ICANEQUAL multi-stakeholder partnership: Reducing inequalities in liver cancer (HCC) diagnosis, treatment and care across the UK

Patient and Public Involvement

The British Liver Trust and Egality Health were co-applicant on the ICANEQUAL grant and contributed as a part of research team in providing patient perspectives and prioritizing research questions to address the need of patients and underserved patient population The ICANEQUAL Community of Expertise included interested individuals and patients and carer with lived experience and those with a carer role via the British Liver Trust and Egality Health and their work with Can-Survive UK, BME Cancer Communities, and Wai Yin Society. They were involved from the beginning of the project and contributed to the development of recruitment strategies and discussions on key aspects of research questions, project design, and delivery, helped raise awareness about this research, and led innovative recruitment strategies using social media and charity webpages. This was done to ensure that the workshop design and materials were culturally appropriate and sensitive. They were also involved in agreeing plans for dissemination of the study results to participants and linked communities by reviewing and advising on suitability of the research findings summary, visual materials, ICANEQUAL website and dissemination plans. Patients and the public members who have contributed substantially and meet ICJME criteria are listed as co-authors.

ICANEQUAL partnership development

Two elements of the partnership activity are reported in this paper: building a Community of Expertise (Domain 1) and conducting a series of three workshops (Domains 2 and 3). The primary aim of the workshops was to develop a more nuanced understanding of the landscape of HCC diagnosis and treatment for underserved populations. There were three key objectives: 1) to map the current landscape of HCC care in the UK, 2) to understand and articulate the key issues and gaps for underserved populations, and 3) to co-design solutions to address the identified issues. An independent committee of representatives from key stakeholder groups provided an overview of project duration (Figure 1).

Figure 1. ICANEQUAL framework.

Participants and recruitment

The preliminary partner network and the growing ICANEQUAL Community of Expertise supported recruitment for the workshops. The aim was to recruit up to 10 participants per stakeholder group to balance information power against achieving a sense of group and purpose within each online session, and to recruit individuals to participate who could act as spokespersons for their area of expertise. This included health and social care professionals from primary, secondary, and tertiary care, including transplant co-ordinators, individuals with lived experience, patient and public (PPI), community organizations, and researchers. Existing community and professional networks, the NIHR ‘People in Research’ website, and social media were also used to recruit stakeholders from across the different health boards, including the social and geographical differences across the UK.

A letter of invitation to join the Community of Expertise and participate in the workshops was developed in collaboration with PPI group members: the British Liver Trust and Egality Health, a community-focused small enterprise. Terms of reference and rules for engagement were also developed and shared with the potential participants. Participants were asked to join all three workshops; however, if a participant was unable to attend more than one workshop, further representation was sought from the stakeholder group to ensure balance in each workshop.

Invitations to join the partnership and participate in the workshops were sent (emailed) to 57 stakeholders and additional stakeholders were approached using the snowballing method. Invitations were targeted to health care professionals (n=22), including GPs involved in early diagnosis in primary care, nurses, consultant hepatologists, oncologists, and transplant surgeons; patients and public members (n = 18), including outreach project managers, community groups, representatives from diverse ethnic communities and patient advocacy groups, and those living with and beyond cancer; and researchers (n=17), including experts in cancer epidemiology, health economics, research/trial methodology, diversity and inclusion in trials and implementation science, and global health/health inequalities.

Workshop delivery

All the three workshops were conducted online (ZOOM, licenced by University of Aberdeen) and facilitated by SJM, ST and MB. This reduced the required travel time commitment for each workshop and supported attendance for all; for example, individuals managing complex health needs or HCPs balancing clinical and research commitments. A delegate photo directory was produced and shared with all participants ahead of the first workshop to facilitate their engagement and increase their sense of community. The workshops were recorded and transcribed using a third-party university-approved transcription service. Informed verbal consent to record the workshop discussion and transcribe the recording was obtained from all participants before the workshop. NVivo (version 12 for Windows, QSR International, NVivo Leading Qualitative Data Analysis Software (QDAS) by Lumivero) subscribed by University of Aberdeen was used to organize the data and support analysis.

