Abstract
Introduction:
Essential tremor (ET) is a common neurological disorder associated with functional impairment. Emerging evidence shows that some ET caregivers experience burden, but the unique interpersonal aspects of caregiving in the context of ET have not been fully examined.
Research design:
Open-ended questions were administered to 98 ET care-recipient–caregiver dyads. Responses were analyzed using conventional content analysis.
Results:
The unique visibility of disability and feelings of embarrassment that occur with ET prompts caregivers to be highly attuned to care-recipient emotions. Providing companionship, promoting independence, and reducing embarrassment are three themes we found that describe the ET caregiving experience.
Discussion:
Caregiving in ET often goes beyond aiding with activities of daily living; it has a significant emotional component especially in relation to independence and embarrassment. Future studies on caregiving in ET and conditions in which disability is visible should consider using a definition of caregiving that includes emotional caregiving.
Essential tremor (ET) is the most common movement disorder in older adults, with worldwide prevalence in those age 65 and older estimated at 4.6% (Louis & Ferreira, 2010). ET is a progressive neurodegenerative disease of heterogeneous origin characterized by tremor of the hands, head, voice, and/or other areas of the body (Benito-Leon & Louis, 2007; Louis & Ferreira, 2010; Louis & Ottman, 2003). These tremors can cause functional disability in activities such as writing, eating, and drinking (Louis et al., 2001a). Additional symptoms such as cognitive impairment, impaired balance, and sleep dysregulation can result in added challenges for those with ET (Louis, 2016). For many, these impairments affect employment and participation in social or community activities (Tröster, Pahwa, Fields, Tanner, & Lyons, 2005). Existing medications are limited in effectiveness (Louis, Rios, & Henchcliffe, 2010; Zesiewicz et al., 2011). While cognitive and motor impairments in ET are typically not severe enough to require constant care or paid caregivers, individuals with ET may rely on their family members and friends to act as informal caregivers (Kellner et al., 2017; Morgan et al., 2017). These informal caregivers may assist individuals with ET with activities of daily living such as cooking, eating (e.g., cutting food), and dressing as well as with tasks such as writing checks, completing forms, or using a computer (Kellner et al., 2017; Morgan et al., 2017). For individuals with severe voice tremor, caregivers may act as interpreters to help them communicate with others. Although most caregivers provide few hours of direct care per week, some provide >25 hr of care per week and/or report high levels of burden (Morgan et al., 2017).
While there is considerable literature describing the caregiving relationship in other conditions such as dementia, cancer, and stroke (Adelman, Tmanova, Delgado, Dion, & Lachs, 2014), ET presents unique challenges. ET is often highly visible and a large proportion of those with ET experience embarrassment related to their tremor or even social phobia (Louis & Rios, 2009; Schneier, Barnes, Albert, & Louis, 2001; Traub, Gerbin, Mullaney, & Louis, 2010). Tasks that require assistance in public, such as completing forms or eating, could result in heightened embarrassment for the individual with ET. Recent research also shows that caregivers’ perception of their care-recipient’s embarrassment is associated with increased caregiver burden (Kellner et al., 2017; Monin et al., 2017; Morgan et al., 2017). Another study showed that caregivers’ perceptions of suffering in their care-recipients was associated with caregiver depression (Monin et al., 2017; Morgan et al., 2017). In other words, there is evidence to suggest that ET caregivers are attuned to and affected by the care-recipient’s emotional states.
While studies on caregiving in other conditions have occasionally noted that providing emotional support is an aspect of the caregiving relationship, embarrassment is rarely mentioned apart from being a potential factor contributing to caregiver burden (Van Exel, Koopmanschap, van den Berg, Brouwer, & Van den Bos, 2005; Wright & Aquilino, 1998). Given the general lack of research on emotional support in caregiving, the importance of embarrassment in ET, and the scarcity of ET related caregiving studies, we conducted the first qualitative study exploring the interpersonal processes within the ET caregiving relationship. Qualitative analysis seeks to understand problems from the perspectives and experiences of the population under study and can provide a broader and richer view of an issue than quantitative data (Strauss & Corbin, 1998). This research aims to provide an overview of the caregiving relationship in ET from both the caregiver and care-recipient perspective. This effort could provide a richer understanding of the emotional side of caregiving, which future researchers could incorporate into their definitions of caregiving. Also, by providing a more accurate definition of family caregiving in ET, advocates and policy makers may better argue for health-care benefits that meet these families’ needs. Furthermore, this research could have implications in the clinic regarding caregiver education and support, especially for conditions where the care-recipient is likely to experience embarrassment.
