Renegotiating dimensions of the self: A systematic review and qualitative evidence synthesis of the lived experience of self‐managing rheumatoid arthritis

Abstract Background As chronic illnesses, such as rheumatoid arthritis (RA), place an increased burden on health‐care systems, the ability of individuals to self‐manage these diseases is crucial. Objective To identify and synthesize the lived experience of self‐management described by adults living with RA. Design A systematic search of five electronic databases (MEDLINE, CINAHL, EMBASE, PsycINFO and ASSIA) was undertaken to identify relevant studies. Data were extracted and quality‐assessed using CASP guidelines. A meta‐synthesis was conducted based on Thomas and Harden's thematic synthesis approach. Results The search identified 8423 publications. After removing duplicates, 6527 records remained of which 32 studies met the inclusion criteria. Quality of studies was moderate to high, yet a considerable lack of reflection on researcher bias was evident. Our analysis identified 28 dimensions of self‐management RA across six domains: (a) cognitive‐emotional, (b) behavioural, (c) social, (d) environmental, (e) physical and (f) technological. Cognitive‐emotional experiences dominated the analysis. Renegotiating ‘the self’ (self‐concept, self‐esteem, self‐efficacy) was a key focus of self‐management among individuals with RA. Conclusion Our findings highlight the focus of ‘the self’ as a central concern in the self‐management of RA. Standardized self‐management programmes may primarily focus on disease management and daily functioning. However, we suggest that personal biographies and circumstances should move to the fore of self‐management support. Registration PROSPERO International Prospective Register of Systematic Reviews 2018: CRD42018100450. Patient or Public Contribution Patient and public involvement was not explicit in this review. However, three authors provided a patient perspective on the self‐management of arthritis and autoimmune disease.


| INTRODUC TI ON
Rheumatoid arthritis (RA) is a highly prevalent chronic autoimmune disease affecting between 0.3% and 1% of the global population. It is more prevalent in developed countries and among women. It is characterized by inflammation of the joints causing pain, stiffness and swelling. It can affect all areas of life, including work, 1,2 family 3 and leisure activities, 4 as well as physical and mental health. 5 Symptoms tend to emerge between the ages of 20 and 40, and within ten years, at least 50% of patients in developed countries are unable to maintain full-time employment. 6 The term 'self-management' refers to the 'day-to-day tasks an individual must undertake to control or reduce the impact of disease on physical health status'. 7(p05), 8 It involves the patient taking increased responsibility for their health in terms of the decisions they make and activities they engage in and can greatly improve health outcomes and the individual's quality of life. 9 Self-management may require individuals and their families to make extensive practical as well as psychosocial adjustments. For example, individuals may find they are unable to perform previously taken-for-granted behaviours, routine tasks and activities, and require greater practical and emotional support from others. 10 Patient education is recognized as integral to the success of self-managing a chronic illness, and great strides have been made in the development and implementation of Chronic Disease Self-Management Programmes (CDSMP). [11][12][13][14] However, despite advances in patient education programmes and biologic treatments, adherence to long-term therapies remains a challenge. [15][16][17][18] Non-pharmacological treatment, such as exercise, diet and lifestyle modifications are particularly difficult behaviours to adopt. 19 Moreover, effective supports and interventions might not be suitable or accessible to everyone. 11 For example, people from low socioeconomic and disadvantaged groups report poorer outcomes and lower levels of adherence in CDSMPs. 20,21 In turn, this may exacerbate the social gradient in chronic disease outcomes. 22 Thus, consolidating qualitative evidence around the lived experience of self-managing RA could inform the development of self-management support and resources.
A recent mega-ethnography 23 summarized evidence from nine qualitative evidence syntheses exploring experiences of RA and established that living life with RA is a precarious and marginalizing experience, which includes RA as an emotional challenge, as an invisible illness and presenting a biographic disruption. 23 While there is much qualitative research exploring the lived experience of RA in recent years, to date, to the best of our knowledge there is no qualitative evidence synthesis addressing the lived experience of self-management. The purpose of this paper was to systematically identify, appraise and synthesize prior qualitative studies of RA to examine the lived experience of self-management. We seek to answer the following research questions: (a) How do patients experience the self-management of RA? and (b) What aspects of their experience are described as most pertinent to living with and managing the condition?

