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: Ashley N. Newton, MHA, MAcc, CPA, Michael F. Price College of Business, Division of Finance, University of Oklahoma, 307 W. Brooks St., Rm. 360, Norman, OK 73019-0450; e-mail: 

A Comprehensive Cost-Effectiveness Analysis of Treatments for Multiple Sclerosis

Being a parent can be demanding and stressful, especially for people with chronic diseases such as multiple sclerosis (MS). Parenting can be disrupted by flareups, disease worsening, and other MS symptoms, including mobility problems, pain, fatigue, and cognitive impairment. Mood disorders, such as depression and anxiety, have been found to occur at much higher rates in people with MS than in the general population. Surprisingly, less is known about which factors may predict mood disorders in parents with MS. This study aims to identify potential demographic, clinical, and self-reported predictors that contribute to mood disorders measured by the Hospital Anxiety and Depression Scale.

A total of 285 parents with MS completed an anonymous online questionnaire combining sociodemographic, clinical, and family characteristics and scales, validated in Italian, related to coping strategies and social support. Associations between each variable and mood disorders were assessed using univariate and multivariate logistic regression analyses.

Disability level, emotional and dysfunctional coping strategies, and perceived social support were significant predictors of mood disorders in parents with MS.

These findings confirm the importance of identifying risk factors for mood disorders in parents with MS so that early intervention can minimize mood disruptions caused by the disease.

From Scientific Research (JP, MMU, AT, LP, MMB, GB, MP) and AISM Rehabilitation Service (MMB, GB), Italian Multiple Sclerosis Foundation, Genoa, Italy; and the Department of Physiopathology, Experimental Medicine, and Public Health, University of Siena, Siena, Italy (MAB). 

: Jessica Podda, PhD, Scientific Research, Italian Multiple Sclerosis Foundation, Via Operai 40, 16149, Genoa, Italy; 

 Michele Messmer Uccelli, MSc, is currently employed by Biogen. The other authors have disclosed no relevant financial relationships.

 Funding for this study was provided by the Italian Multiple Sclerosis Foundation (CUP H52I150001900001).

Parenting is a complex process that can be influenced by a combination of factors both internal and external to the individual. Being a parent can be demanding and stressful, and its success can be influenced by chronic diseases such as multiple sclerosis (MS).

 People with MS face a variety of challenges related to living with this disabling disease, which impacts physical and social functioning, including daily activities, roles, and relationships. Thus, the parent-child relationship can be disrupted by flare-ups, disease worsening, and other characteristic MS symptoms, particularly mobility problems, pain, fatigue, cognitive impairment, and mood disorders (MDs).

 The physical and emotional symptoms of MS have been found to specifically affect parents’ participation in their children’s lives, but parental emotions and mood have a greater impact than the physical limitations caused by MS.

 Indeed, MDs have been found to highly impact the lives of people with MS.

 Depression and anxiety occur at much higher rates in people with MS than in the general population.

 of people with MS, and anxiety has been reported to be present in up to 22.1% of people with MS.

 Furthermore, anxiety can overlap with somatic (eg, muscle tension) and nonsomatic (eg, concentrating difficulty) manifestations of the disease, increasing the possibility that MDs and their causes go underrecognized and, consequently, untreated, impacting other clinical outcomes as well.

Parents with MS often report an idealized representation of what they have to do to be “good” parents, and although they want to be able to live up to their ideals, many felt that MS limited them, resulting in feelings of distress and guilt.

 Indeed, mothers with MS had regretted or felt guilty about their inability to fully participate in their children’s physical activities or even to transport them to those activities, especially when hampered by fatigue or mood changes.

 Parents with other major diseases, such as cancer, also frequently report this feeling of inadequacy. Parents with advanced cancer report higher levels of depression and anxiety than patients without dependent children,

 and they struggle with concerns about the impact of an incurable illness on their children and the challenges of being a “good parent”

Interest in understanding factors that may promote psychosocial adjustment and protect against negative outcomes in parental MS is growing. Among these protections, coping strategies can play a pivotal role in reducing psychological pressure and managing stressful situations. The chronic and unpredictable nature of MS suggests that people need to find ways to cope with change and challenges in a continual process of regulation. Coping skills have proved to be crucially important for adjusting to the adaptive demands of chronic diseases, and in the past few years they have received growing interest in MS. For example, evidence indicates that people with MS with higher disability levels have higher depression scores and exhibit avoidance, emotion-centered, and less adaptive coping strategies.

 people with MS routinely adopted avoidance strategies and relied less on task-oriented and positive attitude strategies compared with the general population.

Social support has also been identified as a factor that consistently predicts how well a patient adjusts to living with MS. Increased social support is associated with better mental health outcomes, including reduced MDs and higher quality of life, and also serves as a buffer against the adverse effects of stress.

 A recent qualitative study suggested that the parent with MS’s partner may play an important role in moderating the impact of parenting with MS by providing practical help with housework and emotional support.