The ICANEQUAL project management team facilitated and moderated the workshops. Each workshop was three hours long and structured using a model previously validated by a research team²⁵. Each workshop was divided into three parts and followed the same structure: a plenary session during which the purpose of the workshop, rules of engagement, and instructions for the small discussion groups, small group discussions, and a final plenary session in which key points from each group were presented back and discussed by the entire group. The discussion groups for the first two workshops were assigned to include participants from the same area of expertise (HCP, PPI or Researcher). During the third workshop, groups were assigned to include participants from various areas of expertise. Structured discussions during the workshops were provided by the NIHR INCLUDE Ethnicity Framework²³.

Analysis

The data from the workshops were coded both inductively and deductively using the Theoretical Framework of Acceptability (TFA)²⁶. Data from the first two workshops were analyzed inductively. Data from the third workshop were analyzed inductively and deductively, the prioritization process was inductively coded, and the proposed research projects were deductively coded using TFA. Any relevant statements that did not correspond to the TFA domain were coded inductively under the code proposal development.

A preliminary inductive analysis following each workshop allowed the next workshop to begin with a summary of the previous workshop. A summary document was also provided to all participants before the second and third workshops. During the plenary session, the group was asked to make any corrections, clarify any issues, and add any missing issues to the presented summary. These were incorporated in the final version of the summary document.

The selected transcripts and sets of themes were then read by multiple researchers. The research team discussed any inconsistencies in the results. Multiple coding using ST and SJM tested the reliability of the designated codes.

Building the Community of Expertise

The ICANEQUAL Community of Expertise is central to equitable and sustainable partnerships. Collective work and dissemination to stakeholders and the public are supported by the website (see below Figure 2- Screenshots of the ICANEQUAL website landing page). The medium-to long-term objective of the ICANEQUAL partnership is to develop an ICANEQUAL Training Academy for early career researchers.

Figure 2. Screenshots of the ICANEQUAL Website landing page.

Multi-stakeholder workshops for building a shared understanding of the problems and the solutions

Participants. Participants in the ICANEQUAL project represented urban, rural, and coastal communities in Scotland and England. A total of 38 stakeholders with various levels of expertise participated in either one or multiple workshops (see Table 1). Fifteen stakeholders participated in all the three workshops.

Workshop 1: Understanding the nature of the problem (Barriers & facilitators)

Workshop 1 focused on examining existing clinical pathways and services for people with HCC across the UK. Participants shared examples of good practice across the UK (including other contexts), examples of problem areas around early diagnosis and treatment allocation, and knowledge of key research and service redesign projects within the UK.

It has been agreed that current models of practice for liver cancer vary across the UK. Challenges are amplified for under-served populations due to systemic, geographical, social, and cultural barriers; for example, rural areas lack major treatment centers, expert health care services, patient limitations with the English language, and stigma attached to liver disease. Additionally, poor engagement with underserved communities and a lack of understanding of their needs, historically exploitative medical practices, and complex healthcare systems discourage communities from seeking care and support. All underserved populations were felt to need better consideration in HCC diagnosis and care; no one group was prioritized. This sub-theme underpins all qualitative data analyses.

The analysis of Workshop 1 highlighted areas of ongoing practice and challenges to patients’ entry into the HCC care pathway, provision of care, and held evidence. These were categorized into three themes: 1) early diagnosis, effective treatment, and supportive care; 2) research and evidence; and 3) development of an ideal pathway.

Theme 1: Early diagnosis, effective treatment, and supportive care. Both PPI and HCP participants discussed challenges in timely diagnosis, treatment and effective care across the UK e.g. lack of awareness, screening opportunities, and logistical challenges as “treatment tend to be centralised around large hospitals within a region and that’s hard for people to travel… going in for surveillance or treatment then it’s much more difficult for those individuals.” HCP, Male, England. In existing surveillance programs “there are disparities across different regions, so some people get an ultrasound, some people get a blood test, some people get MRIs, it’s all very varied so the pathway is quite different depending on where you are or what hospital you’re under.” Patient Advocate, Female, England.

Challenges regarding communication within healthcare systems in the transfer of care and with the patient were also identified as key issues. HCPs highlighted that it was important to build systems that allow quick turnaround on diagnostics and investigations for multidisciplinary teams (MDTs) across the UK and that as “time to treatment is significantly shorter” there is a need to “spend more time on patients” early diagnosis and treatment rather than “discussing the patients who don’t need to come to the MDT”. The need for national HCC screening programs has also been highlighted.

