Protocol for Establishing a Stakeholder Group for Primary Care Research into Cancer Using a Modified 7P Framework and an e-Delphi Process

Introduction Currently, no group specifically supports and coordinates primary care focused cancer research in Ireland. The aim of this project is to establish an inclusive stakeholder group for primary care focused cancer research in Ireland, to coordinate research efforts and build capacity in researchers and institutions. Methods We will convene a stakeholder group, recruiting individuals with personal and professional experience of cancer care in a community setting. “Core stakeholders”—patients, clinicians, researchers, and policymakers—will attend regularly. Additional “specialist stakeholders”, such as representatives of secondary care, private healthcare, health insurance, industry, cancer charities, and health research funders, will participate on an ad hoc basis. An e-Delphi consensus process will be used to assess the stakeholders’ views on: (1) the relevance and importance of primary care focused cancer research; (2) the potential role and scope of the stakeholder group; (3) how best to engage with lived experience stakeholders and healthcare professionals affected by the research; (4) how to encourage the dissemination of results and the translation of findings into practice. Round 1 will be open-ended and will invite the independent suggestions of stakeholders; in Round 2 and 3, group members will vote on the inclusion of these suggestions in a position statement by the group, with consensus defined as ≥75% agreement. Discussion The formation of a broad stakeholder group to support primary care focused cancer research will ensure research is relevant, patient centered, and more readily translated into practice. It is also hoped that the group will support capacity building and strategic planning in this important research space.


Introduction
Currently, no group specifically supports and coordinates primary care focused cancer research in Ireland.The aim of this project is to establish an inclusive stakeholder group for primary care focused cancer research in Ireland, to coordinate research efforts and build capacity in researchers and institutions.

Methods
We will convene a stakeholder group, recruiting individuals with personal and professional experience of cancer care in a community setting."Core stakeholders"-patients, clinicians, researchers, and policymakers-will attend regularly.Additional "specialist stakeholders", such as representatives of secondary care, private healthcare, health insurance, industry, cancer charities, and health research funders, will participate on an ad hoc basis.An e-Delphi Any reports and responses or comments on the article can be found at the end of the article.

Background
Cancer imposes a significant healthcare burden in Ireland with an average of 35,825 invasive cancers diagnosed annually between 2018-2020 1 , costing €207 per capita or 5% of all healthcare expenditure costs annually-in addition to €116 per capita in indirect costs, due to lost earnings 2 .Internationally, cancer research is a major destination for research funding 3 , receiving approximately 25% of all disease-specific funding in the UK between 2004 and 2018 4 .In Ireland, the Health Research Board (HRB) reported that €35.8 million out of a total €131.8 million in 2021 was awarded to cancer related trials 5 .
In the United Kingdom, the National Cancer Research Institute was established to promote collaboration between cancer research funders to maximise the value and benefits of cancer research for patients and the public 6 .In Ireland, the National Cancer Control Programme (NCCP) has established a National Cancer Research Group which provides strategic leadership to the NCCP, Department of Health and other partners to implement the National Cancer Strategy 7 .Similarly, the All-Island Cancer Research Institute provides a cancer research framework from "discovery to clinical implementation" in and between the Republic of Ireland and Northern Ireland 8 .
The Irish Department of Health's National Cancer Strategy (2017-2026) recommends that "clinical cancer research, and the staff who deliver it, become a fully integrated component of cancer care delivery" 7 .Primary care professionals have a key role across the continuum of cancer care-in prevention, screening, early detection, treatment, survivorship and end of life care [9][10][11] .General Practitioners (GPs) are the initial point of contact for most patients presenting with symptoms of cancer 12 .The delivery of primary care across the cancer continuum is influenced by unique structural and systems factors 13,14 .This requires specific study designs, methods, and funding to address primary care policy, practice, service, workforce design, and intervention development and evaluation 15 .
The research skillset and infrastructure they demand, and the relative underfunding of primary care research (vis-à-vis other areas of health services research) [15][16][17][18][19] , foster a research ecosystem with unique challenges and priorities.
However, in contrast to basic science and secondary care research, currently no group specifically supports and coordinates primary care focused cancer research in Ireland.This represents an important gap in the Irish research ecosystem because: (1) primary care shares a large part of the cancer control burden in any health system; (2) research is needed to inform and improve care; and (3) research findings from secondary care setting are typically not generalisable to primary care.
We believe that, to address these primary care-specific challenges and priorities, a stakeholder group which specifically supports and coordinates primary care focused cancer research is needed.
Aims and objectives 1. Convening a stakeholder group This protocol describes the establishment of an inclusive stakeholder group to support primary care focused cancerrelated research in Ireland.This group will aim to address three broad needs: (1) to serve as a forum for key stakeholders, including the public, patients, clinicians, funders, researchers and policymakers to collaborate, enabling strategic planning and preventing duplication of research; (2) to build capacity in primary care cancer researchers, equipping them with knowledge and strengthening competence; (3) to improve research impact through strategic dissemination of research findings to clinicians and policymakers.

