‘It struck at the heart of who I thought I was’: A meta‐synthesis of the qualitative literature examining the experiences of people with multiple sclerosis

Abstract Background People with multiple sclerosis (MS) have varied experiences and approaches to self‐management. This review aimed to explore the experiences of people with MS, and consider the implications of these experiences for clinical practice and research. Methods A meta‐synthesis of the qualitative literature examining experiences of people with MS was conducted using systematic searches of ProQuest, PubMed, CINAHL and PsycINFO. We incorporated feedback from team members with MS as expert patient knowledge‐users to capture the complex subjectivities of persons with lived experience responding to research on lived experience of the same disease. Results Of 1680 unique articles, 77 met the inclusion criteria. We identified five experiential themes: (a) the quest for knowledge, expertise and understanding, (b) uncertain trajectories (c) loss of valued roles and activities, and the threat of a changing identity, (d) managing fatigue and its impacts on life and relationships, and (f) adapting to life with MS. These themes were distributed across three domains related to disease (symptoms; diagnosis; progression and relapse) and two contexts (the health‐care sector; and work, social and family life). Conclusion The majority of people in the studies included in this review expressed a determination to adapt to MS, indicating a strong motivation for people with MS and clinicians to collaborate in the quest for knowledge. Clinicians caring for people with MS need to consider the experiential and social outcomes of this disease such as fatigue and the preservation of valued social roles, and incorporate this into case management and clinical planning.


| INTRODUC TI ON
Multiple sclerosis (MS) is one of the most common inflammatory neurological conditions and a major cause of non-traumatic neurologic disability among younger adults. 1 Worldwide, more than 2.2 million people, mostly female, are estimated to be affected. 1 MS varies in its presentation, clinical course and the frequency and severity of symptoms experienced. Many people present initially with a relapsing-remitting form of the disease, characterized by symptom-free periods and recovery which follow attacks or relapses. 2 For others, MS begins as a primary progressive form, or develops into secondary progressive MS, with gradual worsening of neurological symptoms and increasing disability over time. 2 Although several risk factors have been identified, the cause of MS remains unknown and to date, there is no known cure. 2,3 Many disease-modifying therapies are available that can reduce symptoms and relapse frequency, with the ultimate aim of preventing all disease activity. 4 Most of these treatments modify immunity and are administered variously via oral, intramuscular, subcutaneous and intravenous routes. All treatments carry risk of side effects, including pervasive flu-like symptoms as a direct consequence of treatment (type 1 interferons), heightened susceptibility to infections as a result of immune suppression, and drug hypersensitivity and injection site reactions, 5 which can impact people's willingness to use them. 6 Overall, the relationship between therapies and disease outcomes is uncertain for any particular person, as is the range of side-effects a person may experience.
Perhaps because of the heterogeneity of disease experiences of MS, the literature has tended to atomize, rather than synthesize these experiences. Qualitative studies have focused on the experiences of people with MS at particular points in time (eg diagnosis, early stage and relapse), 9,10 in specific populations (eg women and mothers), [11][12][13][14] in relation to specific assessments or interventions (eg rehabilitation, physical activity, disease-modifying therapies or alternative therapies), [15][16][17][18][19] or of specific symptoms or consequences (eg fatigue or sexual dysfunction). [20][21][22] The purpose of this review was to: (a) conduct a systematic

| ME THODS
We used the scoping review approach described by Arksey

| Research question
The overarching question underpinning this review was as follows: How do people experience living with MS? Two further questions were defined: (a) What are the key experiences explored in the qualitative literature? and (b) What common themes underpin these experiences?

| Searches
Systematic searches were conducted in ProQuest, PubMed, CINAHL and PsychINFO using the search string ('multiple sclerosis') AND (experienc* OR perception* OR perspective* OR attitude* OR belief* OR value* OR view*) AND (qualitative OR 'focus group*' OR interview* OR narrative*).

| Study inclusion and exclusion criteria
Inclusion criteria were studies with empirical qualitative data about adults' subjective experiences of living with MS (2010 to January 2019). Mixed-method studies were included if qualitative data could be extracted. Studies that focused on the experience of others (eg carer/family/health-care professionals) were excluded. Studies in the grey literature and those not written in English were also excluded.
Experiences of the person with MS included physical, social and/ or psychological impacts of the disease, health systems and services, health-care professional interactions and disease management.
Studies describing experiences related to specific interventions or treatments (eg a specific activity programme as opposed to all physical exercise or a specific drug as opposed to all disease-modifying therapies) were excluded.

| Study quality assessment
All included studies were appraised using the Critical Appraisal Skills Program (CASP) qualitative checklist 25 by two researchers working independently. Title and abstract, and full-text screening was performed by two reviewers. Any disagreements were resolved by a third reviewer.

| Charting the data
A thematic analytical approach was adopted to provide a rich description of MS experiences. 26 Data familiarization was achieved through several stages of article review. Coding and interpretation began at title and abstract screening, and were refined as the data were reviewed. Initial coding involved arranging-related types of experiences conceptually into categories, capturing disease domains (diagnosis, progression and relapse, physical and psychological symptoms) and contexts of people's lives (work, social and family life; the health sector). We coded and compared the breadth and commonalities of experience across these domains and contexts. Final coding was conducted using NVivo 12, a qualitative data analysis computer software package. 27 We undertook blinded audits to ensure consistency of codes and concepts between reviewers. Any differences in approaches were resolved through discussion across the research team.