 Practical and emotional support from others and the perception that others are available to provide support is a significant indicator of parenting satisfaction.

Although much research and many resources now focus on children or adolescents with MS

 as well as the possible effect of a parent’s MS on children’s and family members’ moods,

 surprisingly less is known about which factors may predict MDs in parents with MS. For these reasons, the aim of this study was to investigate potential variables that contribute to the experience of MDs in a sample of parents with MS.

Individuals were eligible to participate in the study if they had a diagnosis of MS and were parents of at least 1 child younger than 18 years. Individuals provided their consent to participate after reading information about the study and clicking on the option “proceed with the questionnaire,” in accordance with the revised Declaration of Helsinki.

 Participants completed an anonymous SurveyMonkey questionnaire related to coping strategies and social support composed of questions about sociodemographics (ie, sex, age, years of education, current employment), family characteristics (ie, living with/without a partner, the number and age ranges of children), and clinical characteristics (ie, disease duration, disease course, whether a diagnosis of MS was received after the birth of a child). Questions about psychological variables and self-reported scores on an Expanded Disability Status Scale

 derived from the original Expanded Disability Status Scale

 and the Patient-Determined Disease Steps scale

 (both validated in Italian) were also collected. The study received ethical approval from the Regional Ethics Committee, Liguria, Italy.

MDs were evaluated using the Hospital Anxiety and Depression Scale (HADS),

 which consists of 2 separately scored subscales measuring anxiety (HADS-A) and depression (HADS-D). The usefulness of the HADS has been validated as a marker of major depression and generalized anxiety disorder in the MS population. Items are rated on a 4-point Likert scale, with higher scores indicating greater anxiety and depression. A score of 7 or less is normal, 8 to 10 is borderline abnormal, and 11 or higher is abnormal. A score of 8 or higher reflects the presence of anxiety or depression.

Individual coping strategies in difficult or stressful situations were tested using the Brief Coping Orientation to Problems Experienced (Brief COPE)

 self-questionnaire. The Brief COPE measures 14 theoretically identified coping responses classified into 3 categories: problem-focused coping (ie, active coping, instrumental support, planning), emotion-focused coping (acceptance, emotional support, humor, positive reframing, religion), and dysfunctional coping (behavioral disengagement, denial, self-distraction, self-blame, substance use, venting). As a short, 28-item self-report questionnaire (with 2 items for each of the measured coping responses),

 the Brief COPE provides a way to rapidly measure coping responses. Because the scores for the different coping strategies comprise varying numbers of items, and thus are not comparable, a standardized total score is calculated by dividing the total score by the number of items for each of the 3 categories.

Perceived social support was assessed using the Multidimensional Scale of Perceived Social Support (MSPSS).

 This scale is designed to measure perceived social support from 3 sources: family, friends, and a significant other. Participants indicate their level of agreement or disagreement to 12 items on a scale from 1 (very strongly disagree) to 7 (very strongly agree). A higher score represents higher perceived support.

The sample size was considered appropriate to detect an association between mood and potential predictors in parents with MS. Post hoc power analysis—considered at least 5% of the variance in the dependent variable (MDs measured by the HADS) at a 5% significance level and with a sample size of 285 individuals—produced a power of 86%. Sample characteristics are described with means, SDs, ranges, and frequencies.

The HADS score was considered the primary outcome. The cutoff score of 8 for both anxiety and depression was used in a univariate logistic regression model to assess sociodemographic, clinical, and family characteristics; coping strategies; and social support. Variables with a 

 less than .20 were considered for the multivariate model. Separate multiple regression models were fitted for mild symptoms of anxiety and depression. The predictors were simultaneously entered into the model. The results are presented as odds ratios (ORs) and 95% CIs. The multiple regression model was checked for multicollinearity. A variable was eliminated if the variance inflation factor was greater than or equal to 10 or the tolerance limit was less than 0.1. Analyses were performed using Stata Version 15 (StataCorp).

A total of 416 parents with MS were recruited through the social media channels of the Italian Multiple Sclerosis Society. Twenty-four parents with MS did not meet the inclusion criteria, and 107 were excluded because they did not complete the questionnaire. The final sample consisted of 285 parents with MS. The sociodemographic, clinical, and family characteristics; coping strategies; and social support of the 285 parents with MS are reported in 

The univariate logistic regression analyses found that parents with a HADS-A score of 8 or higher (59.0%; n = 168) were more often women (OR, 2.19; 

 = .012), reported the use of dysfunctional coping strategies (OR, 4.25; 

 < .001), and had a lower level of perceived social support (OR, 0.62; 

; all supplemental tables are available in a PDF at the bottom of this article). Parents with a HADS-D score of 8 or higher (30.5%; n = 87) had higher levels of disability (OR, 1.34; 

 < .001); had a primary progressive MS course (OR, 2.61; 

 = .019); were not taking disease-modifying drugs (OR, 0.41; 

 = .005); reported the use of problem-focused (OR, 0.70; 

 < .001) coping strategies; and had a lower level of perceived social support (OR, 0.53; 

). After the multivariate analyses, anxiety symptoms were correlated with disability level (OR, 1.18; 

 = .033), dysfunctional coping strategies (OR, 4.07; 

 < .001), and a lower level of perceived social support (OR, 0.69; 

 = .014) (Table S1). Depression was associated with disability level (OR, = 1.38; 

 < .001) coping strategies, and a lower level of perceived social support (OR, 0.73; 

As a chronic, disabling disease that frequently occurs in young adults in the most productive period of their lives, MS can contribute to psychosocial problems. These can be exacerbated when people with MS face the stress and demands of parenting.