e-Delphi exercise
We will also conduct an e-Delphi exercise to gain consensus from stakeholders on: (1) the relevance and importance of primary care focused cancer research; (2) the potential role and scope of the stakeholder group; (3) how best to engage with lived experience stakeholders and healthcare professionals affected by the research; (4) how to encourage the dissemination of results and the translation of findings into practice.

Methods
We will convene a stakeholder group, guided by the established recommendations for multistakeholder engagement in health research 20 .Then we will conduct an e-Delphi exercise, in accordance with the Guidance on Conducting and REporting DElphi Studies (CREDES) 21 .Any deviations from the methodology described by this protocol will be reported, justified by a rationale and applied systematically.

Amendments from Version 1
The main change is to the title which adds details on how the stakeholder group was a established (i.e., via the 7P Framework and an e-Delphi process) and therefore highlights why the paper may be of interest.
Other peer review comments (on the reason for publishing this protocol, the nature of the proposed group compared with a project steering committee, and the lack of consideration of EDI concerns in the original 2012 Concannon framework) were addressed by clarification, rather than edits to the manuscript.
Ming Chuen Chong has been added as a co-author because they contributed to the revised version of the article, responding to the suggestions and comments of the peer reviewers.

Any further responses from the reviewers can be found at the end of the article
To achieve the goal of an inclusive membership, Concannon's 7P taxonomy for stakeholder engagement will be utilised to ensure broad stakeholder representation and adapted to the Irish primary care context 20  To ensure that the perspectives of all key stakeholders of primary care cancer research are represented, we propose to recruit a minimum of two members from the four core stakeholder groups and recommend representation of all four at every meeting.However, the requirements for a quorum, as with all the operational methods, will be definitively determined by the group.
To facilitate the participation of additional stakeholders with more niche interests, we propose to distinguish between the core stakeholders described above, and "specialist stakeholders", who are expected to contribute only when the meeting agenda concerns their area of expertise or interest.This distinction will serve to boost inclusivity by ensuring efficient stakeholder participation, enabling some stakeholders to decline meetings which are not relevant to their role, scope of practice or specialist interests.
The three remaining categories of Concannon's 7P taxonomy: "Purchasers" (of health insurance), "Payers" (i.e., health insurance companies), and "Product" (device and drug manufacturers), are more applicable to the US healthcare setting, and the activities of the stakeholder group may only occasionally be of relevance to them.Hence, we plan to invite representatives from private healthcare, health insurance, and industry (pharmaceutical companies, device manufacturers, and medical software) to participate on an ad-hoc basis as specialist stakeholders.The various stakeholders of primary care focused cancer research and their role within the proposed group is illustrated in Figure 1.

