Series: Public engagement with research. Part 2: GPs and primary care researchers working inclusively with minoritised communities in health research to help address inequalities.

Public engagement in health research is vital for addressing health disparities and promoting inclusivity among minoritised communities who often face barriers to accessing healthcare. Minoritised communities are groups, which have been made minorities by a dominant culture, race, ethnic group and/or social class and may experience health inequalities as a result. By incorporating diverse perspectives and lived experiences of minoritised communities, this approach aims to achieve contextually relevant research outcomes that reduce health inequalities and improve overall well-being. However, underrepresentation and lack of inclusivity challenges persist, necessitating the establishment of inclusive partnerships and grassroots participatory methodologies.To foster inclusive public engagement, it is important to overcome structural and cultural barriers, address socioeconomic challenges, and build trust with minoritised communities. This can be achieved by promoting a cultural shift that values inclusivity, providing comprehensive training to researchers, and collecting rigorous data on engagement demographics for transparency and accountability. Involving minoritised communities in decision-making through participatory research approaches enhances trust and yields successful outcomes. Additionally, allocating sufficient resources, collaborating in co-production, and prioritising the diverse needs and perspectives of stakeholders contribute to fostering inclusive public engagement in research.Overall, inclusive engagement practices particularly in primary care research have the potential to reduce health inequalities and cater to the unique requirements of minoritised communities, thereby creating more impactful outcomes and promoting equitable healthcare access.

There is an important need to engage with minoritised communities in primary care researchEngaging diverse communities in research helps produce relevant research to address health inequalities.The exclusion of minoritised communities from research can be addressed by taking action towards more inclusive engagement.

The most important problems and needs of rasopathy patients with a noonan syndrome spectrum disorder.

BACKGROUND: Noonan syndrome spectrum disorders (NSSDs) constitute a group within the Rasopathies, and are one of the largest groups of syndromes with impact on multi-organ involvement known. The extreme variability of the clinical phenotype is, among others, due to the numerous different genes that are involved, and the differences in clinical presentation over the life span. We have studied the needs of patients and their relatives aiming to develop, evaluate and choose focus in research, medical care and policy to better meet their perspectives. METHODS: Using the participatory and interactive Dialogue method, 80 patients and relatives mentioned 53 different problems or needs (topics) that were categorized into eight themes. These themes and the topics within each theme, were subsequently prioritized by putting them in order of importance methodologically. RESULTS: The four highest prioritized themes were: (1) Physical problems (non-musculoskeletal related); (2) Social, emotional and behavioral problems; (3) Cognitive functioning and information processing; and (4) Problems related to the musculoskeletal system. Nineteen out of the 53 topics were physical problems. According to the total group of respondents, the top 3 prioritized topics within theme 1 were coagulation problems, heart problems, and feeding problems. Also data stratified by age groups, phenotype (NS and other NSSDs) and gender showed some remarkable results. For instance, feeding problems were prioritized as the most important topic of the highest prioritized theme, according to patients aged 0-12 years. Also feeding problems show a significant difference in its prioritization according to female patients (2) compared to male patients (7). On the other hand, heart problems were not mentioned in the top three prioritized topics in the youngest age groups, although heart problems are generally considered most important for patients with NSSD. CONCLUSIONS: With our results we underline the importance of methodologically inventorying the needs of NSSD patients, not only at the group level, but to also focus on specific needs according to e.g. age, phenotype and gender. For instance, it is remarkable that both the current Clinical Guidelines and the Noonan Syndrome diagnostic criteria give little to no attention to feeding problems, though our results indicate that, to the youngest patients, these problems have top priority. A similar situation appears to apply to the clinical management of e.g. coagulation, neuropsychological and musculoskeletal problems (like physiotherapy or occupational therapy) and to a need for (educational) tools to support patients at school or at work. Our study may help to shape targeted (clinical) management, research and policy inside and outside medical (research) institutes and shed light on the complex phenotypes of NSSDs, the families' and patients' perspectives on the everyday consequences of the many different problems, as well as their needs.

Decision-Making Dilemmas within Integrated Care Service Networks: A Systematic Literature Review.