 The psychosocial impact of MS is modulated by several factors that interact in a complex manner, such as MDs.

 However, although many studies shed light on mood alteration in children or adolescents with MS

 or on the possible effect of a parent’s MS on children’s and family members’ moods,

 less is known about which factors may predict MDs in parents with MS. The objective of this study was to identify factors that influence MDs in parents with MS, including sociodemographic, clinical, and family characteristics; coping strategies; and social support. First, this study confirmed that parents with MS have higher occurrences of anxiety and depression (59.0% and 30.5%, respectively) than the prevalence reported in the MS literature.

 Furthermore, for the first time, to our knowledge, these findings highlight that disability level, coping strategies (emotional and dysfunctional), and perceived social support significantly predict MDs in parents with MS.

The relationship between disability level and MDs is not clearly understood and deserves further attention. For example, although some previous studies show that individuals with more severe disability report higher levels of anxiety,

 others do not find any significant correlation.

 The present results suggest that parents with MS experiencing mobility limitations have difficulty assisting their children in recreational activities and transportation,

 and this, in turn, can have a negative impact on mood. Furthermore, people living with a chronic disability use more emotional and avoidance coping strategies, including social support seeking, emotion venting, and mental/behavioral disengagement.

Although problem-focused coping strategies were more often reported in the present sample, the results align with previous evidence that indicates a frequent use of emotion-focused coping in people with MS in general.

 Because people with MS are not able to change the chronic and disabling nature of the disease, it is not surprising that they more frequently use emotional coping skills. Emotion-based coping strategies (eg, acceptance, emotional support, humor, positive reframing, religion) were observed in individuals with higher levels of depression, confirming a tendency to resort to passive behavior.

 people with MS use emotion-focused coping to regulate anxiety, depression, anger, and grief, emotions tied to the stressful conditions associated with having a chronic disease. Interestingly, although less reported compared with problem- and emotion-focused strategies, multivariate analyses reveal that dysfunctional coping strategies have a significant impact on MDs in parents with MS, especially when relevant concomitant physical disability exists.

 Studies show that people with MS more frequently use detachment and avoidance coping strategies, such as mental/behavioral disengagement and denial, than the general population.

 This use of less adaptive coping strategies, such as behavioral disengagement, denial, self-distraction, self-blame, substance use, and venting, may put those living with MS at greater risk for depression or anxiety.

The chronic and progressive nature of MS suggests that people with MS are engaged in a continual process of adjustment to changes and challenges. Evidence indicates the link between poor psychologic adjustment to MS and ineffective coping.

 Although no coping strategy is, per se, maladaptive, some strategies are more effective than others in counterbalancing mental distress. Results suggest that denial and restraint coping may be useful for parents with MS experiencing distress because they help reduce emotional pain. On the other hand, avoidance may have a negative impact on adaptation, particularly in the face of life challenges that require direct engagement,

Social support is an important factor related to mood in parents with MS. In line with previous evidence,

 lower social support was associated with higher depression and anxiety symptoms. Parenting is a complex social role that often places parents’ health care needs behind those of their child. To accomplish parenting tasks and roles, parents with MS rely heavily on their partners (where present), as well as on extended family, friends, and community resources.

 Appropriate social support, however, may not always be accessible or sufficient for the well-being of parents with disabilities. In this case, other positive personal factors, including environmental mastery, self-acceptance, and purpose, could contribute to daily role participation and parental satisfaction in parents with MS.

The present study has several limitations. Participants’ characteristics may limit the interpretation of the results. The sample included middle-aged, low-disability parents with MS with at least 1 child younger than 18 years. Evidence indicates that when people with MS experience a lower level of physical disability, symptoms may be ambiguous, with a variety of manifestations, leading people to question the source of the symptoms and their possible evolution, thus affecting work and social roles. Furthermore, 74.7% of people in the present study had relapsing-remitting MS. The number of relapses over time, their unpredictability, and the exacerbation phase could significantly increase anxiety and depression symptoms

 and lead to more uncertainty about the future than in patients with more apparent progression, in the absence of relapses.

 Furthermore, collecting data via online questionnaires may create bias because technology access and proficiency may exclude some people from participating.