Identification and recruitment
We will adopt different recruitment strategies across the different stakeholder groups.
Patients and carers will be recruited via our Public and patient involvement (PPI) partners, the HRB PPI Ignite and HRB Primary Care Clinical Trials Network.Cancer advocacy organisations (the Irish Cancer Society, Marie Keating Foundation, and Breakthrough Cancer Research) will also be contacted to invite a representative to contribute to the stakeholder group.
Invites to clinicians and academics working in cancer care will be sent via established primary care and cancer research mailing lists and social media channels by the authors, who are involved with organisations such as the Association of University Department of General Practice in Ireland (AUDGPI), the HRB Primary Care Clinical Trials Network, the Irish College of General Practitioners (ICGP), and cancer research organisations.Policymakers and government representatives will be approached via the National Cancer Control Program and the National Cancer Registry.

Operational methods of the stakeholder group
The operational methods of the group will not be fixed in advance, but rather will be decided upon by the participating stakeholders, informed by the results of the e-Delphi exercise described below.

e-Delphi exercise
We will employ three rounds of an e-Delphi study to gather stakeholders' beliefs, prior to the group's first meeting, on four areas of interest: (1) the relevance and importance of primary care focused cancer research, (2) the potential role and scope of the stakeholder group, (3) how best to engage with lived experience stakeholders and healthcare professionals affected by the research, and (4) how to encourage the dissemination of results and the translation of findings into practice.
A Delphi process was chosen because it is a well-established methodology designed to systematically collate expert consultation and build consensus among a diverse group 22,23 .
Additionally, the Delphi technique may be performed remotely, which will be convenient for the stakeholders.Stakeholder group members will be invited to participate in the e-Delphi exercise if they feel that they have an interest in primary care cancer research in general-i.e., all the core stakeholders and some of the specialist stakeholders.

Software
We will use Microsoft Forms to run the e-Delphi exercise.This will be piloted by the RCSI Department of General Practice staff and PPI contributors.

Stages of the e-Delphi exercise
Round 1 is open-ended; participants are free to make an unlimited number of suggestions in response to the following questions: In Round 2, the participants will be presented with the same 5 sections, but the task is voting (on the items generated from the previous round) rather than free-text answers.In sections 1 to 3, the participant will vote on inclusion in a finalised list to be published as a position statement.In sections 4 and 5, the participant will vote on whether a suggestion (a) should be included as a primary priority, (b) should be included as a secondary priority, or (c) should not be included at all in a finalised list to be published in the position statement.In Round 2, the participants are permitted to comment with their justification for any of the decisions/recommendations.
In Round 3, the participants will vote again on the same items as in Round 2, however this time they will be presented with both the results and justifications from Round 2.
After Round 3, if a consensus has not been achieved, the process will be terminated to prevent stakeholder fatigue, and a resolution will be sought at an in-person meeting of the stakeholder group.Both the failure to achieve consensus via the e-Delphi procedure, and the subsequent resolution achieved thereafter, will be reported.
The time between rounds is expected to be approximately one to two weeks, owing to the requirement to summarise the free-text answers from the previous round and to follow up with each participant who will be completing the exercise at a time of their choosing.

Definition of consensus
In sections 1 to 3 of the e-Delphi exercise, consensus is defined as ≥75% agreement that the item should be included or excluded 24 .In sections 4 to 5 of the e-Delphi exercise, consensus is defined as ≥75% agreement that the item should be included in a given category.Any subsequent deviations from this definition of consensus will be justified and reported.

Informational input and risk of bias
Since Round 1 of the e-Delphi study is open-ended, no information will be provided to the participants so as not to bias them towards a particular answer or answers.After Round 1, the investigators will summarise the results of the open-ended round by merging and paraphrasing "suggestions" believed to possess the same meaning.This summarised list will be presented to the participants for Round 2 for voting-i.e., while the participants will vote on paraphrased suggestions from Round 1, they will be presented with the suggestions in their original form.After Round 2, the investigators will merge and paraphrase "justifications" believed to possess the same meaning.Again, in Round 3, participants will be presented with the justifications in both their paraphrased and original form.

