Discussion

Our study used surveys and semi-structured interviews to characterize the population of MSA caregivers and understand many of the day-to-day challenges, concerns, and positive aspects that shaped their experiences. It also identified ways the health care system and clinicians can support MSA caregivers.

Caregiver burden has been a focus of the literature on caregiving ^{Reference Hoffmann and Mitchell25} and was assessed in our study. Caregiver burden consists of objective caregiver burden (the tasks the caregiver is responsible for) and subjective caregiver burden (the emotional demands of caregiving). ^{Reference Hoffmann and Mitchell25} In terms of objective burden, participants outlined the many activities required to support the person with MSA. These roles were myriad, including physical tasks (assisting with activities of daily living), administrative activities (such as scheduling appointments), emotional support, and advocating for the PwMSA within the health care system (Table 7). Caregiving activities performed by the MSA caregivers in our study were consistent with activities performed by caregivers in other conditions. ²⁴ Since participants with MSA varied in their symptoms and the length in time since diagnosis, the objective burden of activities required to assist PwMSA varied from “not very much work” to a long list of tasks every day accounting for a significant amount of time. Subjective burden was assessed using a modified 24-item Zarit Burden Interview, and scores similarly ranged from mild burden to severe burden.

Through statistical analysis, we assessed whether caregiver burden was associated with other measured variables. Caregivers had mild anxiety on average, and caregiver anxiety correlated with caregiver burden. Caregivers of people with Parkinson’s disease (PD) have a similar association of higher anxiety with higher caregiver burden. ^{Reference Macchi, Koljack and Miyasaki9,Reference Martinez-Martin, Forjaz and Frades-Payo26,Reference Torny, Videaud, Chatainier, Tarrade, Meissner and Couratier27} In PD caregivers, there is also an association between higher depression scores and higher caregiver burden that was not found among our caregiver participants. ^{Reference Macchi, Koljack and Miyasaki9,Reference Martinez-Martin, Forjaz and Frades-Payo26,Reference Torny, Videaud, Chatainier, Tarrade, Meissner and Couratier27}

Patient quality of life is a factor that has been found to correlate with caregiver burden in studies in PD, ^{Reference Macchi, Koljack and Miyasaki9,Reference Torny, Videaud, Chatainier, Tarrade, Meissner and Couratier27} dementia, ^{Reference Farina, Page and Daley28} and amyotrophic lateral sclerosis. ^{Reference Thomas, Warrier and Sadasivan29,Reference Geng, Ou and Miao30} Our results did not find a link between the QoL of PwMSA and caregiver burden; however, our sample size was small and unlikely to find such an association. Disease severity and symptoms are other patient factors that have been associated with caregiver burden, ^{Reference Torny, Videaud, Chatainier, Tarrade, Meissner and Couratier27,Reference Smith, Perrin, Tyler, Lageman and Villaseñor31} but quantitative measures of disease severity were too homogenous in our study to assess this.

Though caregivers in our study were on average not depressed, we found a correlation between patient and caregiver depression. This correlation has previously been reported in studies of caregivers of people with cancer ^{Reference Nipp, El-Jawahri and Fishbein32} and patients in primary care clinics. ^{Reference Jeglic, Pepper, Ryabchenko, Griffith, Miller and Johnson33} Whether the relationship between patient and caregiver depression symptoms is causal, and the direction of causation, cannot be determined by our results.

Qualitative results identified safety of the PwMSA as a primary concern of caregivers, similar to previous research in PD ^{Reference Hand, Oates, Gray and Walker34} and Parkinson-plus syndromes. ^{Reference Wiblin, Durcan, Lee and Brittain8} In our study, home care services were identified as essential to keeping PwMSA safe at home. Our participants had access to publicly funded home care based on their assessed needs without a time limit on the services. Caregivers were thankful for home care that allowed them to work outside the home, run errands, and participate in hobbies. However, despite access to publicly funded home care and health care navigation services, participants still encountered challenges with home care. These included limits to hours and the lack of flexibility of home care schedules to address day-to-day variations in care requirements. Similar difficulties were noted across the range of household incomes in our study. As a way to gain more control over their home care services, some PwMSA and families in our study opted into a publicly funded self-managed home care program. ³⁵ In this model, patients are assessed and given a budget to work with. Hiring, training, and scheduling employees is then the responsibility of the patient or family. ³⁵ While some participants preferred self-managed care for the added control it gave them over scheduling and hiring, others chose not participate in the program due to the management responsibilities required.