Introduction: The diverse nature of people's care needs requires collaboration between different organisations and sectors. One way of achieving such collaboration is through integrated care service networks. Decision-making is considered an important aspect of network governance and key to achieve further integration of care services. As integrated care scholars only implicitly seem to touch upon the issue of decision-making, we aimed to identify multiple decision-making dilemmas. Theory and Methods: A systematic literature review was conducted of eighteen empirical studies in which decision-making dilemmas in integrated care service networks were inductively identified. To frame and understand these dilemmas, we partly drew on Provan and Kenis' governance models and their hypothesised decision-making dilemma for service networks. Results: Identified decision-making dilemmas included 1) autonomy versus interdependence, 2) diversity versus coherence, and 3) self-interest versus common goals. In line with Provan and Kenis' hypothesis, we highlight a cross-cutting dilemma of inclusiveness (all viewpoints are considered hence widely supported decisions) vs. efficiency (reaching timely decisions). Discussion and conclusion: We believe that network- and 'systemic' stakeholders both need to reflect upon and learn from decision-making dilemmas to work towards widely supported and adequate decisions. This is important for achieving aligned and holistic care services that many people desire.

An impact model to understand and improve work-life balance in early-career researchers in radiation oncology.

Purpose: The COVID-19 pandemic had a substantial effect on mental health and work productivity of early-career researchers working in Radiation Oncology (RO). However, the underlying mechanisms of these effects are unclear. The aim of the current qualitative study was therefore to achieve a better understanding of how these effects arose and could be managed in the future. Methods: This study was conducted jointly by RO and qualitative health researchers. Data was collected in four online Focus Groups with 6-11 RO researchers (total N = 31) working in Europe. The transcripts were analysed through a qualitative cross-impact analysis. Results: Causal relations were identified between seventeen variables that depict the impact of disrupted working conditions. Mental health and work productivity were indeed the most important affected variables, but relations between variables towards these impacts were complex. Relations could either be positive or negative and direct or indirect, leading to a cascade of interrelated events which are highly personal and could change over time. We developed the model 'impact of disrupted working conditions' depicting the identified variables and their relations, to allow more individual assessment and personalised solutions. Conclusion: The impacts of disrupted working conditions on RO researchers varied due to the complexity of interrelated variables. Consequently, collective actions are not sufficient, and a more personal approach is needed. Our impact model is recommended to help guide conversations and reflections with the aim of improving work/life balance. The participants showed high levels of personal responsibility towards their own mental health and work productivity. Although being an individual issue, a collective responsibility in developing such approaches is key due to the dependency on organizational variables.

Mapping the complex everyday challenges and needs of people with rheumatic disease and their surroundings using a multi-actor approach.

INTRODUCTION: This study aimed to gain insight into the real-world complexity of the challenges experienced by patients, their significant others, care professionals and the work and education environment concerning rheumatic diseases as well as the interrelation between these challenges; it also aimed to prioritise the identified challenges. METHOD: Using the Dialog Model, 21 people with various rheumatic diseases, 24 care professionals, 9 significant others, and 3 education and work representatives were asked about rheumatic disease-related challenges and needs in a series of focus groups and interviews. Data were inductively coded and analysed, resulting in a mind map thematically displaying the challenges. The mind map was translated into a survey, and respondents (N = 1802) prioritised themes and challenges. RESULTS: Of the six identified themes, 'physical complaints' was prioritised the most, followed by 'collaboration in healthcare', 'social and mental wellbeing', 'self-management', 'information and options in healthcare' and 'work and education'. Challenges of people with rheumatic diseases appeared to be complexly interrelated. For instance, fatigue and pain affect everyday functioning, but can also heavily impact social and mental wellbeing. To facilitate support for these challenges, which many patients desire, patients and care professionals said that better collaboration between primary and secondary care professionals is needed. Additionally, patients felt that their experiential expertise deserves more acknowledgement from care professionals. Results were similar across different rheumatic diseases. CONCLUSION: Many patients desire more support to manage life with their disease. To facilitate this, collaboration and communication between healthcare professionals, and between healthcare professionals and individual patients, should be improved.

Monitoring and Evaluation of Patient Engagement in Health Product Research and Development: Co-Creating a Framework for Community Advisory Boards.