Although perceived support has been found to be more strongly related to quality of life than received support, further examining the differences between perceived and received support might be useful in developing interventions to increase social support for people with MS. Furthermore, information about socioeconomic status, sleep disturbances, and personality traits that may also contribute to MDs in parents with MS should be taken into account.

The present results strongly support the importance of early identification of MDs in parents with MS. Data are required to make strong, evidenced-based clinical practices, services, and policies that will maximize the capacities of parents with MS and their partners. Given that mood has a significant impact on the quality of life of parents with MS and on other family members (eg, affecting sibling psychological adjustment), it is important that health care providers proactively and directly engage patient concerns regarding their role as parents in the context of managing a potentially disabling illness such as MS.

Future research should continue to examine how psychological factors, such as coping strategies and social support, influence MDs and how they can support the mental health and well-being of parents with MS. This is highly important for planning interventions to minimize the disruption of disease on parents’ moods.

Predictors of Mood Disorders in Parents With Multiple Sclerosis: The Role of Disability Level, Coping Techniques, and Perceived Social Support

People with moderate to severe multiple sclerosis (MS) and their family care partners do not engage in sufficient physical activity (PA) for health benefits. Dyadic PA interventions need to be developed to benefit each individual and the dyad. The objective of this study was to engage expert stakeholders in prioritizing and refining key intervention content, delivery methods, and the practical/logistical aspects of a dyadic PA intervention for persons with MS and their care partners.

Thirty-two stakeholders (14 clinicians, 11 people with MS, 5 MS care partners, and 2 representatives of organizations that provide support services for people with MS and/or MS care partners) completed 2 rounds of a modified e-Delphi survey. In round 1, participants rated items across 3 domains: key intervention content (n = 8), delivery methods (n = 9), and practical/logistical aspects (n = 4). Participants contributed additional ideas about these domains, which were incorporated into round 2. Items that did not reach consensus in round 1 were forwarded to round 2 for rerating. Data were analyzed using descriptive statistics and content analysis.

A 24-item list of recommendations was generated, including ensuring that presentation of the intervention content encouraged lifestyle activities in addition to exercise, using videoconferencing rather than teleconferencing as a delivery platform, and stressing the importance of flexibility during the support calls.

Feedback will be used to improve the quality of the intervention. The next step in this line of research involves evaluating the refined intervention in a pilot feasibility trial.

From the School of Rehabilitation Therapy (AF, MF) and the School of Kinesiology and Health Studies (AEL-C), Queen’s University, Kingston, ON, Canada; the Interdisciplinary School of Health Sciences (OM, RK, LAP), Biomedical Sciences, Faculty of Science (RK), Faculty of Medicine (MSF), and Brain and Mind Research Institute (LAP), University of Ottawa, Ottawa, ON, Canada; and the Ottawa Hospital Research Institute, Ottawa, ON, Canada (MSF). 

 Afolasade Fakolade, PhD, School of Rehabilitation Therapy, Louise D. Acton Building, 31 George St, Queen’s University, Kingston, ON K7L 3N6, Canada; 

 The authors declare no conflicts of interest.

The pathology of multiple sclerosis (MS), including neurotoxicity, inflammation, and neurodegeneration, contributes to a complex array of heterogeneous symptoms and disability.

 With increasing disability, many people with MS have difficulty with completing daily activities and maintaining social relationships.

 These individuals require ongoing support to manage associated life roles as the disease progresses and disability accumulates. Informal care partners (ie, family or close friends) are the main providers of support for people living with MS.

 Many care partners, particularly those caring for persons with moderate to severe MS (ie, significant walking limitations that require support for gait), experience poor physical and mental health outcomes due to their caregiving role.

 For example, approximately 50% to 70% of care partners of people with severe MS experience anxiety and depression.

 This evidence points to the need for effective health promotion strategies for these groups.

Despite advances in the pharmacologic treatment of MS, disease-modifying therapies have limited long-term efficacy in preventing the accumulation of disability.

 Best practices for comprehensive MS care recommend a combination of pharmacologic and nonpharmacologic approaches.

 Physical activity (PA) is a health-promoting behavior in the general population, and a cost-effective, nonpharmacologic approach for managing the impact of MS.

 Although there is growing evidence for the benefits of PA, people living with MS have consistently reported low PA levels compared with the general population.

 There is also evidence that care partners of persons with moderate to severe MS disability do not engage in sufficient PA for health benefits.

 These findings demonstrate the importance of acknowledging the unique interrelatedness of health outcomes among MS dyads. Findings also underscore an opportunity for dyadic PA to improve the health of both partners.

In response, we developed Physical Activity Together for MS (PAT-MS), a group-based, remote-delivered, dyadic PA intervention for people with moderate to severe MS and their care partners. We applied the Medical Research Council (MRC) framework

 to guide the design and development of PAT-MS. The MRC framework provides an iterative process to developing complex interventions. The framework consists of 4 cyclical phases: (1) development (identifying existing evidence and theory, applying theory to develop the intervention, and modeling processes and outcomes); (2) feasibility/piloting (testing procedures for feasibility and acceptability); (3) evaluation (understanding the process of change and assessing effectiveness and cost-effectiveness); and (4) implementation (assessing and monitoring long-term effectiveness and dissemination).