Research Design and Methods
Parent Study
Participants were previously recruited from a 230-participant, longitudinal study of cognitive function in ET which began in 2014 (Clinical Pathological Study of Cognitive Impairment in Essential Tremor [COGNET], NINDS R01NS086736) approved by the Yale University institutional review board. Each participant signed written informed consent. Participants must have had a diagnosis of ET, been willing to undergo comprehensive cognitive testing, and agreed to postmortem brain donation.
Participants were evaluated in a series of home visits by trained study personnel (SK, TEKC) who administered a battery of neuropsychological tests in two 2-hr sessions from January 2016 to February 2018 (Collins et al., 2017). Participants were evaluated on tremor severity and tremor disability by a senior movement disorders neurologist (EDL; Louis et al., 2000; Louis, Gerbin, & Mullaney, 2010; Louis et al., 1997). Semi-structured interviews with informants were used to calculate the 8-Item Lawton Instrumental Activities of Daily Living Scale score, which asks about ability to independently perform activities such as doing laundry and preparing food (Graf, 2008) and the Clinical Dementia Rating (Morris, 1993; Woolf et al., 2016).
Substudy Recruitment
The institutional review board of Yale University approved this substudy and recruitment occurred between November 2017 and March 2018. Subjects were recruited for this substudy by trained study personnel (MAZ), who contacted and consented COGNET participants by telephone. After giving verbal consent over the phone, subjects were invited to complete an online survey about their quality of life and people with whom they have close relationships. Interested individuals were requested to identify a caregiver, that is, someone who helped them with daily tasks, provided emotional support, and/or could provide insight into their well-being, henceforth referred to as “caregivers.” MAZ then contacted, consented, and invited caregivers to complete their portion of the survey. Subjects from the COGNET cohort were contacted in alphabetical order (no participants being biologically related) until 100 individuals with ET (henceforth “care-recipients”) were enrolled along with their corresponding caregiver, totaling 100 dyads. Quotes included in the paper are referenced as either care-recipient (CR) or caregiver (CG) and a randomly assigned number corresponding to the dyad (i.e., CR 59).
Survey Development and Distribution
The surveys distributed to care-recipients and their caregivers included a series of open-ended questions asking each one about their experiences with ET and how it has affected their relationship (Tables 1 and 2). Open-ended questions were designed based on aspects of the ET caregiving relationship that may have particular importance based on previous findings. Previous studies have found that those with ET experience embarrassment at high rates and that ET caregivers experience increased burden when they perceive more embarrassment in care-recipients and that they experience increased depression when they perceive more suffering (Kellner et al., 2017; Monin et al., 2017). This, combined with the significant public component of embarrassment, guided our focus on caregiving situations both in public and private that may cause strain or distress for both the caregiver and care-recipient. These questions were also informed by expert opinion from both a senior movement disorders neurologist with a special interest in tremor (EDL) and a research psychologist specializing in older adult caregiving (JKM). We asked similar questions to both the care-recipient and caregiver, allowing comparison within dyads as well between dyads.
Care-Recipient Open-Ended Questions.
Caregiver Open-Ended Questions.
Open-ended questions were further supplemented by having respondents complete several psychometric scales. Care-recipients were asked to complete a modified Essential Tremor Embarrassment Assessment (ETEA; Traub et al., 2010). Meanwhile, caregivers were asked to complete the Essential Tremor Enfeeblement Scale (ETES; Trujillo Diaz et al., 2019), the perceived ETEA (pETEA; Kellner et al., 2017), and the Zarit Burden Inventory–Short Form (ZBI-12; Zarit, Reever, & Bach-Peterson, 1980).
Surveys were distributed by e-mail to individuals with ET and to their caregivers using the Yale Qualtrics online survey platform. Participants without a viable e-mail address or who were otherwise unable to answer the online survey on their own were instead interviewed by MAZ over the telephone. Due to the sensitive nature of the questions, participants were instructed to answer the survey independently from and without the influence of their counterpart in the dyad.
