Understanding leisure‐time physical activity: Voices of people with MS who have moderate‐to‐severe disability and their family caregivers

Abstract Background Physical activity (PA) is beneficial for all people, yet people affected by multiple sclerosis (MS) find regular PA challenging. These people may include individuals with the disease who have moderate‐to‐severe disability and their family caregivers. For researchers and clinicians to effectively promote PA among caregiver/care‐recipient dyads with moderate‐to‐severe MS, a comprehensive understanding of the shared PA experiences of these dyads would be beneficial. Objective We explored shared experiences of caregiver/care‐recipient dyads affected by moderate‐to‐severe MS about PA and directions for intervention. Methods Six focus groups with 23 people with moderate‐to‐severe MS and 12 family caregivers were conducted. Data were analysed using a constant comparative approach. Results Three major themes emerged as follows: (i) PA is a continuum, (ii) cycle of disengagement and (iii) cycle of adjustment. The first theme captured the dyads understanding that PA falls along a continuum ranging from highly structured to unstructured activities. Cycle of disengagement captured the experiences of dyads engaging in little or no PA. These dyads perceived internal and external issues as drivers of the cycle of disengagement, while availability of supportive programmes and services or people helped the dyads to break out of the cycle. When the cycle of disengagement was broken, the dyads described moving towards the cycle of adjustment, where they were able to learn skills and take action to incorporate PA into daily routines. Conclusion This research highlights the need to adopt an integrative approach that acknowledges the caregiver/care‐recipient dyad with moderate‐to‐severe MS as a focus for PA intervention.


| INTRODUCTION
The progressive nature of multiple sclerosis (MS) and associated motor, cognitive and psychological symptoms mean that people with this condition may become increasingly disabled as the disease progresses. [1][2][3][4] Consequently, many people with MS (PwMS), especially those individuals with moderate-to-severe disability (ie, significant walking limitations that require support for gait PDDS 3-7 or EDSS ≥6), often need emotional, physical and instrumental support to manage associated life roles and maintain independence. 5,6 Providing this on-going support falls primarily on the family caregivers 7,8 who may spend up to 10 hours per day for caregiving activities. 9,10 Together this evidence suggests that the impact of MS does not rest solely on the individual with disease and that PwMS and their family caregivers often need to adapt to its presence as an interdependent caregiver/ care-recipient dyad. 11 The definition of caregiver/care-recipient dyad adopted for this study is as follows: the reciprocal partnership of two or more persons who enact caring roles towards one another. This partnership may include the individual with a disease and a close friend or relative usually a spouse, adult child, sibling, unmarried partner, or parent. 12,13 Researchers have demonstrated that regular participation in physical activity (PA) has beneficial effects on all aspects of health and quality of life for all people including PwMS. [14][15][16] Yet recent estimates suggest that approximately 60% of PwMS are physically inactive (ie, fail to meet public health guidelines of ≥30 min/d of moderate-tovigorous intensity activity) compared with 23% of the general population. 1 Furthermore, people with progressive subtypes of MS have lower PA levels compared to those who have a relapsing clinical course, 17 suggesting that a higher disability level is associated with physical inactivity. 18 With increasing caregiving responsibilities, family caregivers of people with moderate-to-severe MS-related disability may have limited time and opportunity to participate in PA. 19,20 To date, PA promotion interventions in the MS literature have been individually oriented and focused primarily on individuals with mild-to-moderate MS-disability. 17,[21][22][23][24] However, the developmentalcontextual model of coping with chronic disease suggests that the disease has an impact on both caregivers and care-recipients and that dyads who engage in collaborative activities such as PA are more likely to experience better adjustment. 25 Furthermore, research in other chronic disease contexts (eg, cancer and dementia) has demonstrated that a dyadic approach to PA improves several health-related outcomes for the dyad including muscle strength, physical function and psychological health. [26][27][28][29][30] Collectively, this work highlights the importance of targeting the dyad as an equal and interactive unit and suggests that PA promotion interventions that are mutually beneficial to the needs of PwMS and caregivers are warranted.
For researchers and clinicians to effectively promote PA among caregiver/care-recipient dyads with moderate-to-severe MS-related disability, a comprehensive understanding of the shared PA experiences of these dyads would be beneficial. Qualitative data are extremely valuable for providing insights to guide the development and implementation of appropriate interventions. 31,32 However, the majority of qualitative studies on PA among PwMS do so from the perspective of those with mild disability, with little consideration given to the experiences of those with moderate-to-severe disability. 33,34 Furthermore, no study has documented shared perspectives of caregiver/care-recipient dyads affected by MS about PA. In response, this qualitative study was undertaken to explore shared perceptions of caregiver/care-recipient dyads affected by moderateto-severe MS-related disability about PA. A secondary objective was to identify directions for intervention among these groups in order to inform the development of an intervention that supports their joint engagement in PA. The main question that guided our research was as follows: What are the shared perspectives of caregiver/care-recipient dyads with moderate-to-severe MS-related disability about PA?