Stakeholder group membership
We will describe the characteristics of stakeholder group members: their areas of interest, expertise, professional role and scope of practice, and institutional affiliations where appropriate, as well as their demographic characteristics.We will report separately the makeup of the e-Delphi "expert panel", per the requirements of the CREDES reporting guideline.

e-Delphi results
We will outline the results of the e-Delphi exercise under the following headings: (1) the relevance and importance of primary care focused cancer research, (2) the potential role and scope of the stakeholder group, (3) how best to engage with lived experience stakeholders and healthcare professionals affected by the research, and (4) how best to disseminate results and advocate for translation of findings into practice.
We will report results for each round separately, to make transparent the evolving of consensus over successive rounds.This will include figures showing the average group response, changes between rounds, as well as any modifications of the survey instrument such as deletion, addition, or modification of survey items based on previous rounds.

Other results
At the first in-person meeting of the group, members will agree upon the operational methods of the group, a finalised list of objectives, and an action plan for the coming year, informed by the results of the e-Delphi exercise.This will form the basis of a "Terms of Reference" document for the group and will be reported as an appendix.
The stakeholder group will also develop a position statement on primary care focused cancer research in Ireland, informed by the results of the e-Delphi exercise -this will be included for publication (as an appendix).The position statement will comment on (1) the relevance and importance of primary care focused cancer research; (2) the potential role and scope of the stakeholder group; (3) how best to engage with lived experience stakeholders (the public, patients, and carers) and healthcare professionals affected by the research; (4) how best to disseminate results and advocate for translation of findings into practice.
We will include a reflection of potential limitations of the e-Delphi exercise and their impact on the resulting Terms of Reference and position statement of the stakeholder group 21 .
Where it is achieved, we will report endorsement of the position statement by relevant professionals and professional bodies to boost dissemination 21 .

Dissemination
In addition to a peer-reviewed open access publication and academic conference presentations, we will also disseminate the findings to a broad range of professional networks with relevance to primary care cancer research.Moreover, we intend to report on the progress of the project at a public event focusing on cancer control in primary care, planned for late 2023.
Once convened, the stakeholder will identify appropriate audiences to engage when sharing the outcomes, such as researchers, policymakers, funders, and patient and clinical communities.

Conclusion
The formation of a broad stakeholder group in primary care focused cancer research, representing both professional and lived experience stakeholders, will ensure research is relevant, patient centred, and more readily translated into practice 25 .

Study status
At the time of submission, the stakeholder group had not yet been convened.It was planned that this would occur at during the period July to September 2023.

Ethical considerations
The Research Ethics Committee at RCSI granted approval for this project (REC202301024) Overall, this is a good protocol which should help guide other groups, even beyond cancer care or primary care, to convene effective stakeholder groups.
With this said, there are still a few points for improvement in the current manuscript.
1a) The title implies that the authors are presenting a novel protocol for establishing a stakeholder group.In fact, they are simply presenting Concannon et al's 2012 protocol.
1b) The authors are appropriately using a Delphi process to assess and consolidate stakeholder views.However, there is nothing particularly novel about the approach or how they are using it.
1c) Therefore, although this will be an interesting study, and I look forward to reading the outcomes, I'm not sure what we are gaining from a published protocol in advance of actual findings.However, SEE BELOW for some suggestions on how this could still be a valuable protocol.
1d) Even if it does proceed, a better title would focus on 'Using an e-Delphi process to...' Because that is what this is mostly about.
2) Issues with the engagement approach 2a) Although Concannon et al. is a good start and does indeed set the framework for a strong engagement process, it is getting a bit dated in terms of PPI scholarship (2012) and omits to address several issues that have emerged since.
2a.i) Concannon does not directly address issues of representation, including EDI issues and reaching underrepresented or underserved segments of the population.Without this, steering groups tend to contain the 'usual suspects'.This would be an EXCELLENT opportunity for this protocol to ADD to Concannon and suggest something novel.
2a.ii) Concannon does not directly address the ethical considerations surrounding steering group recruitment and selection.These are numerous but big ones are duality of relationships for PPI contributors (e.g., serving on the same panel as their health care provider and the power and vulnerabilities this can create); and researchers' duty of care toward vulnerable PPI contributors.Again, an excellent opportunity for the authors to ADD to Concannon.