Analysis
Once 100 care-recipients and caregivers had completed the survey, responses were analyzed in two ways. First, qualitative responses were evaluated for agreement with the following statements: (1) Caregiver self-identifies as a “caregiver,” (2) caregiver helps care-recipient with daily tasks, (3) caregiver aids with any task due to ET, (4) Caregiver agrees with definition of caregiving provided, and (5) care-recipient agrees with definition of caregiving provided. Responses were rated by three research assistants (RH, TEKC, and DTD) as 0 (disagree), 1 (agree), or 2 (response was insufficient to determine agreement), and discrepancies were resolved by choosing the majority rating.
Second, responses were analyzed using conventional content analysis (Hsieh & Shannon, 2005) with ATLAS.ti (version 8.3.1) as a data management tool. This method of qualitative analysis is best suited for studies describing a phenomenon with limited previous literature. While there is significant literature on caregiving in other disease and disability contexts, the lack of research relevant to the ET caregiving experience informed our inductive approach. Conventional content analysis draws from the naturalistic paradigm, which emphasizes the importance of context and an individual’s experience (Given, 2008). As such, responses were read primarily in pairs so that each dyad member’s responses could provide context to the other. Responses were first read in their entirety several times to gain an overall sense of the data. Then, responses were coded at a granular level, coding sentence-by-sentence and repeatedly questioning what the data were indicating. Memos were created for both whole interviews and especially meaningful quotes. Through this process, categories of codes were developed. Codes were organized based on conceptual relationships, and clusters of codes formed the basis for themes. Codes and developing themes were constantly compared to the responses using Boeije’s five steps (within the same interview, between interviews of same group, between interviews of different groups, within dyads, and comparing dyads, not necessarily in that order; Boeije, 2002) to ensure that they accurately reflected the experiences of the respondents. For example, instances of the code “normalization,” which eventually was subsumed under the theme of reducing embarrassment, were checked within individual interviews, between interviews of the same group (i.e., spousal dyads), between groups (i.e., between spousal dyads and child–parent dyads), and between dyads to ensure the meaning was consistent and accurately reflected the responses. This primary analysis was performed by a researcher previously trained in content analysis (RH). Developing themes were discussed with the other investigators at several points throughout the analysis, and the final themes are discussed below.
Results
Sample Characteristics
We initially recruited 100 care-recipient/caregiver dyads; however, one dyad was excluded due to incomplete responses and another dyad was excluded because the caregiver who responded was a formal (paid) caregiver. Demographic data from 98 complete dyads are shown in Table 3. In general, more than 50% of the care-recipients and caregivers were female, and both groups skewed White and highly educated. Most caregivers were spouses and lived with care-recipients.
Sample Characteristics.
Note. Frequencies presented with percentages. Normally distributed variables as assessed by Kolmogorov–Smirnov tests are presented with means and standard deviations; nonnormally distributed variables are presented with medians and interquartile range (IQR). MoCA = Montreal Cognitive Assessment; IADL = instrumental activities of daily living; ETEA = Essential Tremor Embarrassment Assessment; ETES = Essential Tremor Enfeeblement Scale; pETEA = Perceived Essential Tremor Embarrassment Assessment; ZBI-12 = Zarit Burden Inventory–Short Form.
Among care-recipients, 15 had questionable dementia (CDR = 0.5), two had mild dementia (CDR = 1), and three had moderate dementia (CDR = 2). Although tremor evaluated by a tremor disability questionnaire and total tremor score from a videotaped evaluation was generally more severe than tremor evaluated in previous ET community-based studies (Louis, Barnes, et al., 2001; Louis, Ford, et al., 2001), our cohort remained highly functional, with a median instrumental activities of daily living (IADL) score of 8 (i.e., requiring no assistance with the given instrumental activities).