| Study design
We chose a qualitative, cross-sectional, descriptive study design to answer our research question. 35 A focus group method was used, as it facilitates interactive discussion and exchange of ideas among participants. [36][37][38] Such interactive focus group discussions are reported to provide deep insight into multiple views on health behaviours. 39

| Setting and participant recruitment
Focus group participants were recruited from three communities with populations ranging from 39 000 to 474 800 located within a single Canadian province. Potential participants were informed of the study through (i) a recruitment phone call to individuals who had participated in a previous study conducted by investigators in our research team and consented to being contacted for additional research projects, (ii) newspaper advertisements and (iii) information flyers distributed through local MS clinics and MS Society chapters.
The principal investigator (PI) or a trained research assistant (RA) conducted a telephone screening using a pre-defined script developed for this study. During the phone screening, we asked the primary contact person (ie, either the PwMS or caregiver) to provide contact details of a possible partner for the focus group. Once provided, the PI or RA made a separate phone call to the partner to screen for eligibility.
Potential participants were eligible to take part in the study if they met the study's eligibility criteria in Table 1.

| Participants
A total of 97 individuals (58 PwMS and 39 caregivers) expressed interest in the study. Nineteen PwMS and 22 caregivers decided not to be screened after receiving more information about the study and what was involved. After completing the screening process, nine PwMS and five caregivers were deemed ineligible (due to disease severity or transportation difficulties). Another seven PwMS were eligible but chose not to participate because the group timing was inconvenient. Thirty-five eligible participants comprising of 23 people with moderate-to-severe MS-related disability and 12 caregivers returned a copy of the signed consent materials, and these individuals were enrolled in the study. A flow chart summarizing the recruitment and enrolment process is presented in Figure 1.

| Procedure
The focus groups were conducted between April and August 2015.
Participants were assigned to groups based on practical reasons (ie, timing and locations where participants were recruited) in order to make participation as straightforward as possible. Each focus group was composed of a mix of dyads and single individuals. These single individuals were PwMS whose caregivers did not attend the groups. semistructured discussion guide was used (Data S1). All the focus groups were digitally recorded and professionally transcribed.

| Data management and analysis
Descriptive statistics were used to report the sample characteristics. Focus group data were analysed using a constant comparative approach. 40 This approach was chosen to enable us to explore areas where the views of each partner converged or diverged in order to produce a shared understanding of the dyad experience of PA. The PI reviewed the transcripts against the digital recordings for accuracy and replaced participants' names with pseudonyms. The PI and RA independently coded the transcripts after multiple readings. The coding process began with making notes in the margins of the transcripts about the participants' comments and what was interesting about them. The next step was to develop an initial list of codes by identifying concepts embedded within the data. During this process, the two researchers compared and discussed their codes until they reached consensus. When disagreements were encountered, the senior author provided guidance. Once all the initial codes were identified, the two researchers met with the senior author to review and refine the codes, as well as develop a coding framework.
The search for themes involved condensing the codes into descriptive categories that offered a conceptualization of the shared dyad experience of PA. Another meeting with the senior author was held, in which a hierarchy of overarching themes and subthemes was discussed. This process was complemented by the development of a thematic map, which provided an overall conceptualization of the patterns within the data and relationships between them. Different iterations of the thematic map were discussed and revised with the senior author to ensure the patterns and suggested relationships represented on the map were consistent with the agreed interpretation of the data. Exemplar quotations were extracted from the transcripts to provide a basis for understanding the themes and their unifying properties.
The final themes, subthemes and definitions are provided in Table 2. ATLAS.ti v7 software (Scientific Software Development GmbH, Berlin Germany) was used to facilitate data management and analysis.

| Trustworthiness
The strategies recommended by Lincoln and Guba 41 were used to ensure rigour. Dependability and confirmability were achieved using an audit trail to connect the raw data and codes with themes and subthemes. To enhance credibility, two researchers independently analysed the data, discussed and compared findings and consulted with the senior author in case of a disagreement. Each participant was mailed a summary of his/her individual focus group approximately 2 weeks after the group for member-checking. Each participant was asked to comment on the summaries if inaccuracies were detected. Participants were given the option to decline if they did not wish to be part of this procedure. Of the 35 participants, two declined to participate because of vacation plans, 21 responded and expressed no concerns with the summaries and 12 did not return the feedback form despite three follow-up attempts.
Transferability was achieved by presenting sufficient contextual information and raw data in this paper to allow readers to evaluate the themes and to decide whether they can be applied to their own situations.