Theme 2: Research and evidence. Participants emphasized that the current research and evidence on detection in HCC lacks adequate consideration of the needs and priorities of groups that are under-served in health research. Large cancer registry data that could provide insight into population-level health are missing from the health equity lens.

It was felt that there is an issue with the lack of data quality “specially about which subgroups of patients are needing additional resources to improve equity for access”. There is a need to generate more appropriate data that allows for a better understanding of patient-level (e.g., ethnicity, deprivation, etc.; PROMs and PREMs) and systems-level characteristics (e.g., number of points of contact; time between points of contact). Participants noted that existing evidence suggests that blood tests at 6 months intervals can have a significant impact on monitoring tumor occurrence and treatment response.

Theme 3: Development of an ideal pathway. Due to the identified heterogeneity of current practice, the development of an ideal pathway for research, practice, and guidelines was suggested and supported by the entire group. It was agreed that this does not currently exist in the UK Kingdom. However, it was flagged that this is not a simple task because of the complexities of NHS structures and commissioning arrangements and the lack of accepted clinical practice guidelines in the area.

The adaptability of an ideal pathway was considered very important as challenges vary across time, levels of care (primary, secondary, and tertiary), and geography (e.g., urban vs. remote, rural, and coastal; England vs. Scotland). The patient’s perspective is crucial in unpacking these variations in practice, experience, and outcomes.

It was felt that creating well-resourced and sustainable roles (such as patient/cancer pathway navigators) within health systems, especially in rural areas, could be important. The need for systems that allow faster reporting on diagnostics and investigations for multidisciplinary teams (MDTs) across the UK has also been raised, such as the hub-spoke models used in the South of England. Exploring other models within existing services in different parts of the country could be beneficial, such as the European Centre for Disease Prevention and Control (ECDCs, England), Rapid Cancer Diagnostic Service pilots (Scotland), MDT pre-meet, cancer pathway navigators and MDT co-ordinators, and Clinical Nurse Specialists (CNS). At the same time, the group felt strongly that it was important not to reinvent the wheel in a system that was stretched and resource-limited (See Table 2).

Workshop 2: Three key solutions

During workshop 2, the group was asked to critically examine the gaps that were identified in the previous workshop and map the barriers and facilitators to equitable early diagnosis and treatment allocation for underserved populations. The focus of workshop 2 was to understand the specific needs of different underserved groups in HCC and prioritize areas for moving forward. The research priorities to address the identified gaps were informed by the NIHR INCLUDE Ethnicity Framework²³: 1) the recipient of the proposed solution, 2) the feasibility and acceptability of the proposed solutions, 3) the engagement of all the relevant groups, and 4) the context for the delivery of the solution. This was achieved through structured discussions and consensus building at the end of the workshop.

Theme 1: Raising knowledge & awareness of HCC across the UK. Lack of knowledge, awareness and access to available treatment were identified as major barriers to early diagnosis and provision of care for under-served populations across the UK especially “that’s disproportionately affecting certain regions with high levels of deprivation, certain groups of patients”. HCP, Male, England. Participants agreed that “if patients have better knowledge and understanding of their risk and what they can do to change their course, then you know we’re potentially empowering them at that early stage which is really important when we can do something about it.” HCP, Male, England. This was felt to be important for several different groups, extending beyond the considerations of ethnicity and social deprivation.

Therefore, in the development of any screening program, defining underserved populations as well as better articulation of strategies to reach, recruit, stratify data, and implement the program is important. This would need to be deliverable, practicable, and sustainable, and not place extra stress on already overburdened parts of the workforce.

The lack of consideration of underserved populations was felt to be evident within the identified gaps, for example, the lack of help-seeking behavior from patients, especially due to stigma attached to liver disease, the lack of a national screening program to support presentation for detection/diagnosis, and the lack of knowledge and awareness across of HCC across the UK. However, it was agreed that tailored community-based interventions, strong partnerships with third-sector organizations, and learning from existing initiatives around the inclusion of under-served populations could overcome these barriers.

Theme 2: Providing peer support & mentoring. The participants highlighted the importance of mentoring and peer support via community group/patient groups. Those running these programs could decipher the complexity and communication flow of clinical settings, and that this could be an enabler to enhance awareness, access, and engagement with surveillance or treatment from different communities.