Caregiving Definition and the “Caring Giver”
Caregivers and care-recipients were each given a typical definition of caregiving based on assistance with IADLs (Tables 1 and 2; Schulz & Beach, 1999) and asked whether they agreed with the definition and whether they saw themselves or their loved one as a caregiver. Responses were assessed for agreement with several statements of interest pertaining to caregiving (Table 4). Despite 80 of 98 dyads mentioning providing or receiving help with at least one caregiving task, only 28 of 98 caregivers said that they saw themselves as a caregiver. Interestingly, dyad members did not always agree with identification as a caregiver or care-recipient. For example, one spouse responded that she cuts food, writes, performs small repairs, assists with dressing, and does the driving for her husband who has ET. The husband agreed that she was a caregiver, but she responded that she only sees herself as a “loving wife.”
Rated Agreement With Statements.
a N = 98. bCaregiving definition provided: “helping someone with at least one of the following activities due to a chronic condition: bathing, dressing, toileting, transferring bed/chair, eating, using the phone, shopping, preparing meals, performing housework, driving or using public transportation, managing medication, and managing finances.”
Looking to the literature, a review article of dementia caregiving states: “Although no standard definition of family caregiving exists, there is general consensus that it involves the provision of extraordinary care, exceeding the bounds of what is normative or usual in family relationships” (Schulz & Martire, 2004). Considering the relatively mild nature of functional impairment in many ET cases, most people who provide care would not be considered a caregiver under this definition. However, many with ET do require assistance in tasks that require fine-motor skills. Tasks that were specifically endorsed by our dyads as receiving or providing assistance with included writing (e.g., signing or writing checks, completing forms), meals (e.g., cutting food, serving plates at buffets, pouring drinks), emotional support (e.g., providing general emotional support, helping when upset), personal care (e.g., dressing, bathing), miscellaneous fine-motor tasks (e.g., getting cards out of wallet, home maintenance tasks such as wiring), and management (e.g., of finances, living situation). Although many in our study did not self-identify as a caregiver, previous literature on caregiving in ET suggest that aiding with these tasks is associated with caregiver burden (Morgan et al., 2017).
From our participants’ responses, it became apparent “emotional caregiving” was a particularly important component of being a caregiver otherwise missing from the definition we provided. Emotional caregiving was not only a specific task that participants reported receiving or providing assistance with but also a way of providing care itself. The three themes outlined below, providing companionship, promoting independence, and reducing embarrassment, were developed through coding and recoding of the responses. These three themes can be understood not only as ways of providing emotional care but also as ways of providing all types of care in an emotionally conscientious manner. As one care-recipient described his wife, a caregiver who provides emotional support in these ways is “more of a caring giver, than a caregiver” (CR 73).
Providing Companionship
While an IADL-based definition of caregiving may be the most objective, participants noted that providing emotional support was an important component that was missing. For some, providing emotional support and companionship constituted the majority of the caregiving relationship. For example, one daughter noted that while her mother’s partner has a “more day-to-day role” in helping with tasks because they live together, her contributions at this point are aiding with bill payment and providing emotional support over the phone (CG 13). For other individuals with tremor not severe enough to warrant assistance with physical tasks, providing companionship can be a way of caregiving. One care-recipient noted that while her tremor was not severe enough to require help with instrumental tasks, her daughter still cared for her by providing companionship. My daughter is someone I can talk to and share with her my feelings of embarrassment that I have because of my ET […] I think there is a moral support that we need more than just help with activities. I need my daughter to want to do activities with me like going to a movie or out to eat. This is what she helps me with at this time. She is someone who knows my situation and isn’t embarrassed to be with me. (CR 99)
Social avoidance due to tremor can exacerbate social isolation and loneliness, which are health risks for the elderly (Steptoe, Shankar, Demakakos, & Wardle, 2013). Providing companionship can combat isolation associated with ET, as one caregiver described while responding to the caregiving definition provided: Good definition, however there needs to be an element of companionship. Chronic illnesses tend to be isolating. A good caregiver addresses that, both with positive interaction and encouraging the ill one to engage socially. (CG 116) My friends and I used to go out to eat often but that is almost impossible now. They take me to the doctor and the retina specialist and the dentist when necessary but it’s so hard and embarrassing for me. (CR 54)
Especially for spouses, providing companionship also encompasses being a supportive partner in facing health challenges. The “communal coping” framework of Helgeson, Jakubiak, Van Vleet, and Zajdel (2018) describes an attitude about a disease based on an understanding that the illness is shared between both members, which positively affects outcomes. One caregiver wrote that helping with her husband’s tremor has “only made us stronger and closer and determined to be in control, not the disease process ruling us” (CG 72). Supportive companions can share the disease burden both physically and emotionally.
