaluation, including outcome measurement | |
5. Justify methods, considering appropriate use of existing data sets and secondary analysis as these may produce rapid information | |
6. Justify methods of empirical studies considering mixed methods, observational studies and randomised trials | |
Study team | 7. Ensure involvement from: (i) consumers, patients and caregivers; (ii) relevant clinicians; (iii) relevant methodologists to develop study questions, questionnaires and procedures; and (iv) researchers familiar with the challenges in EoLC studies |
8. Ideally, involvement should be well established and continuing, beyond a specific study, with joint meetings or rotations between clinical and research staff | |
Ethics | 9. Note in ethics committee application MORECare recommendations that it is ethically desirable for patients and families in EoLC to be offered involvement in research and MORECare evidence of patient willingness to be approached |
10. Work within legal frameworks on mental capacity, consent and so on, to ensure that those who may benefit from interventions are offered an opportunity to participate if they wish | |
11. Collaborate with patients and caregivers in the design of the study, vocabulary used in explaining the study, consent procedures and any ethical aspects | |
12. Attend the ethics committee meeting with a caregiver or patient, as a means to help the committee better understand the patient perspective | |
13. Ensure proportionality in patient and caregiver information sheets, appropriate to the study design and level of risk, as excessive information in itself can be tiring/distressing for very ill individuals | |
Participants | 14. Adjust eligibility criteria to recruit those patients who may benefit most from intervention, ensuring equipoise |
Procedures | 15. Minimise burden for existing clinical staff for participation in the study |
16. Clearly distinguish between service received and research activity interviews in study arms when multiple interviews with patients are undertaken in trials, for example, using a graphical system [25] | |
Outcome measures | 17. Choose outcome measures that meet the following criteria: |
• established validity and reliability in relevant population | |
• responsive to change over time | |
• capture clinically important data | |
• easy to administer and interpret (for example, short and with low level of complexity) | |
• applicable across care settings to capture change in outcomes by location (for example, patients’ home, hospital, hospice) | |
• able to be integrated into clinical care | |
• minimise problems of response shift (see below) | |
18. Consider including patients’ experience of care, as this is central to many interventions | |
19. Select time points of outcome measurement to balance the value of early recording, to reduce attrition, but to allow enough time for the intervention to have had an effect | |
20. Consider the potential effect of response shift (that is, a change in a person’s internal conceptualisation or calibration of the aspects measured). Questionnaires that include anchor points or descriptions of each response category may be less problematic in this regard | |
Missing data and attrition considerations | 21. Estimate in advance levels of, and reasons for, attrition and missing data, integrating these into sample size estimates and planned collection of data from proxies |
22. Monitor during the study and report all levels of, and reasons for, attrition and other missing data | |
23. Assume missing quantitative data NOT to be at random unless proven otherwise | |
24. Test results from different methods of imputation – noting that ‘using only complete cases’ is a form of imputation | |
25. Use the MORECARE classification of attrition to describe causes of attrition: that is, | |
• ADD – attrition due to death; | |
• ADI - attrition due to illness; | |
• AaR - attrition at random. | |
26. Consider reasons for missing data which are not due to attrition, for example missed questionnaire, or missed data item in questionnaire. Consider these in analysis and the potential imputations | |
Mixed method studies | 27. Mixed methods can be appropriate in all phases of development and evaluation |
28. Ensure appropriate multi-disciplinary skills mix or training of team | |
29. Define the theoretical paradigm and method of integrating results and safeguards to ensure rigour at the outset | |
30. Plan investigation to avoid undue burden of qualitative and quantitative questionnaires – perhaps dividing data collection or selecting questions and/or sampling appropriately | |
31. Take into account any potential therapeutic effect of qualitative interviews where participants can express their feelings, if these are similar to components of the intervention | |
32. Ensure that those collecting data are appropriately trained in qualitative data collection | |
Implementation | 33. Consider implementation implications, including workforce and training needs, in all phases of the study |
Cost-effectiveness | 34. Integrate into preliminary evaluations and test feasibility of methods |
35. Collect data on use of services including health, voluntary, social and informal care, to take societal approach to care costs | |
36. Justify appropriate outcome measures to generate cost effectiveness |
Recommendations | |
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Ethics | 1. Create a Research Ethics Network for Palliative and End of Life care to further and disseminate best practice. |
2. Train those working on ethics and research governance committees in the specific issues and wishes of patients in palliative and EoL care and their families. | |
3. Seek to amend the law regarding consent so that advance consent for studies other than clinical trials of medicinal products is legally effective. This would permit research among people who might develop problems with mental capacity later. | |
Clinician/researcher collaboration | 4. Increase collaboration and understanding between clinicians and researchers in EoLC through rotations, joint departments and exchanges |
5. EoLC organisations to create a research-aware culture for practitioners by informing practitioners and patients on admission to a service that the organisation is actively involved in research | |
6. Develop specific training for practitioners in palliative and end of life care about research practice, its value and how to recruit | |
7. Introduce screening questions about patient/family willingness to be approached for research (as a general principle) in routine initial assessments on entry to palliative care services | |
Funders | 8. Develop collaboration to ensure that funding supports advancement in knowledge, where one study builds from the finding of another and there is progression to multicentre studies, full evaluations and cost effectiveness studies |
9. Assess study proposals against the MORECare statement | |
National bodies/strategy | 10. Develop repositories of routine data and from specific studies which can be used for secondary analysis to quickly answer current questions |
11. Develop collaboration to take forward the MORECare statement | |
Journal editors/referees | 12. In statistical assessment take account of the MORECare statement: |
• that attrition due to death and illness is to be expected and should be planned for when designing EoLC studies. It is not an indication of a poor study unless it is markedly different to that planned, but indicates that a relevant population of patients and families have been included, giving external validity | |
• that lack of attrition or missing data is not necessarily a positive finding; it could mean the population studied is less relevant to EoLC | |
13. Use MORECare statement to consider good research practice for conducting EoLC studies, alongside established checklists for reporting, for example, STROBE, CONSORT |