PURPOSE: While patient engagement is becoming more customary in developing health products, its monitoring and evaluation to understand processes and enhance impact are challenging. This article describes a patient engagement monitoring and evaluation (PEME) framework, co-created and tailored to the context of community advisory boards (CABs) for rare diseases in Europe. It can be used to stimulate learning and evaluate impacts of engagement activities. METHODS: A participatory approach was used in which data collection and analysis were iterative. The process was based on the principles of interactive learning and action and guided by the PEME framework. Data were collected via document analysis, reflection sessions, a questionnaire, and a workshop. RESULTS: The tailored framework consists of a theory of change model with metrics explaining how CABs can reach their objectives. Of 61 identified metrics, 17 metrics for monitoring the patient engagement process and short-term outcomes were selected, and a "menu" for evaluating long-term impacts was created. Example metrics include "Industry representatives' understanding of patients' unmet needs;" "Feeling of trust between stakeholders;" and "Feeling of preparedness." "Alignment of research programs with patients' needs" was the highest-ranked metric for long-term impact. CONCLUSIONS: Findings suggest that process and short-term outcome metrics could be standardized across CABs, whereas long-term impact metrics may need to be tailored to the collaboration from a proposed menu. Accordingly, we recommend that others adapt and refine the PEME framework as appropriate. The next steps include implementing and testing the evaluation framework to stimulate learning and share impacts.

Persuasive communication in medical decision-making during consultations with patients with limited health literacy in hospital-based palliative care.

OBJECTIVE: Both patients in the palliative phase of their disease and patients with limited health literacy (LHL) have an increased risk of being influenced by healthcare providers (HCPs) when making decisions. This study aims to explore to what extent persuasive communication occurs during shared decision-making (SDM) by (1) providing an overview of persuasive communication behaviours relevant for medical decision-making and (2) exemplifying these using real-life outpatient consultations. METHODS: An exploratory qualitative design was applied: (1) brief literature review; (2) analysis of verbatim extracts from outpatient consultations and stimulated recall sessions with HCPs; and (3) stakeholder meetings. RESULTS: 24 different persuasive communication behaviours were identified, which can be divided in seven categories: biased presentation of information, authoritative framing, probability framing, illusion of decisional control, normative framing, making assumptions and using emotions or feelings. CONCLUSIONS: Persuasive communication is multi-faceted in outpatient consultations. Although undesirable, it may prove useful in specific situations making it necessary to study the phenomenon more in depth and deepen our understanding of its mechanisms and impact. PRACTICE IMPLICATIONS: Awareness among HCPs about the use of persuasive communication needs to be created through training and education. Also, HCPs need help in providing balanced information.

Establishing the research agenda for oral healthcare using the Dialogue Model-patient involvement in a joint research agenda with practitioners.

Engagement of patients in the composition of a research agenda is essential to reduce the gap between research and practice and thereby generate more impact. The aim of this study was to develop a research agenda for oral health. Experienced challenges and needs with oral health(care) of practitioners and patients formed the input for the research agenda. We describe the identification of research priorities of patients and the integration of these with previously identified research priorities of practitioners, using a participatory multi-phase approach for research agenda setting (Dialogue Model). Via focus group discussions, 32 research topics were generated. Next, 1495 patients prioritized these topics in an online survey. In a dialogue meeting, a joint research agenda of eight research topics was agreed upon. Many topics were contributed by patients, but were prioritized by both stakeholder groups. The most important topics concerned behavior change and the relation between general and oral health. Other topics that were prioritized covered affordability and accessibility as well as health system research and organizational issues. By considering different perspectives, this research agenda has uncovered directions for future research that go beyond evident research topics, as many topics are currently underrepresented in oral healthcare research.

Patient engagement in the design of clinical research in Noonan syndrome spectrum disorders: a scoping review.

