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

Abstract 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.


| INTRODUCTION
Globally, musculoskeletal diseases, among which rheumatic diseases, put a significant burden on societies: they currently rank first among causes of years lived with disability (YLD), with an increase in YLD of 19.8% between 2010 and 2019 (Vos et al., 2020), causing lower quality of life (Branco et al., 2016;Sloot et al., 2016). Studies report impaired mental health (Branco et al., 2016;Kristiansen et al., 2012;Park et al., 2020), negative effects on social interactions (Sloot et al., 2016) and challenges within the healthcare system, such as insufficiently effective medication (Radawski et al., 2019) or limited attention being given to pain (Erwin et al., 2018).
Due to the diverse impacts of rheumatic diseases on a range of societal, physical, social and care aspects, there is an increasing interest in the needs and questions of patients. This has been inspired by studies in other disease areas which have shown that although patients' perceptions may overlap with those of professionals and researchers, they can also be substantially different (e.g. Broerse et al., 2010;da Silva et al., 2010;Tallon et al., 2000). In the area of rheumatic diseases, the objectives for collecting insights on patient perceptions have varied from improving care practices (e.g. Erwin et al., 2018) to building research agendas (e.g. Parsons et al., 2017;Radawski et al., 2019). Recent studies on the quality of life and needs of patients with rheumatic diseases mainly used quantitative surveys (e.g. Hunter & Riordan, 2014;Ingegnoli et al., 2020). While quantitative research has provided relevant insights into the prevalence of challenges such as pain and fatigue, it does not allow insights into the real-world complexity behind survey responses. Additional qualitative research is essential to acknowledge and map contextualised experiences of patients. Various studies have taken such a qualitative approach but have explored needs within a specific care context (e.g. Erwin et al., 2018;van Eijk-Hustings et al., 2013) or have aimed at research agenda creation (e.g. Verwoerd et al., 2021) and have not integrated perspectives of actors in their surroundings that provide their care and support. Non-majority perspectives or conflicts in perspectives among various actors therefore remain hidden.
A recent review by Fairley et al. (2021) concluded that identification of patients' needs that are broader than healthcare is needed to improve person-centred care. We argue that challenges are best investigated holistically to inform a broad architecture of solutions that address these challenges. As highlighted by Fairley et al. (2021), an exploration of the complexity of the life experiences and challenges of rheumatic disease patients is currently missing. Considering the challenges of those who closely interact with patients and are responsible for their care and support can provide a further base for the exploration of solutions. To the best of our knowledge, such a broad, multi-actor perspective has not been performed or documented yet in the scholarly literature.
The current study, therefore, aims to gain insight into the realworld complexity and variety of the daily life challenges experienced by patients and their family and friends, healthcare professionals and their work and education environment in relation to rheumatic diseases in the Netherlands and the interrelations between these challenges. These insights could provide a starting point for the development of a broad array of solutions to these challenges.

| METHODS
This study was part of a larger action research project that aimed to develop strategic aims for the Dutch Arthritis Foundation (DAS).
The first part of the project aimed to explore and prioritise the challenges experienced by patients and those who provide their care and support; we report on this part in this paper (hereafter: 'this study').
We based our approach on the principles of the Dialog Model (DM; Abma & Broerse, 2010). The DM is a transdisciplinary research approach to formulating and prioritising complex health-related problems, that applies different principles to ensure successful multi-actor consultations: active engagement of relevant actors, good social conditions, respect for experiential or practical knowledge, emergent and flexible design, and neutral and structuring process facilitation.
In the first phase of this study we explored the experienced challenges of patients and those who provide their care and support through online, creative focus group discussions (FGDs) and interviews. Such an open approach is essential to acknowledge the richness of real-life experiences and the complexity of different actors' challenges. In the second phase, these challenges were ranked by a broad group of actors through a survey. The data were gathered between May and December 2020.

| Patient and public involvement
In preparation for the overall project, we interviewed various employees of the DAS, healthcare professionals and researchers to get their advice on the design of the project and the actors that should be included. We also interviewed two patient representatives of the DAS to get their advice about the design and execution of the data collection activities during phase 1 of this study. The patient representatives were, together with other employees of the DAS, also part of an advisory board. During the first advisory board meeting, they reflected on the envisioned design of this study, and the actors that should be invited. During a second meeting, they reflected on the study's progress and on a concept version of the qualitative analysis.