This emotional support also includes helping the care-recipient with frustration related to tremor. One caregiver wrote: “[The] hardest part is convincing mother she cannot when she always talks like she can do everything” (CG 40). Where physical ability ends, emotional caregiving steps in to help soothe frustration and adapt to the evolving disease.
Promoting Independence
As opposed to the direct emotional caregiving of providing companionship, promoting independence is a method of doing care. Many people with ET are still able to do tasks if necessary, but it can take longer or be more frustrating. This was a factor in many caregivers’ “no” responses to self-identification as a caregiver. As one caregiver put it: All of those [tasks] are still done with both of us talking about it and doing it. It has not come to the point where I HAVE to do something for him. He tries and if he needs help then I will stick in my 2 cents. (CG 104)
Caregivers noted that it can be challenging when care-recipients do not accept help with tasks. One caregiver said, “I think he gets frustrated that I do certain things for him, if he doesn’t ask me to do it. He thinks I baby him but for me it’s easier to help him” (CG 73). On the other hand, care-recipients noted that it can be frustrating that caregivers are able to do tasks so much more easily and may presume that they want help. One care-recipient responded, “It bothers me that [my wife] feels it’s probably better if she does certain tasks for me” (CR 117). While it might be easier for the caregiver to cut food or write out a check, some care-recipients value independence above efficiency. In the literature, higher “independence centrality” (value of independence) has been associated with more depressive symptoms (Martire, Stephens, & Schulz, 2011; Monin, Schulz, Martire, Connelly, & Czaja, 2014). For care-recipients with ET who place high value on independence, it may be especially important for caregivers to be cognizant of the way in which they provide care.
Dyads mentioned different strategies for navigating independence in the caregiving relationship. Some caregivers only help if specifically asked, even if their loved one is struggling with a task, as one described: “I only help when it is necessary and she asks me to cut something on her plate or pick up something from the floor” (CG 177). This puts control of the situation in the hands of the care-recipient and lets them decide at what point they want the caregiver to step in. However, it also requires the care-recipient to make their needs known, which can be difficult or embarrassing.
To compromise, some caregivers offer help but make sure not to presume or be offended if the care-recipient refuses: The tasks I perform for my mother are not in themselves challenging but the challenge is helping her keep her dignity when needing assistance with remedial tasks […] however, I never assume she wants help. She may ask and/or I may offer—sometimes she wants help and sometimes she doesn’t. (CG 133)
This balance is likely easier to establish among dyads who spend a significant amount of time together. The caregiver can learn when the care-recipient is likely to need help and in what situations they prefer assistance and in doing so, the pair can find a way to help with tasks that considers ability, independence, and efficiency. In a condition characterized by a lack of control, promoting independence is a way that a “caring giver” can give some of that control back.
Reducing Embarrassment
Another way of providing emotional care that emerged from responses was reducing embarrassment. Those with ET often experience embarrassment related to their tremor (Kellner et al., 2017; Louis & Rios, 2009). Among respondents in this study, 51% of participants agreed or strongly agreed that “my tremor is embarrassing to me.” While many situations that provoke embarrassment are unavoidable, caregivers can adapt to provide care in more discreet ways.
Many respondents noted that they aid their care-recipients in ways that are subtle or that eliminate the need for care altogether. One caregiver noted that for her, the caregiving definition “also includes foreseeing and/or anticipating their needs, even before they realize it” (CG 67). By preempting the need for care, caregivers circumvent the need to help in a potentially embarrassing situation. To prevent embarrassment related to spilling while drinking, one caregiver noted that she did the following: If she is thirsty, I will get a bottle of water for her, open it and take a large drink then I will hand it to her. If it is too full, she will spill it. I don’t even ask, I just do it without drawing attention to it. (CG 119)
For some dyads, this becomes a team effort. One spouse noted that “if we are in a situation where, for example, we would need to carry a cup of coffee, I just do that and he carries something he is comfortable doing” (CG 126). In tasks that cannot be completely circumvented, some respondents noted they make sure to help in a subtle manner. One caregiver said: I think my mom feels embarrassed when she needs help zipping her coat or has trouble manipulating food utensils. When I’m with her out of the house, I try to help her on the down low. (CG 144)
For caregivers who provide assistance in public, it is important to be aware of the care-recipients’ emotions in order to best reduce their embarrassment. Mindful strategies like those discussed by our respondents can help reduce embarrassment in situations that can arise from tremor.