, available below this article, illustrates the MRC framework, highlighting the developmental process of PAT-MS.

The full details of the PAT-MS intervention content, including theoretical frameworks, are reported in the published protocol.

 In brief, the content and processes of PAT-MS are theoretically grounded in the Theory of Dyadic Illness Management (TDIM),

 with direction from social cognitive theory (SCT)

 The basic tenet of TDIM is that the way that dyads affected by chronic diseases (eg, MS) appraise disease impact as an interdependent team influences how they engage in health-promoting behaviors (eg, PA), which, in turn, influences dyadic health.

 The TDIM emphasizes the importance of targeting both partners simultaneously to ensure sustainable change in health outcomes for each individual and for the dyad. We used SCT

 to identify key theoretical constructs (eg, self-efficacy from SCT; relatedness and intrinsic motivation from SDT) that influence PA behavior to be included in PAT-MS. Also, SCT

 provided guidance for selecting behavior change techniques (eg, goal setting, problem-solving, and behavioral practice)

 to alter or redirect causal processes that regulate PA behavior (see Table 1 in the published protocol

 for details of behavior change techniques in PAT-MS). PAT-MS includes 3 main components: (1) education (ie, provision of information about PA, shared disease appraisal, and benefits of shared participation in PA as a dyadic coping strategy); (2) guidance from a trained activity coach (ie, an activity coach will provide verbal and written instruction, including behavioral strategies commonly used in dyadic health interventions for persons with neurologic conditions and their care partners

); and (3) social support (ie, provision of practical, emotional, and informational support, including links to community resources for continued PA participation and opportunities to learn from other group members).

Initial works related to the development of PAT-MS have also been published, including a staged needs assessment process to explore dyadic PA

 and a systematic review that provided theoretical understanding of behavioral strategies for change.

 The present study represents step III (modeling processes and outcomes) of the developmental phase. This step involves defining and combining the components of the intervention and identifying potential intervention vulnerabilities.

 Specifically, the most relevant intervention components and ways to tailor and refine intervention contents are identified during this step.

 Researchers have reported the use of various methods, including but not limited to a Delphi consensus process to prioritize and refine intervention components during the modeling step.

 Importantly, the Delphi consensus process provides a pragmatic and flexible methodological tool for engaging a broad range of stakeholders in intervention development work.

 The objective of this Delphi study was to engage people with MS, MS care partners, clinicians, and service providers in prioritizing and refining key content, delivery, and practical/logistical aspects of the PAT-MS intervention.

 the design process involved creating initial survey items based on previous intervention development research

 and the preliminary PAT-MS intervention manual.

Heterogeneous groups (eg, stakeholders from different disciplines) of approximately 30 participants are recommended to provide a range of perspectives and to improve the reliability of the findings.

 We aimed to recruit a purposive sample of 30 participants from 4 stakeholder groups to maximize patient, professional, and public engagement. The stakeholder groups included people living with MS, MS care partners, clinicians with MS-specific knowledge and practice, and representatives of community organizations that provide support services for people with MS and/or MS care partners. The recruitment strategies and inclusion criteria for each stakeholder group are outlined in 

We decided, a priori, to conduct a 2-round e-Delphi survey, with the possibility of a third round for items with no consensus after round 2. Prospective participants were sent electronic invitations that included a study summary, a PDF copy of the preliminary PAT-MS manual, and a personalized survey link via SurveyMonkey (Momentive, Inc). Participants confirmed eligibility and indicated consent online before completing the round 1 survey. Participants who completed the round 1 survey received a personalized email link to the round 2 survey. Participants were provided their previous responses and a summary of the group responses to items from the round 1 survey that required rerating. Each survey round took 20 to 30 minutes to complete and was open for 2 weeks. There was a 2-week interval between survey rounds for data analysis and subsequent survey development. The survey rounds were completed during the COVID-19 lockdown between August 2020 and October 2020. Three research team members tested each survey round to ensure face and content validity and accessibility for participants.

 After the survey was developed, we used an online resource (http://www.readabilityformulas.com/free-readability-formula-tests.php) to improve the readability of the questions. Participants received up to $100 in online gift cards for completing the study. Ethics approval was obtained from the University of Ottawa Science and Health Sciences Research Ethics Board.

The round 1 survey consisted of 4 sections. Section 1 collected demographic information, including evidence of relevant expertise. Section 2 presented 8 questions about content that should be included in PAT-MS and asked participants to rate the importance of each item using a 7-point scale (1 = not a priority and 7 = essential priority). Section 3 presented 9 questions about how PAT-MS should be delivered and asked participants to rate each item using the same 7-point scale. Section 4 presented 4 questions about practical/logistical aspects and asked participants to rate their level of satisfaction with each item on a 7-point scale (1 = very dissatisfied and 7 = very satisfied). Participants were asked to provide qualitative feedback, including rationale for ratings, proposed changes to key intervention components, and concerns with the overall intervention.