| ME THODS
This study presents a systematic review and synthesis of qualitative evidence of the lived experience of self-managing rheumatoid arthritis. Our approach involves a comprehensive search for and retrieval of qualitative research publications, a critical appraisal of primary studies, a classification of results and a synthesis of key findings. 24 This work was conducted following the 'Enhancing Transparency in Reporting the Synthesis of Qualitative Research (ENTREQ)' guidelines, and a copy of the ENTREQ checklist is available in Appendix E. The inclusion criteria for the articles were organized following the SPIDER framework for qualitative literature 25 : sample: adults, aged 18+ with a diagnosis of RA; phenomenon of interest: lived experience of adults with RA with self-management; and design/ evaluation/ research type: qualitative peer-reviewed primary studies of any design in English language. There was no limit on the year of publication. Mixed-methods studies and studies examining the perspectives of mixed participant groups were included only if data pertaining to participants with RA could be isolated. We included primary qualitative data obtained and interpreted using qualitative methods of data collection (including interviews, focus groups and ethnography) and analysis. We excluded non-empirical articles including opinion pieces, commentaries, systematic reviews and grey literature. We included only peer-reviewed journal articles to maximize the methodological quality and reporting of the included studies. 26 A full list of inclusion and exclusion criterion is included (Appendix B).

| Critical appraisal
All included publications were subject to a global assessment of study quality using the Critical Appraisal Skills Programme (CASP). This assessment was conducted by the first author in consultation with the team. It involved a systematic assessment of each component attributing a score to indicate whether the item had been adequately addressed. The studies were scored out of 10. Scores >9 were deemed high quality; scores between 7 and 9 were deemed moderate quality; and scores <7 were low quality. The 'typology' and characteristics of the qualitative evidence (Appendix D) were also assessed and reported. 27

| Data extraction
All text under the headings 'results' was extracted electronically and entered into computer software (NVivo version 11) by the first author [SD]. The remaining sections, such as the conclusions, were scanned for any additional findings and extracted where relevant.

| Data synthesis
Drawing from both meta-ethnography and grounded theory, Thomas and Harden's 28 'thematic synthesis' approach was used to synthesize the extracted data. Our intention was not to simply aggregate findings, rather we aimed to produce descriptive and analytical themes by translating concepts from individual studies into one another resulting in the development of overarching concepts and new insights. The process of deriving themes from the data was inductive.
[SD] performed a close reading of all papers.

| Study selection
The electronic database search identified 8423 citations. After removing duplicates, 6527 records remained. Based on this screening, 298 records were deemed potentially eligible for inclusion and full texts were accessed for further scrutiny. Following full-text screening, 31 studies were deemed to have met the selection criteria. An updated search in January 2019 resulted in the inclusion of one additional study. Thus, a total of 32 studies were ultimately identified for analysis. The process is reported in Figure 1 using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. 29