Is the rationale for, and objectives of, the study clearly described? Yes
Is the study design appropriate for the research question?Partly

Are sufficient details of the methods provided to allow replication by others? Yes
Are the datasets clearly presented in a useable and accessible format?

Not applicable
Competing Interests: No competing interests were disclosed.
Reviewer Expertise: PPI, participatory health research, primary care research, systematic review, community and population health.
I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above.
Author Response 27 Nov 2023

Benjamin Jacob
Comment/Suggestion #1: "The title implies that the authors are presenting a novel protocol for establishing a stakeholder group.In fact, they are simply presenting Concannon et al's 2012 protocol...The authors are appropriately using a Delphi process to assess and consolidate stakeholder views.However, there is nothing particularly novel about the approach or how they are using it....Therefore, although this will be an interesting study, and I look forward to reading the outcomes, I'm not sure what we are gaining from a published protocol in advance of actual findings.However, SEE BELOW for some suggestions on how this could still be a valuable protocol...Even if it does proceed, a better title would focus on 'Using an e-Delphi process to...' Because that is what this is mostly about."

Response #1:
As you've already said, our approach to establishing the stakeholder group involves two aspects: (1) convening a stakeholder group, using a modified Concannon 7P Framework, and (2) exploring stakeholder values in order to guide the process, using an e-Delphi process.Therefore, it feels like the title should not the e-Delphi process without mentioning the 7P Framework also.
We could thus suggest a new title as follows: "Protocol for Establishing a Stakeholder Group for Primary Care Research into Cancer Using a Modified 7P Framework and an e-Delphi Process".It might be considered a little longwinded?But if you find a shorter title-e.g., Protocol for Establishing SPARC-a Stakeholder group for Primary cAre Research into Cancer-to be misleading, we would be happy to accept the former.
Our rationale for publishing the protocol is not because of the novelty of the methods (as you rightly point out, we are following a well-established approach).Rather our primary motivation was academic transparency (and the consequent reduction of publication bias/selective reporting), as well as the reduction of research duplication.You are right to say that we have not proposed a clear EDI strategy in advance of establishing the group, but this is because we want the stakeholders themselves to decide upon the group's responsibilities in accordance with their values (rather than having this enforced a priori by the research team).Hence, we expect the stakeholders to make suggestions along these lines via the e-Delphi exercise, due to the diversity of stakeholders and PPI input in the group.A natural place for these suggestions to be made is Q2b (Role of the group) and Q3 (Engagement with lived experience stakeholders).Thus, we can be reasonably confident that the outcome of the e-Delphi consensus process will ensure that EDI and PPI concerns are addressed, and an action plan published in the planned position statement.

Comment/Suggestion
This group will support and promote high-quality research, which involves the promotion of meaningful PPI input and the inclusion of under-represented and underserved groups in the population.However, this stakeholder group will not act as a steering group for any specific research projects.Futhermore, the under-represented and underserved groups in the population differ depending on the cancer type and clinical context, thus the stakeholder group will best effect positive change through its general influence of research steering committees and will seek a diversity of lay and professional voices in its composition.
Comment/Suggestion #3: "Concannon does not directly address the ethical considerations surrounding steering group recruitment and selection.These are numerous but big ones are duality of relationships for PPI contributors (e.g., serving on the same panel as their health care provider and the power and vulnerabilities this can create); and researchers' duty of care toward vulnerable PPI contributors.Again, an excellent opportunity for the authors to ADD to Concannon."