The round 2 survey presented 13 items, including 4 original items that did not reach consensus in round 1. These items were related to intervention content (n = 2) and delivery (n = 2). Participants rerated these items with the same 7-point scale used in round 1. Six of the 13 items were modified based on participants’ feedback in round 1. These items were related to intervention content (n = 1), delivery (n = 3), and practical/logistical aspects (n = 2). The remaining 3 items were new items suggested in round 1. These items were related to delivery (n = 1) and practical/logistical aspects (n = 2). Depending on the nature of the modified or new item, participants were presented with the same 7-point scale used in round 1 (for modified items) or multiple-choice or 

 options (for new items). Participants provided qualitative feedback on their ratings.

Data analysis occurred at the conclusion of each survey round. Data were downloaded from SurveyMonkey and imported into IBM SPSS Statistics for Windows, version 27.0 (IBM Corp) for analysis. Descriptive statistics were used to summarize participants’ characteristics and expertise and group responses. Consensus was defined a priori as 70% or more of participants rating the item within 2 points. Items achieving 70% or greater consensus in round 1 were automatically included in the final list of Delphi recommendations. Conversely, items rated by 50% to 69% of participants as 

 were retained in round 2 for rerating. Items rated by less than 50% of participants as 

 were excluded. The qualitative aspect of this study was framed by ontological relativism and epistemological constructionism.

 As such, our assumption is that multiple realities and ways of understanding knowledge exist. These realities are subjective, differ between individuals, and are constructed based on multiple factors, including an individual’s values and experiences. Guided by these philosophical assumptions, we provided opportunities to present participants’ unique perspectives and realities through interpretation of the data and the quotes integrated within the presentation of the findings. A content analysis approach was applied to the qualitative feedback from participants. Specifically, an author (R.K.) read the free-text responses several times and generated codes with a focus on elucidating any reasons for mixed responses or arguments for inclusion/exclusion of items that did not reach consensus. Candidate themes were then developed by another author (A.F.) by grouping codes that coherently reflected participants’ feedback. We compared the qualitative data with the extant literature on dyadic health interventions and the theoretical foundations of PAT-MS so that we could decide whether to include the item in the final list of recommendations. After a discussion process among the research team, the final list of recommendations was developed based on items reaching consensus in rounds 1 and 2 and on qualitative feedback.

Of the 201 stakeholders invited to participate in the study, 2 were ineligible, 6 declined, and 157 did not respond to the invitation. Thirty-six individuals agreed to participate and received the electronic invitation to complete the round 1 survey. After receiving this invitation, 2 individuals declined the invitation, and 2 individuals did not activate the survey link. Thirty-two individuals responded to the round 1 survey (89% response rate): 14 clinicians, 11 people living with MS, 5 MS care partners, and 2 representatives of organizations that provide support services for people living with MS and/or MS care partners. A total of 27 participants responded to the round 2 survey (75% response rate). Participants lost to follow-up were 3 clinicians and 2 people living with MS. The characteristics of enrolled participants are presented in 

For ease of reporting, findings from rounds 1 and 2 of the survey are combined. 

 presents a summary of items reaching consensus in rounds 1 and 2 according to stakeholder group. 

 presents a summary of group responses to the items with multiple-choice or 

 response options in round 2. The final list of recommendations for PAT-MS is presented according to intervention content, delivery, and practical/logistical aspects in 

Five of 8 intervention content items (63%) reached consensus (70%–81%) in round 1 (C1–C3, C5, and C7). Of the remaining 3 items, 2 (C4 and C8) were rerated in round 2 and reached consensus (82% and 85%, respectively) in round 2. The remaining item (C6) was modified into a 

 question. Most participants (78%) voted 

Four of 9 delivery items (44%) reached consensus (70%–93%) in round 1 (D2, D4, D7, and D8). Of the remaining 5 items, 2 (D5 and D9) were rerated in round 2. One of these items (D5) reached consensus (74%) in round 2. The other item (D9) did not reach consensus (41% of participants rated the item as 

). However, given the congruity of this item to the design and theoretical foundations of PAT-MS, the research team decided to include this item in the final list of recommendations. Participants’ responses will be considered in modifying the delivery of PAT-MS (eg, by reducing the time allocated for this item during delivery).

The remaining 3 items were modified and presented to participants in round 2 as multiple-choice items (D1, D3, and D6). Most participants (70%) voted for videoconference (eg, Zoom [Zoom Video Communications, Inc]) as the optimal delivery platform (D1). Participants (41%–100%) voted to retain the original order of the module content (D3). Approximately one-third of the participants (33%) voted to include 2 options to track PA: paper-and-pen journaling and a software app (D6). One new multiple-choice item, added in round 2, asked participants about 

the appropriate group composition for the intervention sessions

 (D1B). More than half of the participants (59%) voted for dyadic group sessions (ie, a person with MS and their care partner with 1 or 2 other dyads in each group).