BACKGROUND: Noonan syndrome spectrum disorders are a group of disorders caused by mutations in several genes of the RAS/MAPK pathway. Because of a highly heterogeneity and variable phenotypical manifestations of the disorders, these children and adults have a variable number of symptoms. Inclusion of their perceived experience of their health and developmental problems in research (design) could contribute to increased relevance of the research process and outcomes. The aim of this study is to get insight in what way patients with a Noonan syndrome spectrum disorder have been involved in the research process in order to learn for future engagement practices. METHODS AND RESULTS: To that end, the degree of engagement was measured by the eight levels of the participation ladder of Arnstein. Using a scoping review approach, 18 articles were selected in which patient engagement in the design of studies in patients with Noonan syndrome spectrum disorders was described over the past twenty years. Six of these articles reported engagement on the level of informing (level 3), 8 on the level of consultation (level 4), 2 on the level of placation (level 5)and 2 on the level of partnership (level 6). CONCLUSIONS: The current results do show a positive albeit still modest development of patient engagement over the last few years. A promising way to stimulate engagement is aiming to yield insights in the most important patients' needs by developing a patient guided research agenda. However, this is not automatically followed by patient engagement at higher levels of participation in subsequent research steps. For this reason, in the Netherlands for example, a Dutch Noonan syndrome spectrum disorders research agenda is being developed, in a collaboration between the Dutch Noonan Syndrome Foundation and national scientific and clinical professionals.

A research agenda on oral health care as a boundary object that unites the perspectives of patients and practitioners.

CONTEXT: A research agenda for oral health care was established in the Netherlands using the Dialogue Model. This project served as a case study in which we applied boundary-work theory as a framework to understand boundaries (ie demarcations) between and within groups, and how these boundaries can be overcome. OBJECTIVE: To gain insights into the boundaries encountered when setting a research agenda, we analysed how this agenda served as a boundary object (ie circumstances, situations or material that connect actor groups and allow boundary crossing) that facilitated crossing boundaries and uniting the perspectives of patients and practitioners. METHODS: We used a thematic approach to analyse researchers' observations, meeting materials, emails, interviews with patients (n = 11) and a survey among patients and practitioners (n = 18). RESULTS: Setting the research agenda helped to cross boundaries in oral health care, which demonstrates its role as a boundary object. First, this made it possible to integrate research topics representing the perspectives and priorities of all patients and also to unite those perspectives. It was essential to involve practitioners at an early stage of the project so that they could better accept the patients' perspectives. This resulted in support for an integrated research agenda, which facilitated the crossing of boundaries. CONCLUSIONS: The research agenda-setting project was found to serve as a boundary object in uniting the perspectives and priorities of patients and practitioners. PATIENT CONTRIBUTION: Patient involvement in this case study was structured in the process of research agenda setting using the Dialogue Model.

Appropriate leadership in nursing home care: a narrative review.

PURPOSE: The purpose of this paper is to synthesize the existing evidence on leadership that best matches nursing home care, with a focus on behaviors, effects and influencing factors. DESIGN/METHODOLOGY/APPROACH: A narrative review was performed in three steps: the establishment of scope, systematic search in five databases and assessment and analysis of the literature identified. FINDINGS: A total of 44 articles were included in the review. The results of the study imply that a stronger focus on leadership behaviors related to the specific context rather than leadership styles could be of added value in nursing home care. RESEARCH LIMITATIONS/IMPLICATIONS: Only articles applicable to nursing home care were included. The definition of "nursing home care" may differ between countries. This study only focused on the academic literature. Future research should focus on strategies and methods for the translation of leadership into behavior in practice. PRACTICAL IMPLICATIONS: A broader and more conceptual perspective on leadership in nursing homes - in which leadership is seen as an attribute of all employees and enacted in multiple layers of the organization - could support leadership practice. ORIGINALITY/VALUE: Leadership is considered an important element in the delivery of good quality nursing home care. This study provides insight into leadership behaviors and influencing contextual factors specifically in nursing homes.

"The challenge of managing insecurities": Parents' experiences with the care for their child with congenital diaphragmatic hernia.

PURPOSE: Last decennia remarkable advances have been made in decreasing the mortality rate of children with congenital diaphragmatic hernia (CDH), resulting in a relatively growing patient group with long-term complications and complex care needs. These consequences have a huge impact on the quality of life of both children and their families. To provide practical recommendations for improving the quality of care for this patient group, the present study sought to obtain insights into the experiences and needs of parents with a child with CDH. DESIGN AND METHODS: A qualitative study was conducted on the experiences and needs of parents with a child with CDH living in the Netherlands. Data was obtained by means of a discourse analyses of 17 weblogs written by parents and three online focus groups with 8-12 parents per group (n = 29). The data was analysed thematically and structured by using the model of Lawoko (2007) on parental satisfaction with care. RESULTS: Although parents were generally satisfied with the delivered care, they frequently encountered challenges in managing insecurities throughout the care process. Besides the unpredictable disease progress, insecurities were exacerbated by: (a) limited specialized knowledge of long-term consequences, (b) logistical problems, and (c) nontransparent communication. Providing security through, for instance, a clear care plan and by engaging parents in the decision-making process helped them feel more in control. PRACTICE IMPLICATIONS: This study showed that parents' main challenge was to manage insecurities. Creating securities by providing a care plan and involving parents in the decision-making process helped parents to feel more in control. To improve quality of care for children with CDH, future measures should, therefore, focus on reducing insecurities by managing expectations, improving transparency and stimulating engagement.