The group raised several challenges in providing structured peer support and mentoring. These barriers were noted to be particularly worse for underserved populations or hidden patient communities who face geographical, social, and cultural barriers. Challenges included rural areas that lack major treatment centers, lack of accessible expert health care services, patient limitations with the English language, access to transportation for engagement with healthcare services, digital exclusion/disadvantage, and lack of help-seeking behavior, especially due to the stigma attached to liver disease (e.g., alcohol abuse).

It was highlighted that already existing community-based groups, patient organizations, and third-sector partnerships could provide support through mentoring and peer support at different stages within the care pathway. “The peer support has come through loud and clear quite a few times and the importance of receiving it across the whole pathway”.

Theme 3: Improving information on transition between key points in the care pathway. It was highlighted that there is a communication gap around “access to the pathway and the GPs bottleneck”, as well as the transfer of care from primary care to regional hospitals and transplant centers. Owing to the limited time period for diagnosis and the provision of curative treatment for HCC, delays in the transfer of care have a detrimental impact on patient outcomes.

The lack of an agreed pathway that includes a high or high suspicion in addition to urgent referrals, the lack of robust data for designing an equitable care pathway, and treatment delays caused by diagnostic loops.

There was unanimous agreement that maximizing existing resources such as patient navigators, regional and national pathway co-ordinators, and collaboration between MDTs and Cancer Alliances could be effective in improving the transition between key points in the care pathway. Understanding the roles of patient navigators, MDT co-ordinators, and CNS in place across the UK would be important in designing an effective model/protocol. This model is flexible and adaptable for underserved populations and allows for group-level comparisons and understanding across the cancer pathway. An emphasis on the importance of context and careful consideration of the specific needs of each underserved population was common in all discussions.

Synthesis of the inductive codes from Workshop 1 and Workshop 2. The inductive coding of all discussions during the plenary and individual stakeholder group discussions from Workshops 1 and 2 generated 15 preliminary codes. The frequency of discussions and the emphasis placed on them varied within or across stakeholder group discussions. Multiple researchers analyzed this dataset to synthesize the key themes and priority areas from preliminary workshops (see Table 3 and Table 4). This was categorized into five themes under three priority areas for equitable care for the under-served population (see Table 5). This includes the strengths and barriers of each area.

Workshop 3: Multi-stakeholder groups consensus on priority project areas

Workshop 3 focused on reaching a consensus on the key priorities for improving care and treatment allocation for people with HCC to address the gaps in under-served populations. At the start of the workshop, participants were asked to consider the five priority areas and prioritize the three project areas as the focus of the workshop discussion groups (Table 5). The agreed areas were projects around 1) better data solutions, 2) better screening and surveillance solutions and 3) ‘navigation’ (person-centered and / or health system focused) solutions. It was felt by the whole group that this was not being properly achieved for any under-served population (e.g., rural population, people from lower socio-economic status, migrant population, or people with language barriers); therefore, the analysis presents key principles at the center of change for all under-served populations discussed.

Each discussion group was assigned a priority area and asked to develop an outline for a project to address the needs of the underserved population. This had to include research questions, key outcomes, outline methodology, and potential evaluation strategy. The analysis of the transcripts from Workshop 3 was guided by the prospective application of the Theoretical Framework of Acceptability (using a coding frame; see Table 6).

This deductive analysis had seven a priori themes: affective attitude, self-efficacy, perceived effectiveness, intervention coherence, burden, ethicality, and opportunity costs (See Table 4). There were only a couple of statements coded to ‘opportunity cost,’ so this is not presented in the results section below. Inductive analysis provided the context of the proposed priority projects and generated themes around the need for the project (affective attitude), the strategy and workforce needed to execute (burden), potential feasibility (self-efficacy), and effectiveness of the project (perceived effectiveness), and fit with the stakeholders’ belief systems (ethicality).

A better understanding of equity and effectiveness of care within the HCC pathway (project 1) had three key project outcomes: a) the identification of data gaps, b) the quantification of the unmet needs of underserved populations, and c) the improvement of evidence-based practice and policy. Stakeholders agreed that the project would be acceptable and feasible with the involvement of data scientists, quantitative and qualitative researchers, and experts working with NHS data. The role of voluntary and community organizations was deemed important in identifying and reaching patients with authentic and trustworthy voices that are usually missed.