Two of 5 practical/logistical items (40%) (P2 and P4) reached consensus (70% and 79%, respectively) in round 1. The remaining 3 items were modified and presented as multiple-choice items (P1, P3, and P5) in round 2. Most participants voted that an adequate amount of content was covered in each module (P1, 82%); selected 45 to 60 minutes for the duration of group sessions (P3, 41%); and chose a duration of 15 to 20 minutes for the 1-on-1 support phone calls (P5, 63%).

Nearly all participants (93%) provided qualitative feedback with their rated responses. We present a narrative summary of participants’ comments below with exemplar quotes for context.

Although participants acknowledged the value of including content about PA guidelines to help set benchmarks for PA during PAT-MS, they also acknowledged that they are not universal: “The guidelines are not conducive to everyone who has MS … these suggestions (ie, guidelines) are flexible and general, not definitive – no need to dwell on them” (clinician). Other participants noted that PAT-MS needs to support lifestyle PA alongside exercise training: “…suggest to mention more lifestyle-oriented ways of exercise, indicate other activities that can count, and assure people that everyone is different and that is OK” (person living with MS).

Several participants commended the inclusion of content related to the safety of PA. These participants highlighted the importance of such information, particularly for people with MS, who historically have been told not to engage in PA because of the belief that it can exacerbate the disease process and result in worsening of symptoms such as fatigue: “I find the perspective of exercise as dangerous still persists among clients, especially those not diagnosed recently. Fear can be a significant barrier and addressing it off the bat I think is very important” (clinician).

Participants further emphasized the importance of acknowledging the unpredictability of MS and using a strengths-based approach that highlights opportunities for personal growth and change rather than deficits and problems. This emphasis was particularly relevant to items about motivation, social support, and behavioral strategies. For instance, a participant noted: “Social support is important but as many people with MS lack a social support system and are too tired/fatigued to establish one, too much emphasis on this can be depressing” (person living with MS).

Other comments were related to the importance of ensuring that the additional resources provided in the PAT-MS manual are nationally available at subsidized rates to promote access, particularly for individuals living in small/rural areas: “Too often these services are ‘hidden’ within the community, and no one knows they are available” (person living with MS).

Although more than half of the participants voted for dyadic intervention group sessions, comments indicated a need for the PAT-MS research team to consider a hybrid format that includes a combination of individual (care partner/care recipient alone) and dyadic sessions/activities: “Possibly a blend, where some are specific to the person with MS or to the care partner and some are dyadic. This would allow for certain focus of messaging in the individual groups and then allow the collaboration and sharing between them during the dyadic sessions” (MS care partner).

The use of teleconference as a delivery platform for PAT-MS was a polarizing issue among the participants. Some participants considered teleconferencing “an accessible and realistic approach” (health care provider), stating that “teleconferencing is great for people who need the 2-way interaction” (person living with MS). Other participants were strongly opposed to the idea of teleconference, citing potential challenges regarding group engagement: “It [teleconferencing] is hellish … unsure how the teleconference will work. My experience is that the facilitator will have to ensure engagement of all parties and that can be a challenge” (person living with MS). Several participants suggested videoconferencing with a telephone dial-in option for those with limited or no internet access to promote more group interaction and social support: “Zoom does allow far better relationship building, especially with a small group” (person living with MS).

Participants commended the inclusion of 1-on-1 calls as opportunities to offer additional support and promote engagement during the intervention: “I think this is one of the best options. Having someone who knows what you are doing and can help keep track of what you’re doing is great. Being able to answer your questions and having a bond so you are comfortable to ask anything” (MS care partner). Other comments were related to the need for flexibility during these support calls: “The length of time should differ based on the needs and exhaustion felt by the person with MS or their caregiver that week” (person living with MS).

The purpose of this Delphi study was to engage persons living with MS, MS care partners, clinicians, and service providers as experts in prioritizing and refining key intervention content, delivery, and practical/logistical aspects of PAT-MS. The modified e-Delphi process enabled the PAT-MS research team to clarify any uncertainty within the existing evidence. Specifically, before the Delphi study we had already completed a staged research process

 that identified key components of PAT-MS. The Delphi process was then used to prioritize and refine the intervention content, delivery, and practical/logistical aspects. Below we situate our findings in the extant literature on PA promotion for people with neurodegenerative diseases, including MS, and their care partners.

Overall, the content recommended to include in PAT-MS corresponds with the literature about PA promotion among people affected by MS and care partners in general.

 This content includes, for example, the inclusion of PA guidelines for people with MS and adults in the general population. However, participants cautioned against having a narrow focus on exercise training alone and recommended including content on the benefits of lifestyle PA in addition to exercise. Previous researchers have echoed the importance of a strengths-based approach to health promotion that focuses on a range of enjoyable lifestyle activities that incorporate PA and exercise training for lasting health behavior change.