Expansion of publicly funded home care services would be helpful to PwMSA and their caregivers and would likely be helpful in other chronic health conditions. Home care is also beneficial from a health system perspective, as it is cost-effective compared to the alternatives of hospitalization or admission to a skilled nursing facility. ^{Reference VanderBent and Kuchta36} The limits of home care expansion that would still be cost-effective are not clear; however, in general, home care investment has not kept pace with the aging population. ^{Reference VanderBent and Kuchta36} Our study did not assess for a correlation between home care usage and caregiver burden or anxiety and depression symptoms. The effects of home care on caregiver burden and mood are inconsistent in the literature. One study from South Korea found no effect of home care usage on caregiver burden or satisfaction. ^{Reference Kim and Yeom37} A study in Taiwan found home care buffered the effects of IADLs (instrumental activities of daily living), but not the effects of ADLs or care recipient cognitive impairment, on caregiver depression. ^{Reference Chang38} In both studies, underuse of home care services may have been a factor. ^{Reference Kim and Yeom37,Reference Chang38}

Another important way the health care system supports MSA caregivers is by providing education on the illness as well as on caregiving skills. Caregivers in this study did not receive standardized education, and the information they received from health professionals varied. Standardized education programs for caregivers covering topics such as disease education, nursing skills, and psychosocial education show promise as an intervention for improving caregiver and patient outcomes. ^{Reference Sörensen, Pinquart and Duberstein39} A nurse-led education session for caregivers of people who had recently had a stroke was associated with reduced caregiver strain at time of discharge; ^{Reference Gaio, Buoite Stella, Furlanis, Naccarato, Stefi and Manganotti40} however, another study of in-hospital education for stroke caregivers found no significant difference in patient outcomes. ^{Reference Pedapati, Bhatia and Shakywar41} A hospital-based stroke education program that incorporated an in-home follow-up session resulted in improved caregiver and patient QoL and mood at 3 and 12 months as well as reduced health care costs. ^{Reference Kalra, Evans and Perez42} In PD, a 9-week outpatient educational program for caregivers which incorporated psychoeducational sessions, when compared to an educational program without these sessions, showed improvements for both patients and caregivers in psychosocial adjustment and for caregivers in coping skills. ^{Reference Navarta-Sánchez, Ambrosio, Portillo, Ursúa, Senosiain and Riverol43} A similar 8-week program showed improvement in patient and caregiver mood and decreased psychosocial problems for caregivers, though there was a large variation in scores within intervention and control groups. ^{Reference AʼCampo, Wekking, Spliethoff-Kamminga, Le Cessie and Roos44}

Our caregiver participants recognized the importance of maintaining their own health to continue in their caregiving role. Caregiver health is particularly relevant when the care recipient still lives at home, and may influence admission to long-term care. In a survey of caregivers of patients with dementia in long-term care, 49% of family caregivers cited their own health as a factor in the decision to move their relative into long-term care. ^{Reference Buhr, Kuchibhatla and Clipp45}

The health effects of providing care for a family member are somewhat of a paradox. Despite higher levels of stress and depressive symptoms, in several studies, caregivers have had reduced mortality rates compared to similar non-caregivers. ^{Reference Roth, Brown, Rhodes and Haley46,Reference Mehri, Kinney, Brown and Rajabi Rostami47} These findings have not been consistent, and the influence of duration and intensity of caregiving on mortality are not well studied. ^{Reference Mehri, Kinney, Brown and Rajabi Rostami47} One possible explanation for caregivers’ reduced mortality is the “healthy caregiver hypothesis,” which states that healthy individuals are more likely to become caregivers and continue in that role. ^{Reference Mehri, Kinney, Brown and Rajabi Rostami47} A competing hypothesis is that helping others provides a psychological protective effect against the negative health effects of stress. ^{Reference Roth, Brown, Rhodes and Haley46,Reference Mehri, Kinney, Brown and Rajabi Rostami47} Caregiver participants in our study did report psychological benefits from caregiving, including satisfaction from knowing they were doing everything possible for the PwMSA. Still, they reported risks to their own health due to their role, including risk of injury. Caregiving can be physically demanding, and a survey of Canadian caregivers found that 8% of spouse caregivers and 6% of those caring for their parents have sustained injuries from their caregiving work. ^{Reference Turcotte48} Another survey of caregivers who helped their family member with activities of daily living such as dressing, bathing, and transfers found 94% experienced musculoskeletal discomfort in the preceding 4 weeks, most commonly low back pain. ^{Reference Darragh, Sommerich, Lavender, Tanner, Vogel and Campo49} Transfers were the most physically demanding task. ^{Reference Darragh, Sommerich, Lavender, Tanner, Vogel and Campo49} Even in early stages of MSA, our caregiver participants described helping patients stand up and catching them from near falls. More advanced PwMSA required help to transfer in all situations. Caregivers were not necessarily trained to safely perform these tasks. Physiotherapist or occupational therapist education for all caregivers on transfers and falls would be helpful. ^{Reference Kalra, Evans and Perez42}