| Phase 1: Problem identification (May-October 2020)
role in providing (in)formal care and support for patients; (2) family and friends (hereafter 'significant others'; 3) primary, secondary and tertiary healthcare professionals working with rheumatic disease patients, and (4) employment and education experts.
Participants with rheumatic diseases and significant others were recruited through the DAS 0 network and invitations on various (online) platforms (e.g. DAS website and network). We identified healthcare, work and education professionals with rheumatic diseases expertise through an Internet search. We invited them to participate by email and telephone, either personally or through an open invitation to organisations or departments. Using Google forms, patients, significant others and professionals signed up directly for an FGD or group interview. We use the term 'FGD' for sessions of four to seven participants, and 'group interview' for sessions two to three participants. One patient was not able to participate online, and we, therefore, interviewed them by telephone. Because a limited number of significant others signed up for the FGDs, we conducted one additional interview by telephone after the FGDs. After consultation with the advisory board, we decided to conduct additional interviews with two GPs and a psychologist, as these professions had not yet been represented in the two FGDs with healthcare professionals. In total phase 1 included 57 participants.

| Data collection
Due to 2020s COVID-19 restrictions, all data collection (Table 1) occurred online. We used Zoom for a series of two-hour FGDs and group interviews, guided by two facilitators. Table 2 shows the set-up of all FGDs and (group) interviews, for which we used Padlet (an online application for digital Post-it brainstorming) as a conversation tool. We provided all participants with extensive technical support to allow full participation. All Padlet boards were saved, and sessions were audio and video recorded after consent was given.

| Analysis
Directly after initiating data collection, we started open coding the Padlets, field notes and audio recordings. We inductively coded participants' experienced challenges (Strauss & Corbin, 1990) using Atlas.ti 9. Expressed needs were translated into challenges by formulating them as something that is currently missing. Through iterative open and axial coding (Strauss & Corbin, 1990), codes were compared, refined and connected to understand different components of the challenges and the relationships between them. Through selective coding (Strauss & Corbin, 1990), related challenges were clustered into themes using the 'networks' function of Atlas.ti to create a mind map of challenges. Open, axial and selective coding was done through a continuous iterative process. By ongoing analysis of the data directly after each FGD or (group) interview, we gradually induced predominant themes for the challenges mentioned by multiple actors. This led to purposeful data collection during subsequent sessions by prompting with the previously identified themes in mind (Birks & Mills, 2015), enabling us to gradually improve our understanding of the (causal) relationships between challenges. Authors SH, JN, LLL, MM and CP contributed to coding through frequent conversations, negotiations and decision-making regarding the final set of themes.

| Survey design
To gain insights into the way patients and other actors prioritised the identified challenges and needs, we designed a survey using Qualtrics online software (detailed in appendix A). The previously induced themes were translated into 'topics' and grouped in clusters. To limit the number of topics in the survey, various highly interconnected themes were merged. The survey asked respondents to suggest what "more attention should be paid to…", asking them to choose two topics per cluster, wherein ranking something first meant 'needs the most attention' and ranking something second meant 'needs the next (second) level of attention'.

| Survey distribution
The survey was open to all interested individuals and was distributed among the patient panel of the DAS, the IPSOS rheumatic disease patient panel, a large group of researchers working on rheumatic diseases and all participants in phase 1. It was also dispersed through the DAS's social media and newsletter, reaching donors, members and other actors affiliated with the DAS.

| Analysis of survey
Survey data were transferred from Qualtrics to SPSS (version 26). For the analysis, we followed a descriptive statistics methodology similar to the one used by Broerse et al. (2010). Priorities given to themes were allocated points (#1 priority: two points, #2 priority: one point). For example, if the topic pain was prioritised 150 times; 50 times as #1 priority and 100 times as #2 priority, the prioritisation score was 50 � 2 + 100 � 1 = 200. Higher priority scores resulted in a higher ranking. We stratified our results for patients, significant others, healthcare professionals and 'other', and for all patient groups bigger than N = 100.

| RESULTS
In this section, we first present the predominant challenges induced by our analysis. We discuss the relationships between the challenges HARMSEN ET AL.   As employers or education facilities did not respond invitations to participate, other experts/representatives who could provide insights on the impact of rheumatic diseases on work and education in general, and for employers and education facilities specifically, were included.