Response #3:
These ethical concerns are less applicable to this kind of stakeholder group, as it is concerned with shaping overarching research priorities, capacity building and impact strategies rather than acting as a typical project steering committee (handling the nitty gritty details of study design and project management etc.).
For clarity, none of the PPI contributors in this stakeholder group will serve on the same panel as their health care provider (albeit this can occur in project steering committees).
Secondly the researchers' duty of care towards vulnerable PPI contributors doesn't clearly apply because the "project" they are working on is not a "research study" being "led" by a "researcher", and to which vulnerable patients, for example, are contributing in a PPI capacity.Rather, in the case of this stakeholder group, PPI contributors and researchers are just two of wider range of possible collaborators, working together to support primary care cancer research at a strategy level.
Competing Interests: None declared.

Desmond Leddin
Department of Medicine, Dalhousie University, Halifax, Nova Scotia, Canada Thank you for the opportunity to review the protocol, The authors are very well qualified to carry out the work and are in an academic environment which will be supportive.Is the rationale for, and objectives of, the study clearly described?
The rationale is clearly described.The administrative relationship of the proposed new group to the NCRG or All Island CRI is not clear.The authors mention that there are basic science and secondary care research groups.It would be of interest to know how they relate to the NCRG.

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Is the study design appropriate for the research question?
It is.The authors propose to use a modified 7P structure, which is certainly permissible within the method described by Concannon.

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Are sufficient details of the methods provided to allow replication by others?Yes.More detail on how the suggestions from Round 1 will be extracted for presentation in subsequent rounds would be helpful.

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The aims seem to change a little as the process moves to methods.Page 3.Under Aims 2. Edelphi four questions are listed.Relevance and importance, role and scope, engagement, dissemination, advocacy and translation into policy and practice.Arguably this is six questions.This is somewhat different from the previously listed aims.Benefits for patients may bring advocacy and translation back in and perhaps that is meant by impact?Impact was listed on page 3 under stakeholder objective but not under the eDelphi exercise.○ This is somewhat different from the previously listed aims.Benefits for patients may bring advocacy and translation back in and perhaps that is meant by impact?Impact was listed on page 3 under stakeholder objective but not under the eDelphi exercise." ○ RESPONSE #1: We have made slight alterations to the three sections for greater correspondence and clarity: Page 3 & 5 ("2.e-Delphi exercise") both now read:

Figure 1 .
Figure 1.The stakeholders of primary care cancer research, and their role within the proposed group.

○ Page 5
Edelphi exercise.Same as above ○ Page 5. Stages of the e Delphi exercise lists relevance, scope, role, engagement, and impact.○ ○ ○

○○Page 5 Page 5 .
Overall, a well written, and well-constructed proposal.My comments are minor.Thank you for allowing me to read it.Is the rationale for, and objectives of, the study clearly described?YesIs the study design appropriate for the research question?YesAre sufficient details of the methods provided to allow replication by others?PartlyAre the datasets clearly presented in a useable and accessible format?Not applicableCompeting Interests: No competing interests were disclosed.Reviewer Expertise: Colon cancer screening, environment and healthI confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.seem to change a little as the processmoves to methods.Page 3.Under Aims 2. Edelphi four questions are listed.Relevance and importance, role and scope, engagement, dissemination, advocacy and translation into policy and practice.Arguably this is six questions.Edelphi exercise.Same as above ○ Stages of the e Delphi exercise lists relevance, scope, role, engagement, and impact.
#2: "Although Concannon et al. is a good start and does indeed set the framework for a strong engagement process, it is getting a bit dated in terms of PPI scholarship (2012) and omits to address several issues that have emerged since... Concannon does not directly address issues of representation, including EDI issues and reaching underrepresented or underserved segments of the population.Without this, steering groups tend to contain the 'usual suspects'.This would be an EXCELLENT opportunity for this protocol to ADD to Concannon and suggest something novel."