Better screening and surveillance (including knowledge of individuals and training of HCPs) (Project 2) focused on facilitating better entry into the HCC pathway. There were three key project outcomes. These were: a) knowledge transfer from other contexts and cancer types, b) presentation to screening, delivery of screening, and referral to treatment, and c) training of the different stakeholder groups. Multi-sector collaborations, including researchers, industry, community organizations, and voluntary organizations, were also needed to address the identified challenges in the mode of engagement and establish credibility with different under-served populations.

Experience and effectiveness of care within the HCC pathway (Project 3) focused on an evidence-based, mentoring program would be effective and empowered for patients. It was agreed that patients’ voices were a key element in both the design and delivery of care. The stakeholders agreed that there were three key project outcomes. These were: a) an audit of existing mentorship programs from other contexts and cancer types to tailor to HCC; b) clear articulation of a program outline that could be tailored to rurality, ethnicity (cultural/race), language, digital barriers, deprivation/poverty, gender, age, and living arrangements; and c) the collaborative involvement of the different stakeholder groups. The role of current mentors and mentees/peers, community organizations, and charities around liver conditions was deemed crucial. Also important was the improved referral to mentoring by HCPs.

Understanding equity and effectiveness of care within the HCC pathway: better data solutions. Stakeholders in this group agreed that a mixed-method study accessing data from a few diverse centers (location and size) across the country would be feasible and suitable for quantifying the problem and identifying gaps in data availability. It was agreed that data from patients could provide an understanding of differences in the diagnostic/treatment pathway, whether there is a delay in staging, who the patients are missing, and how they are affected by health inequalities (age, sex, ethnicity, rurality, or deprivation index). Data gathered from services and healthcare professionals could contribute to the understanding of why there is delay in treatment, or if access to certain treatments is determined by availability and workforce issues and differentially impacts those attending. Community services and organizations could provide an understanding of who is struggling to enter the system for diagnosis and/or treatment (although they may not have quantitative data).

Perceived effectiveness: Stakeholders agreed that the proposed priority project could be effective in identifying what data are missing, how to benefit from working with existing initiatives in terms of data, and how to identify specific groups for future projects. It was also believed that comparisons of group-level primary data with current guidelines would help inform the extent to which current practices in the UK are evidence-based and equitable.

Stakeholders also agreed that, in addition to accurate data to facilitate better understanding of differences in presentation for diagnosis and receipt of treatment, the project could also help improve current practice and provision of care, such as implementation science, audits, and feedback.

Burden/workforce: The patients in the discussion did not identify any burden of the project on underserved populations. However, the overall discussion highlighted the burden of the proposed plan on various professional groups, such as data scientists, quantitative and qualitative scientists, and data experts within the NHS. This is a major limitation of innovation in the NHS. Closer working with voluntary and community organizations (such as health or community voluntary services) could help identify and assist in engaging patient groups that are usually missed.

Ethicality: Stakeholders agreed that, given the lack of focus on equity in currently available data, significant consideration needs to be given to the disparity in access to diagnosis and care. This is increasingly being recognized by policymakers and funding bodies, but a better definition is required. Those working within the area and affected by HCC cannot confidently demonstrate the specific needs of different underserved groups. However, there was a clear agreement on the need for any proposed priority project to take a person-centered approach. This would include primary data collected in partnership with community groups, including under-served communities, to be inclusive.

Facilitating better entry (diagnosis) to the HCC pathway. Stakeholders in the group that focused on facilitating better entry into an HCC pathway and timely diagnosis discussed potential projects around better screening and surveillance solutions, knowledge and awareness of individuals, and education and training solutions for HCPs. They highlighted the importance of “…improving engagement with surveillance and screening, recognising that the pre cursor to this is raising knowledge and awareness but with the desire of translating this into behaviour because raising knowledge and awareness without that is necessary but not sufficient for anything to change.” Academic, Group two, Female, Scotland

Intervention coherence: The stakeholders felt that identifying the population at risk should include using FibroScan, adjunct blood tests, and a bespoke risk assessment questionnaire. The use of mobile units for formal screening and follow-up should be both feasible and acceptable. Community-based approaches would also have to be linked with traditional routes to presentation, such as symptomatic presentation to GP. An audit of existing project delivery programs to improve knowledge and awareness and understand the strengths and limitations of both community groups and healthcare professionals would also be useful.