| Characteristics of included studies
The 32 studies included in this meta-analysis describe the lived experience of approximately 665 people with rheumatoid arthritis.
Most studies (n = 640) sampled people with rheumatoid arthritis.
Except two studies which included mixed disease populations, incorporating people with psoriatic arthritis (n = 6), unspecified polyarthritis (n = 2) 30 and juvenile idiopathic arthritis (n = 2). 31 In some cases, samples were drawn from larger studies (eg clinical trials) and there may be duplication of participants as a result. [32][33][34][35][36] Comorbidity was discussed in one study but only to highlight it as an exclusionary criteria. 37 Apart from an Iranian study, all participants were from developed countries, predominantly within North America or Europe.
The publication period ranged from 1993 to 2018. The disciplinary affiliation of lead authors was predominantly clinical (nursing n = 9; medicine n = 7; physio/occupational therapy n = 4; public health n = 3), while social scientists represented a minority (health sciences n = 6; social work n = 2). In terms of research design, the studies referred to using a phenomenological approach, grounded theory or ethnography and employed some variation of content or thematic analysis. In nine of the studies, the sampling approach was unspecified. Where sampling was stated, purposeful sampling was most often used. Most studies (n = 28) used semi-structured interviews as the main instrument for data collection, sometimes referred to as 'open-ended' or 'conversational' interviews. In four studies, focus groups were used. Locations of data collection varied from a hospital or similar clinical site, such as an office or private room at a rheumatology clinic or health centre (n = 9); in seven studies, interviews were conducted at non-clinical sites only (eg at the participants home, workplace or university), while a further seven stated that interviews were carried out at a mix of clinical and non-clinical sites.
In one study, interviews were conducted by telephone. Eight studies did not specify a setting or location of interviews. A comprehensive breakdown of the characteristics of included studies is included in Appendix D.

| Quality of included studies
Using the CASP assessment tool, we determined that 17 of the studies were high quality; 15 were moderate quality; and none were considered low quality. All studies provided a clear statement of aims and findings and used appropriate qualitative methodologies. Some weaknesses were revealed in terms of rigour, specifically limited consideration of the relationship between researcher and participant (eg a critical examination of the researcher's own role and potential bias). In 14 studies, the researcher failed to critically examine their own role, potential bias and influence. In terms of credibility, it was sometimes unclear whether ethical issues had been taken into consideration. While researchers tended to state that ethical approval had been granted by a relevant institutional committee, it was observed that meaningful engagement with ethical issues was unclear in 16 studies. In seven studies, the value of the research was not addressed. Recognizing the limitations of applying quality appraisal tools to produce consistent judgements, and to avoid the risk of overlooking more insightful studies, publications were not excluded based on the CASP assessment. [38][39][40] However, the process of systematically assessing and characterizing the literature served to highlight overall strengths and weaknesses in our sample (Appendix C).
As part of the emotional work of self-management, individuals were required to deal with feelings of frustration, anger and depression. Anger was a prominent emotional response 34,[43][44][45][46]48,49,52,53,[57][58][59][60] and formed part of the burden of coping with RA. This was often rooted in frustrations around limitations imposed by pain and fatigue.
Participants described a sense of injustice towards developing the illness. This created a need to identify a cause. 36 Participants were concerned how they would manage their illness in the future. Despite advances in the treatment, participants managed their illness under the perceived threat of immobilization, deformity and dependence. To counter this, strategies of optimism, positivity and humour were frequently adopted. 30,31,[33][34][35]37,42,46,49,53,54,58,59 A small number of studies described how religious belief and practice provided hope and helped to maintain a positive outlook. [46][47][48]56,57,62 The predominant feature of the lived experience of self-managing RA related to 'the self', a broad category constructed to reflect various dimensions of self-concept (beliefs about oneself), self-esteem (self-worth and value) and self-efficacy (confidence in ability to deal with health problems). These concepts were found in 29 of the 32 studies. 31