Two factors which make MSA a uniquely challenging illness are its rapid progression and the prominence of symptoms affecting communication. ^{1,Reference Wiblin, Lee and Burn4,Reference Wiblin, Durcan, Lee and Brittain8} The rapid progression of MSA was a stressor for caregivers. In addition to the emotional toll from watching the PwMSA’s symptoms worsen, some participants expressed grief or frustration over the relentless progression despite their best efforts to help. Worries about future disease progression were common and have previously been found to affect caregiver QoL in Alzheimer’s disease. ^{Reference Farina, Page and Daley28} Caregiving activities escalated as illness severity increased, which likely impacted caregivers’ health. Transition to heavy caregiving, defined as helping with activities of daily living (ADLs) has been associated with increased depression symptoms and poorer health behaviors compared with non-caregivers and those who transition to moderate caregiving, defined as helping only with IADLs. ^{Reference Burton, Zdaniuk, Schulz, Jackson and Hirsch50}

Dysarthria is a nearly universal symptom of MSA and was present in 100% of PwMSA in one study. ^{Reference Rusz, Tykalová, Salerno, Bancone, Scarpelli and Pellecchia51} Speech often becomes unintelligible even to family members. Since cognitive concerns occur late in MSA, ^{Reference Krismer and Wenning3} this leaves the PwMSA unable to make their wishes known or be “heard," yet still have much to say. For caregiver participants, unintelligible speech was a source of loss and frustration. Inability to discern the PwMSA’s wishes may have led caregivers to doubt their own effectiveness and reduced their satisfaction with caregiving, as feelings of competence are associated with satisfaction with the caregiver role. ^{Reference Quinn, Clare and Woods52} Technologies such as voice augmentation and communication boards were trialed by several participants but were often useful for a brief window of time due to worsening dysarthria and motor dysfunction. Communication difficulties, as well as the dependence of the PwMSA on the caregiver, caused profound changes in some participants’ marital relationships. This finding is in contrast to a previous qualitative study in MSA and PSP, in which the quality of participants’ marital relationships was unchanged despite communication symptoms. ^{Reference Wiblin, Durcan, Lee and Brittain8} Some caregivers in our study described a gradual shift in roles from being a spouse to being a caregiver. Participants were often unprepared for this shift, and information or counseling on how to maintain a positive relationship through changing roles may be helpful. ^{Reference Martire, Schulz, Helgeson, Small and Saghafi53}

As reflected in participants’ comments, caregiving was a multifaceted experience – neither all negative nor all positive. Caregivers identified advantages to caregiving, most importantly being able to spend more time with their loved one. Several supportive factors were identified. Support from family was valued, even when other family members were not directly involved in caring for the person with MSA. Satisfaction with social support networks has previously been linked to a positive view of caregiving, ^{Reference Lee and Choi54} and social isolation in caregivers has been linked to caregiver burden and depression. ^{Reference Zhong, Wang and Nicholas55} Caregivers who do not feel socially supported may particularly benefit from interventions such as respite care, support groups, or psychotherapy. ^{Reference Sörensen, Pinquart and Duberstein39} Aside from social support, supportive factors identified by caregivers in this study included being able to access health services in a timely fashion when needed and having someone to talk to about their experiences.

This study has several strengths as well as several limitations. Its mixed methods design allowed a fuller picture of caregivers’ experiences than either quantitative or qualitative methods alone. However, the convenience sampling recruitment strategy meant that several demographics were absent from this study. All participating PwMSA had the parkinsonian subtype of MSA (MSA-P), except for one patient who had a mixed type. No patient had the cerebellar subtype of MSA (MSA-C). This reflects underlying prevalence: MSA-P is more common than MSA-C, and in Western countries, MSA-P accounts for up to 80% of MSA. ^{Reference Krismer and Wenning3} MSA-C is most common in Asian ethnicities ^{1,Reference Krismer and Wenning3} and progression of disability is slower than in MSA-P; ^{Reference Watanabe, Saito and Terao56} therefore, the experience of MSA-C caregivers might differ in areas such as objective caregiver burden and cultural values related to caregiving. Immigrant or refugee patients and caregivers were not represented in the study; they often face additional barriers to accessing health care and may lack a social network to rely on. ^{Reference Williams, Sethi and Duggleby57} Rural caregivers were underrepresented, and studies have found they have more financial barriers ^{Reference Bouldin, Shaull, Andresen, Edwards and McGuire58} and lower self-rated health status ^{Reference Sanford and Townsend-Rocchiccioli59} than urban caregivers. Because recruitment took place at a specialized movement disorders clinic, we did not include PwMSA without access to a specialized movement disorders clinic, who likely have higher unmet needs.

Our results suggest several areas where the health care system and individual clinicians can assist MSA caregivers. These include providing more information on MSA, ensuring training on caregiving skills such as transfers and how to assist a person who has fallen, providing information and counseling for spouse caregivers experiencing illness-related changes in their marriage, and expanding publicly funded home care services. Caregivers are an essential part of the health care team, and more can be done to support them in their important role.