Setting Priorities for Optimizing Vascular Access Decision Making--An International Survey of Patients and Clinicians.

BACKGROUND: Many decisions around vascular access for haemodialysis warrant a collaborative treatment decision-making process, involving both clinician and patient. Yet, patients' experiences in this regard have been suboptimal. Although clinical practice guidelines could facilitate collaborative decision making, they often focus on the clinicians' side of the process, while failing to address the patients' perspective. The objective of this study was to explore and compare kidney patients' and clinicians' views on what vascular access-related decisions deserved priority for developing guidelines that will contribute to optimizing collaborative decision making. METHODS: In the context of updating their vascular access guideline, European Renal Best Practice surveyed an international panel of 85 kidney patients, 687 nephrologists, 194 nurses, and 140 surgeons/radiologists. In an electronic questionnaire, respondents rated 42 vascular access-related topics on a 5-point Likert scale. Based on mean standardized ratings, we compared priority ratings between patients and each clinician group. RESULTS: Selection of access type and site, as well as prevention of access infections received top priority across all respondent groups. Patients generally assigned higher priority to decisions regarding managing adverse effects of arteriovenous access and patient involvement in care, while clinicians more often prioritized decisions around sustaining patients' access options, technical aspects of access creation, and optimizing fistula maturation and patency. CONCLUSION: Apart from identifying the most pressing knowledge gaps, our study provides pointers for developing guidelines that may improve healthcare professionals' understanding of when to involve patients along the vascular access pathway.

Patient involvement in research programming and implementation: A responsive evaluation of the Dialogue Model for research agenda setting.

BACKGROUND: The Dialogue Model for research agenda-setting, involving multiple stakeholders including patients, was developed and validated in the Netherlands. However, there is little insight into whether and how patient involvement is sustained during the programming and implementation of research agendas. AIM: To understand how the Dialogue Model can be optimised by focusing on programming and implementation, in order to stimulate the inclusion of (the perspectives of) patients in research. METHODS: A responsive evaluation of the programming and implementation phases of nine agenda-setting projects that had used the Dialogue Model for agenda-setting was conducted. Fifty-four semi-structured interviews were held with different stakeholders (patients, researchers, funding agencies). Three focus groups with patients, funding agencies and researchers (16 participants) were organized to validate the findings. RESULTS: Patient involvement in programming and implementation of the research agendas was limited. This was partly related to poor programming and implementation, partly to pitfalls in earlier phases of the agenda-setting. Optimization of the Dialogue Model is possible by attending to the nature of the agenda and its intended use in earlier phases. Attention should also be given to the ambassadors and intended users of agenda topics. Support is needed during programming and implementation to organize patient involvement and adapt organizational structures like review procedures. In all phases the attitude to patient involvement, stakeholder participation, especially of researchers, and formal and informal relationships between parties need to be addressed to build a strong relationship with a shared goal. CONCLUSION: Patient involvement in agenda-setting is not automatically followed by patient involvement in programming and implementation. More attention should be paid, in earlier stages, to the attitude and engagement of researchers and funding agencies.

The involvement of gynaecological patients in the development of a clinical guideline for resumption of (work) activities in the Netherlands.