| Behavioural
Behaviour relates to what the person does; it is an act or an action taken. Across the 32 papers analysed, the central idea that appeared in relation to behaviour concerned adjusting and adapting to RA. [30][31][32][33][34][35]37,41,42,[44][45][46][47][48][49][50][53][54][55]57,58,62 People with RA described how they altered their approach to daily tasks and activities in line with reduced strength and functioning. As Ostlund found, 'behavioural adjustments included more rest, such as going to bed earlier or taking a nap during or after work, in order to be able to fulfil their duties'. 50(p210) Adjusting and adapting to an illness required resilience and the ability to draw from accumulated knowledge to employ effective selfmanagement behaviours.
Pacing and planning daily activities commonly featured in individual's experiences. 30 There was a sense of trading-off and 'dosing' certain activities. Lutze observed that participants 'took the risk of stretching their limits because they so dearly wanted to do special things, or to just feel the pleasure of being "normal"'. 34(p66) People with RA reported a need to carefully plan their lives in a way that minimized impact on their joints, preserved their energy and avoided additional pain and fatigue.
Consequently, self-management reduced spontaneity and created a cognitive burden. Added to this was the unpredictability of symptom flares. The lack of a clear cause of a flare created a sense of uncertainty and reduced self-efficacy.
Physical symptoms of RA created difficulties in managing aspects of self-care, such as exercise and diet, self-maintenance and hygiene (ie getting dressed or brushing hair/ teeth). 31 However, there was also a sense of dependency and being a burden.  7 Optimism, positivity, humour 'What has been most helpful to me (laughter) a sense of humor' 37(p512) 'I think, I have begun to think really positively, that I will get better'. 34(p68) 8

| Social
Religion and spirituality 'I go to church every day to pray to God to recover from the disease'. 43 64 whereby people struggled to convince family, friends and colleagues that they were legitimately 'ill'. Yet, the support of others was described as an essential part of self-managing RA. Paradoxically, some participants reported a desire to conceal-or mask-their illness. RA affected personal and social relationships. [31][32][33][34][35][36][37][41][42][43][44][45][46]48,49,[51][52][53][54][55][56][57][58][59][60][61][62] For some, this meant depending more on family and friends for emotional or physical support. Where participants felt unsupported by family, friends or colleagues, it usually concerned a lack of emotional support: 'My family are not what I wished them to be. I wish they accepted me the way I am and understood my special condition'. 56(p246) As Ostlund surmises 'living with RA not only includes the person affected, but also those close to them'. 60

| Environmental
The environment refers to the setting in which people manage RA, including the built environment and climate. People with RA reflected on the accessibility of their environment. 31

| Physical
The physical symptoms described across the studies related to pain, 34

| Technological
Participants identified various technologies they used to manage RA. Technologies refer to the tools that people used to self-manage their illness, including products such as medical devices and medications, as well as services provided by health-care professionals. Assistive devices and aids were often used. 31,36,41,43,47,50,54,56,[62][63][64] Generally, they were perceived as opportunities for greater independence. Some studies described the experience of taking medication, including alternative medicines and treatments. [31][32][33][35][36][37][41][42][43][44]46,47,[49][50][51][55][56][57][58][59][60] Medication was often experienced in relation to time, the long and painful wait for a new medication to work or a flare to pass, and anxieties around how long a treatment will continue to be effective. Experiences of health services were discussed in relation to interactions with healthcare professionals (HCP). 31

| D ISCUSS I ON
We Qualitative evidence on the self-management of RA has typically been generated from standard qualitative methodologies, such as semi-structured interviews. We observe a lack of participatory techniques to understand the lived experience of self-managing RA. While advances are being made in the development of self-management interventions, particularly technological, [78][79][80] it is vital that these interventions are informed by a deep understanding of the lived experience of the patient community and their needs and concerns. We suggest the incorporation of techniques that emphasize participation, such as participatory action research, to deepen knowledge of the dimensions of self-concept, self-esteem and self-efficacy.

| Strengths and limitations
This is the first meta-synthesis of the lived experience of self-managing rheumatoid arthritis. As such, the findings will be of interest to health researchers, patient and public involvement and engagement (PPIE) partners, clinicians, patients and policymakers in the area of chronic disease self-management. There are, however, a number of limitations that should be taken into consideration. The systematic review process generated more studies than expected. It was therefore decided that the original study design of meta-ethnography would be unmanageable. On reflection, using a structured approach to selecting an appropriate qualitative evidence synthesis approach would have been beneficial. 64 We maximized the rigour of study selection through team consensus, development of a standardized screening instrument  65 We have presented a comprehensive overview of self-management strategies reported by people with RA; however, their efficacy is not assessed. Future research could evaluate whether the self-management strategies described here lead to improved health outcomes, particularly across social dimensions of gender, class and race.