Discussion
From the responses of the 98 dyads, it is clear that caregiving in ET goes beyond simply aiding with IADLs. Although some ET caregivers spend a significant amount of time caregiving (Morgan et al., 2017), our study found that most aid with only a few tasks and do not identify with an IADL-based definition of caregiving. Even among dyads who provide little to no assistance with IADLs, emotional caregiving emerged as an important component of the caregiving relationship. Part of emotional caregiving was providing direct emotional support or companionship, but it also included providing care in an emotionally cognizant way through promoting independence and reducing embarrassment.
While these themes are likely components of caregiving in other conditions, the literature is sparse. One related concept is “affective support,” defined as emotionally based (as opposed to instrumental) care. In a study of adult siblings of those with traumatic brain injury, affective support was measured by the presence of 3 items: “checking in,” providing companionship, and providing general support and encouragement (Degeneffe & Burcham, 2008). A few other studies echo that emotional support is a component of caregiving. One qualitative study on caregivers assisting with aid-in-dying found that emotional support was a major theme of caregiving (Buchbinder, Ojo, Knio, & Brassfield, 2018), and another study noted that “being an emotional supporter” was a component of caregiving in multiple sclerosis (Gafari, Khoshknab, Nourozi, & Mohamadi, 2017). While these concepts are closely tied with the “providing companionship” theme, emotional caregiving as described by our study participants also encompassed providing instrumental care in a way that is mindful of the care-recipient’s emotions.
While autonomy in decision-making is a well-studied principle of medical ethics and independence is a desired outcome of care for elderly or individuals with disabilities (Beauchamp & Childress, 2001; Jogé Boumans, van Boekel, Baan, & Luijkx, 2018), there is little in the literature that has described promoting independence as a way of being a caregiver. To our knowledge, reducing embarrassment is a theme that has not yet been described as a way of providing care. However, the themes of promoting independence and reducing embarrassment may have specific significance in ET, given the common occurrence of feeling embarrassment as a result of tremors (Kellner et al., 2017; Louis & Rios, 2009). These two themes could provide insight into caregiving dynamics in conditions that similarly feature visible disabilities or impairments that may cause embarrassment, such as spinal cord injury, tremor-predominant Parkinson’s disease, or even arthritis.
Limitations
Our study had several limitations. Care-recipients were part of a cohort study of highly motivated older individuals, which may not be representative of the entire ET population. Additionally, most participants responded via online survey, which does not allow the researcher to get clarification or more information from participants, as in an in-person interview. Findings were not tested via respondent validation, and coding was conducted only by one researcher, though themes were validated through constant comparison and discussion with the other investigators.
Conclusion
Future studies on caregiving in ET should consider employing a definition of caregiving that includes emotional caregiving, with particular attention to the role of embarrassment. These findings may also be useful in other caregiving contexts with similar challenges to the maintenance of patient autonomy. While research on caregiving has largely used a definition based on instrumental support, emotional caregiving is an important part of a successful relationship. Expanding the definition in future research could provide insight into sources of caregiver burden, provide a new avenue to target interventions, and build a foundation for discussion of the caregiving experience in future qualitative studies. A more accurate definition of what it means to provide care to a family member with ET is also likely to help advocates and policy makers push for health-care benefits that address their unique needs. Clinically, caregivers may benefit from learning about providing support beyond aiding with IADL. This information could both help to prepare new caregivers for potential challenges and give validation to caregivers who are already providing emotional support to their care-recipients that may be going unacknowledged. Having access to resources for providing assistance that reduces embarrassment and increases care-recipients’ feelings of independence may be especially helpful.
Footnotes
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the National Institute of Neurological Diseases and Stroke (NINDS R01NS086736).