CONTEXT: Most initiatives for patient involvement in guideline development have been carried out for chronic diseases. The involvement of patients with incidental and non-threatening diseases is more complicated. Little knowledge is available on how these patient groups can successfully be involved in guideline development. OBJECTIVE: To assess the effectiveness of the involvement of gynaecological patients in the guideline development for resumption of (work) activities after surgery. DESIGN: At three different stages patients were involved in the process: (i) three focus group discussions (FGDs) were organized, (ii) patients were involved for the instruction video, and (iii) patients tested the patient version of the clinical guideline. To assess the effectiveness, an evaluation framework was used. The guideline development process was divided into two parallel trajectories in which patients and professionals were consulted separately. Patients were primarily consulted for the development of the patient version, although their input also influenced the recommendations for resumption of (work) activities after surgery. Professionals were mainly involved in the development of the recommendations of the clinical guideline. DISCUSSION AND CONCLUSIONS: The involvement of gynaecological patients in the guideline development for resumption of (work) activities after surgery was successful in many respects. Consultation of individual patients by means of FGDs and with regular feedback moments has been rather effective for a guideline development process related to an incidental, non-threatening disease for which there is no patient organization. Patients' input contributed to applicability of the clinical guideline in daily practice. Increased patient involvement could be achieved by integration of the two parallel trajectories with additional participatory activities, such as a dialogue meeting.

eHealth program to empower patients in returning to normal activities and work after gynecological surgery: intervention mapping as a useful method for development.

BACKGROUND: Full recovery after gynecological surgery takes much longer than expected regardless of surgical technique or the level of invasiveness. After discharge, detailed convalescence recommendations are not provided to patients typically, and postoperative care is fragmented, poorly coordinated, and given only on demand. For patients, this contributes to irrational beliefs and avoidance of resumption of activities and can result in a prolonged sick leave. OBJECTIVE: To develop an eHealth intervention that empowers gynecological patients during the perioperative period to obtain timely return to work (RTW) and prevent work disability. METHODS: The intervention mapping (IM) protocol was used to develop the eHealth intervention. A literature search about behavioral and environmental conditions of prolonged sick leave and delayed RTW in patients was performed. Patients' needs, attitudes, and beliefs regarding postoperative recovery and resumption of work were identified through focus group discussions. Additionally, a literature search was performed to obtain determinants, methods, and strategies for the development of a suitable interactive eHealth intervention to empower patients to return to normal activities after gynecological surgery, including work. Finally, the eHealth intervention was evaluated by focus group participants, medical doctors, and eHealth specialists through questionnaires. RESULTS: Twenty-one patients participated in the focus group discussions. Sufficient, uniform, and tailored information regarding surgical procedures, complications, and resumption of activities and work were considered most essential. Knowing who to contact in case of mental or physical complaints, and counseling and tools for work reintegration were also considered important. Finally, opportunities to exchange experiences with other patients were a major issue. Considering the determinants of the Attitude-Social influence-self-Efficacy (ASE) model, various strategies based on a combination of theory and evidence were used, resulting in an eHealth intervention with different interactive functionalities including tailored convalescence recommendations and a video to communicate the most common pitfalls during the perioperative period to patients and employers. Fifteen patients in the focus groups, 11 physicians, and 3 eHealth specialists suggested points for improvement to optimize the usability of the eHealth intervention and judged it an approachable, appropriate, and attractive eHealth intervention to empower gynecological patients. CONCLUSIONS: The IM protocol was a useful method to develop an eHealth intervention based on both theory and evidence. All patients and stakeholders judged the eHealth intervention to be a promising tool to empower gynecological patients during the perioperative period and to help them to return to normal activities and work.

Patient involvement in a scientific advisory process: setting the research agenda for medical products.

Patient involvement in scientific advisory processes could lead to more societally relevant advice. This article describes a case study wherein the Health Council of the Netherlands involved patient groups in an advisory process with a predefined focus: setting a research agenda for medical products development. A four-phase approach was developed to stimulate needs-articulation concerning future medical products for a broad range of patient groups covering 15 disease domains. 119 (expert) patients and 92 non-patient representatives were consulted using interviews and focus groups. In a facilitated way, patients appeared capable and willing to provide input useful for an advisory process. A broad range of medical products was defined serving different purposes. This study showed two dilemmas: first, finding a balance between a predefined focus and being sufficiently broad to enable patients and patient representatives to contribute, and second, finding a balance between relevance for many patients groups and saturation of data for a lower number of patient groups. By taking the context of patients' daily life as starting point patient groups provided new insights. The predefined focus was sometimes perceived as constraining. The GR considered the articulated needs constructive and incorporated patients' input in their advice to the Minister of Health.