| Synthesis with knowledge experts
To improve the authenticity of the synthesis, 28

| Ethical approval
This review did not include direct involvement with human participants; it was a secondary analysis of research data, and therefore in accordance with the National Statement on Ethical Conduct in Human Research 2007 (Updated 2018) did not require ethical approval. 30

| RE SULTS
Of 1680 articles identified in the initial search, 77 met the inclusion criteria ( Figure 1). Ages of participants ranged from 18 to 81 years; two-thirds were female. The data collection method used most frequently was interviews (84%), followed by focus groups (14%) ( Table 1). The country of participants' origin most represented in the studies was the UK (23%), followed by the United States (17%); Scandinavian countries (12%); and Iran (12%).

| Quality assessment
The quality of all 77 studies was considered acceptable using the CASP tool (Table S1). The criterion of adequate consideration of the relationship between researcher and participants was met in only 34% of studies. 12% of studies used recruitment strategies which we did not consider appropriate to address the aims of their study. In 13% of studies, ethical issues were either inadequately addressed, or information about consent, recruitment and/or obtaining approval of a human research ethics committee was not provided.

| Qualitative synthesis
We identified five overarching themes describing people's experiences  (Table 2).

| Uncertain trajectories and a need to plan
This theme described the inherent uncertainty people with MS experienced across all aspects of their lives. It was expressed around the time of diagnosis, 9,10,12,17,31-34 regarding treatment, 6,15,41,44,58 and in terms of the potential future impacts of MS progression, especially on work, 35,43,56,59 family and relationships. 11,14,21,47 Several studies included experiences of physical and psychological symptoms presenting themselves acutely and without warning.

| Loss of valued roles and activities, and the threat of a changing identity
The impact and fear of future impact of MS on valued roles and activities were frequently reported, reflecting the way that MS posed challenges to self-perceptions and perceived identity, and difficulties in adapting to a changing body and altered capabilities. 38 Guilt and shame associated with changing roles and abilities affecting family dynamics were expressed, 14,32,50,65 including an inability to provide for family through loss of employment and income. 35

| Managing fatigue, and its impacts on life and relationships
Managing fatigue required constant planning and pacing of tasks to accommodate the anticipated fatigue-related after effects. 12 14,20,47,51,53,54,77,78,80 and physical and psychological health. 16,18,49,78 People with MS described feelings of frustration about the limitations that fatigue imposed on their lives and the resulting loss of spontaneity. 20,31,49 Knowing how to manage this was a source of confusion, with some people highlighting exercise 16,18,49,78 and diet 49 as effective, and others attributing fatigue and relapse to incorrect, or too much, exercise. 16,49 Information and support to manage fatigue were found to be lacking for some, despite the significant impact it had on their lives. 20,22 People described emotional fatigue in relation to seeking information and support, and with interactions with health services. 40,48 This was influenced by an overall perceived lack of legitimacy 40,42 of invisible symptoms, and experiences of having to repeatedly explain or justify limitations to friends, family and workplaces, 33,42,48,52 to fight for needs from health professionals and government organizations, 33,36,40,52 and the need to regularly re-establish relationships with rotating or changing health-care providers. 40

| Adapting to life with MS
Strategies that people with MS used to adapt to MS ranged from, and oscillated between, denying the existence of their condition 45 to total acceptance. 14 Although denial of the diagnosis was experienced by some, 9,32 defiance in the sense of not letting MS and its impacts define identity, personal outlook and everyday life was most often expressed. 14,32,35,51,78 For some people with MS, initial fears surrounding dependence on aids, such as wheelchairs, were replaced with acceptance and relief, as they facilitated adaptation to certain tasks, and assisted in maintaining independence. 31,32,73,81 Technology and devices were valued for enabling people to stay connected to society and community, 82 follow online exercise programmes, 43 monitor activity and fitness levels, 18 and assist with daily living activities 53 and work-related tasks. 59 The most frequently reported strategy for everyday coping was to draw on personal resources. Resources could include work, 66,71 spiritual faith, 35,72,83 family support 12,33,84 (including financial support to access care 15,68 ) and social interaction, including engaging with other people with MS. 10,18,42,49,50,84 Our team members with MS affirmed the themes, and articulated some linkages across the themes, in narratives synthesized into I-poems (Box 1). These poems contributed to the title of our paper.

TA B L E 1 (Continued)
certainty about the very information that would enable them to do so (eg unknown aetiology and unpredictable prognosis, symptom trajectories and responses to treatment). This is challenging in the context of a diverse and information-laden health landscape. 86 In a qualitative study examining people with MS' experiences, needs and preferences for integrating treatment information into decision making, participants described a desire for unbiased and up-to-date information. On the other hand, they reported an excess amount of information available, of which only a small amount was of relevance to them. 87 Overall, participants expressed a desire to develop a 'research partnership' with health professionals to facilitate tailoring of information to meet their unique health needs. 87 Acknowledging the presence of uncertainty with health professionals is the first step to achieve this aim.

| Limitations
A strength of our study is the incorporation of quality appraisal, which is not a requirement of scoping review approaches. 23

ACK N OWLED G EM ENTS
This research was funded by and has been delivered in partnership with Our Health in Our Hands (OHIOH), a strategic initiative of the Australian National University, which aims to transform health care by developing new personalized health technologies and solutions in collaboration with patients, clinicians and health-care providers.

CO N FLI C T O F I NTE R E S T
The authors declare that they have no competing interests.