| CHALLENGES
We identified eight major, highly interrelated themes of challenges (Table 3 and Figure 1).
Patients generally expressed similar challenges; differences between patients appeared less dependent on the type of rheumatic disease and more on their life and disease phase. Challenges seemed more profound during flare-ups (i.e. for AS and RA) and around time of diagnosis. Relatively young people articulated more challenges regarding diagnosis and participation in social life, work and school, while patients of older age (above approximately 70) more often struggled with multiple rheumatic diseases or with different additional impairments.
As Figure 1 displays, certain themes are highly interconnected, and we will therefore discuss them in three parts.
3.1.1 | Physical and mental challenges and impaired function Figure 1 shows the physical challenges that patients experienced, their impairing effect on function and the resulting mental challenges.
The effects of physical challenges on functioning in daily life were often mentioned as part, or the root cause, of another challenge and are therefore discussed within the themes of physical and mental challenges. Some challenges were also recognized by significant others and healthcare professionals.

Physical challenges
During the FGDs, most patients mentioned 'pain' and 'stiffness' as common physical complaints. The most mentioned consequence of these was being less able to move, making certain activities difficult or impossible, such as lifting things at work or hobbies like rowing, biking or gardening. Many patients also experienced (excessive) fatigue, generally and/or after activities.
For many patients, the unpredictability and invisibility of symptoms were particularly challenging as they influence social life (discussed later) and scheduling. Also, pinpointing and maintaining physical limits was reported to be difficult, because negative physical effects are often delayed. Combined with mental challenges, this causes patients to push their limits.
Many patients said they struggled to move (sufficiently). Moving aids stiffness and overall health, but moving can also cause or worsen complaints. Many patients struggled to find the right form and amount of exercise. This can lead to daily dilemmas, that is exercising while in pain to alleviate pain, and movement being both the remedy for and the cause of stiffness.
"Sports feel like a vicious circle. For example, you should exercise more, but if you cannot exerciseif already your abdominal exercises cause pain in your toe, for example -you do not do it anymore.
And then it worsens. You get into a vicious circle." The set-up of all (group) interviews and FGDs

Element Description
Session preparation Several days before their session, patients receive a preparatory booklet containing creative preparatory exercises (cf. Mattelmäki, 2006;Sleeswijk Visser, 2009). This preparation is done to allow for a shorter, less burdensome focus group or interview, by sensitising participants to the topic beforehand.
Significant others receive preparatory questions to deliberate on to sensitise them to the topic.
Session part 1 Exploration of challenges people experienced while living with rheumatic diseases or taking care of people with rheumatic diseases.
1a a Participants write down their experienced challenges on Post-its in the Padlet application. Simultaneously, facilitator 1 organizes their challenges in clusters. Participants can see each other's contributions on the Padlet board.
1b a The challenges are discussed, guided by facilitator 2. The facilitator asks participants to elaborate on their posted answers, aiming to gain deeper insights into the relationships between and causes and impacts of these challenges. Meanwhile, facilitator 1 adds new insights from the discussion to the Padlet board, which all participants can see. 1c Participants are asked to mark the challenges they prioritised.
Session part 2 Exploration of participants' need to overcome the challenges identified in part 1.

Mental challenges
Patients reported that physical challenges and their impact on functioning caused various mental challenges. Many voiced a constant search for balance in life, not only regarding the (amount of) physical activities and sleep they had but also regarding balancing their limited energy between leisure and obligations. Leisure activities improve mental wellbeing but may worsen symptoms. Many of the patients said they did certain activities but accepted that they would 'pay the price' later. Mental challenges link to challenges in social life, which we will discuss later.
3.1.2 | Challenges regarding self-management and information, options and collaboration in healthcare Figure 2 illustrates the main challenges that were faced by patients and healthcare professionals.

Self-management
Almost all patients said that they constantly looked for ways to manage and balance their life with their rheumatic disease via food, exercise and the optimal scheduling of activities. Many expressed a need for support in this self-management.

Information and options in healthcare
The patients noted the challenges that exist regarding the availability of certain types of information and care. The healthcare professionals provided insights into the reasons why diagnoses and the allocation of adequate care can be difficult.

Work and education
Various patients reported that their disease impacted employment.
Two specifically confirmed that there were challenges regarding education. Work and education representatives also explored challenges for employers and schools concerning students or employees with rheumatic diseases.

| Respondents
The initial number of respondents to the survey was 2256, though only 1802 respondents completed the survey.