Perceived effectiveness: A study guided by the audit of existing projects currently delivering knowledge and awareness programs in other contexts and for other cancer types should provide an understanding of the acceptability, feasibility, and cost-effectiveness of the community program. It would also allow for a better evaluation of the reach of and engagement with surveillance and screening by different underserved populations.

Workforce and partnership: Stakeholders highlighted that partnerships among researchers, industry, community organizations, and voluntary organizations would be instrumental in delivering and evaluating the project. All stakeholders were supportive of delivering community-based programs in partnership with multiple stakeholders. It was also recognized that groups that are already engaged in the community could play an important role in ensuring the authenticity and effectiveness of the message being conveyed.

Ethicality: Stakeholders highlighted different sub-populations who are underserved or non-integrated in different ways, for example, migrant populations who have substance abuse. However, there is a consensus that the complexities of defining and meeting the needs of underserved populations must be considered when planning programs to improve the uptake of surveillance and screening.

Improving the acceptability, experience, and effectiveness of care within the HCC pathway. The stakeholder group that discussed peer support and mentoring reached a consensus on the proposal for the ‘navigation’ (person-centered and/or health system focused) and partnership solutions. They agreed that developing inclusive peer mentorship guidelines (endorsed by clinicians, professional bodies, patients, community organizations, and policymakers) could help improve the acceptability, experience, and effectiveness of care within the HCC pathway.

The stakeholders highlighted the need to define mentoring (including key elements and outcomes) and identify gaps in the current mentorship programs. They agreed that building an evidence base around peer mentor expertise could be effective in supporting patients in the HCC care pathway. This could also include guidelines for an effective HCC peer mentoring program.

The co-design of an HCC mentorship program could include HCPs, current mentors and mentetes/peers, community organizations representing specific under-served groups (older adults, ethnic minority groups, non-English speaking), third-sector organizations (HCC-focused and other cancer-related organizations), and charities focused on other liver conditions, including Hepatitis B and C.

Perceived effectiveness: Stakeholders (including those who had experience of being a mentor or a mentee) believed that the development and provision of an equitable evidence-based mentorship program would be effective. They emphasized that, in addition to supporting patients during care, their involvement in providing mentorship could also contribute to patient empowerment within their healthcare interactions.

Burden: Stakeholders highlighted that given the diversity of potential patient populations (both mentors and mentees), the provision of mentorship needs to be considered according to the context and demography. Issues around rurality, ethnicity (cultural/race), language, personal or structural digital barriers, deprivation/poverty, gender, age, and living arrangements were highlighted as potential barriers to providing an equitable mentorship program.

Visual representation

All of the work achieved within this partnership project is summarised below (Figure 3: Visual representation of the ICANEQUAL Partnership project).

Figure 3. Visual representation of the ICANEQUAL Partnership project.

Strengths

This project had a multi-stakeholder representation from both rural and urban locations in the UK. The workshop design and theoretical framework provided a robust method for both project delivery and analysis. A rigorous preliminary analysis between workshops to inform the next stage ensures the effectiveness of each workshop. The virtual platform ensured the effective use of time resources and facilitated participation from all stakeholders, especially those who might be unable to attend in-person sessions because of physical or logistical issues.

Limitations

The main limitation of this study is related to engagement in primary care. All workshops had good representation from healthcare professionals across the UK and represented different clinical roles. However, despite extensive efforts to recruit primary care professionals, this proved a difficult group to obtain engagement from and reflects the current workforce pressures that need to be considered in any system redesign. However, the voice of primary care was present within this project from the team of collaborators and from the views of researchers working in primary care settings.

Future research

Future research should prioritize delivering considered solutions to diagnosis and care in collaboration with patient and community groups. This needs to be inclusive of those from racial and ethnic minority groups, those with lower socioeconomic status, rural populations, older populations, and those who experience physical and behavioral challenges to access care. Evaluating HCC-specific social determinants of health, inclusion, and diversity (e.g., ethnicity, age, rurality, socio-economic status, access to health care) in different geographical areas and health boards within the UK is important in providing tailored care that emphasizes equity in policy and practice.