| Focus groups
In total, there were six focus groups and each group lasted an average of 93 minutes (SD=17.3; range=59-107 minutes). Tables 3 and 4

| Themes
The experiences shared by the participants were captured in three related themes: "PA is a continuum," "cycle of disengagement" and "cycle of adjustment" (Figure 2). Overall, the participants' experiences of PA as a structured-unstructured continuum were consistent across the focus groups. The cycle of disengagement emerged from statements made by participants engaging in little or no PA.
A combination of internal and external issues kept them locked in this cycle. These participants were able to engage in PA when they entered the cycle of adjustment, which occurred over a period of time. Availability and usage of adequate support systems helped them to break out of the cycle of disengagement and enter the cycle of adjustment. In the discussion that follows, the themes and subthemes are described with quotes that illuminate their meanings. At the beginning of each quote, a pseudonym is included with PwMS or CG used to denote a quote from a participant with MS or caregiver, respectively.

| Overarching theme: PA is a continuum
To orient the participants to the phenomenon of interest, we asked them to describe what PA meant to them. The main theme that emerged from the analysis of the focus groups was that PA falls along a continuum ranging from highly structured to unstructured activities.
This theme was captured in the stories shared by both caregivers and care-recipients. We heard some dyads describe PA as any structured, planned and supervised activity that is performed within the bounda-

| Theme 3: Cycle of adjustment
This theme was more apparent among dyads that were currently engaging in some PA and captured their reflections on the adjustments they had to make to incorporate PA into their daily routines. For most of the participants, the process of adjustment occurred over a long period of time, but as soon as they entered into this cycle, they reported needing less support to be able to maintain their PA. For these dyads, the adjustments involved redefining what PA meant to them and shifting their expectations on types of PA to engage in, as well as the mechanism of engagement. This theme was more common in stories shared by dyads who had been living with MS for many years and reflected in two subthemes: (i) acceptance and (ii) innovation and modification.

Acceptance
The dyads talked at length about how having MS affected all aspects of their PA experience. They also shared the importance of accepting the reality of MS and adapting to the changes in their abilities to engage in PA, as well as the need to change the way they think about PA.

| Limitations
This study has some limitations that warrant consideration. First, our sample comprised majorly of middle-aged (40-59 years) and older adults aged ≥60 years. Given the literature suggesting that middle-aged and older adults with MS are less physically active than young adults affected by the disease, 70 it is possible that our findings may have been influenced by age-related factors in addition to MS-disability. Future researchers may want to explore the differences in PA perspectives between young and older adults affected by moderate-to-severe MS-related disability. Second, we did not have objective information on the PA levels of all our participants. Future researchers may want to gather this information in next studies to provide additional depth of analysis.
Third, although we utilized many strategies to recruit both members of the dyad, recruiting family caregivers proved to be challenging. As a result, a "selection effect" may have occurred where the caregivers who participated in the focus groups may have been different from those who chose not to participate. Fourth, we combined caregivers and PwMS in the same groups rather than having separate groups. While this strategy allowed for an in-depth exploration of coconstructed experiences, it may have influenced what the partners felt they were able to say during the groups. For example, caregivers may have wanted to protect their care-recipients by not expressing certain views and vice versa. It is therefore possible that we may have been able to further tease out the nuances of the PA experience between caregivers and PwMS if we had conducted additional separate groups and this would be an interesting avenue for future research.
In addition, it is possible that the amount of caregiving time may influence perspectives on PA. Our study is limited by not having detailed information on caregiving time and tasks. Future researchers in these areas are encouraged to gather this additional information to look at this relationship. Finally, some of the challenges associated with focus groups include domineering or quiet members, the moderator's inability to control the course of discussion and the unnatural setting in which the interviews are conducted. 71,72 In this study, we attempted to manage these challenges through training of the facilitator, supervision by a senior investigator during each focus group, using a small group composition and ensuring that the focus groups occurred at a place and time of convenience for the participants.

| CONCLUSION
This research has highlighted the need for both people with MS and caregivers to adapt to the impact of the disease on their lives and work together to find options to engage in PA. Given that participants described PA as a continuum of widely varied activities, interventions should not solely focus on promoting structured exercise, but also encourage everyday PA while taking the needs of both caregivers and care-recipients into consideration. These findings highlight the vital importance of adopting an integrative approach that acknowledges the dyad as a focus for PA interventions, which is highly relevant given the rising burden of MS on society.