Healthcare professionals
Healthcare professionals' and patients' priorities differed on various topics. The qualitative results combined with those shown in

Significant others
Interestingly, significant others ranked topics connected to social life and how patients are approached by others higher than patients.
These included 'invisibility […]', 'misunderstanding from outsiders due to limitations' and 'awareness of rheumatic diseases and their consequences among the Dutch public'. During the FGDs, these topics were also prominent. Perhaps significant others rank them higher because they themselves also experience (the consequences of) these challenges.

| DISCUSSION
Despite vast improvements in rheumatic disease care since the midtwentieth century, we found that the disease still has a considerable impact on the daily lives of many patients. Physical complaints were prioritised most by patients -mainly fatigue and pain. Previous studies have found that pain is constant and impactful for many patients (Erwin et al., 2018;Hunter & Riordan, 2014). Fatigue has also been recognized as an important issue (de Wit et al., 2013) and is increasingly understood to be a complex phenomenon connected to elements such as sleep and biological and psychosocial factors (Davies et al., 2021). Our research adds to this knowledge by F I G U R E 3 Prioritisation of themes by various (a) main actor groups and (b) patient groups. The percentage indicates the proportion of points allocated to the theme, relative to the total number of points allocated to all themes HARMSEN ET AL.
T A B L E 5 The ranking of themes and topics, stratified for various survey respondent groups. (For topics, the percentage is the proportion of points allocated to the topic, relative to the total number of points allocated to all topics within a theme. For themes, the percentage indicates the proportion of points allocated to the theme, relative to the total number of points allocated to all themes. Rank 1 and 2 are indicated by green or yellow highlight respectively.)

| Strengths and limitations
A strength of the research is the use of the mixed-methods approach, which allows for better interpretation of both the qualitative and the quantitative data. We argue that quantitative results should be seen

| CONCLUSION
We conclude that our cross-domain, context-rich, and multi-actor study on challenges faced by people with rheumatic diseases can provide a starting point for various actors who wish to take action to improve the lives of this group. Any solutions need to take into account the complexity of these challenges.

ACKNOWLEDGMENT
We are grateful to the people who participated in our research who are living with arheumatic disease and to their relatives for their openness and trust and for helping us to better understand the impacts of a rheumatic disease on daily life. We also want to thank the various care professionals who have shared their experiences with us. We thank the Dutch Arthritis Society for their support. The data collection of this research was commissioned by the Dutch Arthritis Society (and part of a larger research/strategy development trajectory). However, the full data analysis and writing of this paper took place after the commissioned project had ended. As such, the Dutch Arthritis Society had no influence nor participation in the analysis of the data or writing of this paper.

ETHICS STATEMENT
According to Dutch law, the ethical approval of a formal medical ethics committee was not required because this study was noninvasive and participants were aged over 18. The research complied with the national Code of Ethics for Research in the Social and Behavioural Sciences involving Human Participants (VCWE, 2018).
All respondents received written and verbal information about the aim, data analysis and later process of the study and were told that they could withdraw from the study at any time without explanation.
All data were anonymised before being analysed. You can click next. Then you will proceed to the second part of the questionnaire. In this section, you are asked to prioritise challenges regarding living with rheumatic disease and opportunities for research and care (which do you think are most important?). This part will take about 10-15 min.

Part 2 Challenges
Discussions with patients, significant others, care professionals and other (care) organisations have revealed various challenges and needs that people with a rheumatic disease experience in daily life.
These challenges are divided into 6 themes. We ask you to indicate your top two per theme: which challenges do you think should receive the most attention?
It concerns the following six themes: 1. Physical complaints 2. Self-management 3. Information and options in healthcare 4. Collaboration in healthcare 5. Social and mental wellbeing 6. Work and education

Prioritisation of topics within themes.
For each theme the following was asked, after which a list of topics appeared: Within the theme [THEME NAME] a number of topics have been mentioned as a challenge and/or need.
Please indicate a top 2 of topics, within the theme [THEME NAME], that you think should receive the most attention. Left of the topic, you can enter a number 1 and 2. In the top 2 you enter, you regard the topic for which you fill in a 1, the most important.
Within the theme [THEME NAME], more attention should be paid to the challenges… [The following challenges were presented, for each theme]