Other recommendations included providing high-quality information about the safety of PA and strategies for maintaining ongoing motivation; highlighting the importance of a strong social support network for PA; and emphasizing PA as a dyadic coping strategy. These recommendations have ample support in existing literature.

 The recommendation to include behavioral strategies was not surprising, and it reflects current best practice guidelines for developing health interventions.

 Similar to the present findings, people with MS

 have previously reported the need for resources to guide them to available community-based supports for PA. The present findings further emphasize that these resources need to be updated regularly, nationally available, and affordable to promote access for people with MS and their care partners.

In the literature specific to dyads with neurodegenerative diseases, there is growing popularity of dyadic interventions that involve both partners participating together in joint sessions and receiving the same intervention materials.

 Although the use of dyadic sessions reached consensus in this study, participants emphasized the need to carefully consider a hybrid format, including a combination of individual (care partner/care recipient in separate sessions) and dyadic sessions/activities. Participants’ comments highlight the importance of retaining a pragmatic balance between issues, such as addressing individual needs and ideal study designs, and time and budgetary constraints. Going forward, we now plan to include some opportunities for individual activities while retaining the overall dyadic format of PAT-MS. This decision will ensure that PAT-MS retains the dyadic intervention approach and its theoretical foundations.

Participants indicated a preference for group-based videoconference delivery. Although telephone delivery has been one of the most widely available telerehabilitation modalities,

 the current COVID-19 pandemic has increased endorsement and use of videoconferencing software (eg, Zoom) for virtual health care. Thus, this finding may reflect the current health care delivery climate, where participants may now have experience delivering and/or receiving health care online. This finding further highlights the importance of capitalizing on technological advancements that have occurred in the past few decades to promote participation in PAT-MS. Recent research indicates that more than 90% of people living with MS own at least 1 computer or a smartphone.

 Incorporating more interactive technologies into rehabilitation interventions may improve participant engagement in these interventions. Nevertheless, when capitalizing on current technological advancements in intervention development, we must consider accessible approaches that meet the needs of diverse audiences (ie, considering e-literacy, access to the internet, and personal preferences) for long-term intervention success.

Overall, these Delphi recommendations helped us think about the way that we may need to approach discussions about PA in a PAT-MS intervention. The recommendations provide key guidance for future evaluation of PAT-MS in terms of understanding the most important components to different stakeholders and the types of content/indicators to include in the intervention. The findings of this study may provide some guidance for future researchers and clinicians interested in developing and implementing dyadic interventions in MS and other neurodegenerative diseases.

This study is the first to provide recommendations for key components for a dyadic PA intervention for persons with moderate to severe MS and their care partners. Broad stakeholder involvement increases the possibility that the final intervention will be acceptable to all groups.

 In addition, the present response rate is consistent with the literature that suggests a 10% response rate for survey-based studies.

 Finally, there was a high level of participant retention between the 2 survey rounds (~85%) with the final sample size more than double the lower limit threshold of 12 participants.

 These features promote the reliability and validity of the present findings.

There are some limitations worth noting. First, there is a possibility that the views of the present participants may differ from those of stakeholders who declined participation and, therefore, may not adequately represent experts in the field of interest. To minimize this risk, we used a comprehensive recruitment process involving a database of previous participants in the present research studies, our professional networks, and a snowballing technique to identify a representative range of stakeholders to participate in the study. Nevertheless, a small percentage of MS care partners (4%; n = 5 of 149 who were emailed a recruitment letter) participated in the study. The reasons for declining to participate are unclear; however, it is possible that the method of email invitation may not have been an effective or appropriate form of recruitment for this group. It is also possible that the COVID-19 pandemic may have affected MS care partners’ ability to participate during this time (eg, additional time constraints, unpredictable changes in caregiving roles and responsibilities, etc). Furthermore, we recruited care partners for this Delphi study primarily from a sample of participants who had recently completed an online survey as part of a longitudinal study examining resilience among MS care partners with our research group. It is possible that these care partners may have perceived taking part in another study, particularly during the COVID-19 lockdown, as an additional burden and chose to opt out of the present study. Another possibility is that few care partners are interested in this type of intervention; however, previous needs assessment work

 established the critical need to develop a dyadic PA intervention that supports joint engagement of people with moderate to severe MS disability and their care partners. Therefore, lack of interest from MS care partners in PAT-MS is unlikely to be a key issue. Nevertheless, several evidence-based strategies

 are included in the PAT-MS intervention design to ensure “buy-in” from care partners and to maximize recruitment and retention.

Prioritizing Components of a Dyadic Physical Activity Intervention for People With Moderate to Severe Multiple Sclerosis and Their Care Partners: A Modified e-Delphi Study

Ashley N. Newton, MHA, MAcc, CPA

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