| CON CLUS IONS
This systematic review and meta-synthesis of qualitative evidence on the lived experience of RA articulates the experience of self-management and points to its many and diverse dimensions. It helpfully conceptualizes a number of domains in which self-management is experienced. It provides a comprehensive and up-to-date summary of the self-management of RA and crucially explores how these dimensions feature in people's lived experience. Our findings highlight the diverse challenges of self-managing RA, including adapting to the illness, balancing roles, pacing activities, feelings of burden, struggles with feelings of dependency and acceptance, and strategies to overcome these challenges identified by people with RA.
Crucially, it emphasizes the importance of negotiating dimensions of the self (self-concept, self-esteem and self-efficacy) in supporting the self-management of RA.

ACK N OWLED G EM ENTS
We would like to acknowledge the contribution of Diarmuid Stokes, Health Sciences Librarian, University College Dublin. We thank colleagues at the UCD Centre for Arthritis Research for their feedback on the development of the review at various stages. As part of a patient and public involvement (PPI) strand within this project, a Research Advisory Group informed our understanding and expertise on self-management. We are grateful for their insight and expertise.

CO N FLI C T O F I NTE R E S T
The authors declare that there is no conflict of interest.

DATA AVA I L A B I L I T Y S TAT E M E N T
The data that support the findings of this study are available from the corresponding author upon reasonable request. A PPE N D I X B

Inclusion criteria
Sample Adults aged 18+ years who have a diagnosis of rheumatoid arthritis.
No restrictions will be applied based on severity or duration of illness or any other demographic variable.

Phenomenon of interest
Perceptions, attitudes, perspectives and experiences of adults with RA towards: The lived experience of RA Self-management, disease management, self-treatment, self-medication Self-care, self-help, self-concept, self-efficacy, self-actualization, self-empowerment Resilience, coping, psychological adaptation Cost of illness, treatment burden Design, evaluation, research type Studies in the English language, from any publication date and any geographical location.
Studies that address the lived experience of self-management or self-care or coping in rheumatoid arthritis patients.
Studies conducted using qualitative methods (eg interviews, focus groups, ethnography). This includes: Primary and secondary qualitative studies. A qualitative study as part of a mixed-methods study.
Mixed-method studies are included (only when qualitative data were reported separately) Peer-reviewed journal articles only.
Studies involving people with diseases other than RA (ie mixed populations) will be included only if outcomes/results for people with RA are presented separately, and only if this is suggested within the title or abstract.

Exclusion Criteria
Sample Children (<18 years) No diagnosis of RA

Phenomenon of interest
Studies that are not qualitative in nature Do not focus on lived experience or address the phenomenon of interest, that is self-management or self-care.
Do not focus on the person with RA specifically (eg experience of spouse; experience of health-care provider/ clinician).
Do not address the disease area of interest specifically or are ambiguous, that is juvenile arthritis, including JIA and JRA, chronic inflammatory arthritis, chronic illness or disease, chronic pain, chronic fatigue, etc Design, evaluation, research type Any perspectives other than people with RA.
Studies that do not state or describe a qualitative method of data analysis.
Non-empirical articles including opinion pieces, commentaries, theoretical articles and treatment guideline documents.
Personal accounts that do not employ qualitative methods of data collection and analysis.
Grey literature/ non-peer-reviewed works, including book chapters, policy reports, technical reports, short reports, conference proceedings and reports, dissertations, commentaries/ editorials.
Dissertations/theses/conference proceedings/published abstracts-however, attempts will be made to source fulllength articles if abstracts are otherwise eligible.