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Affiliate stigma and caregiver burden in intractable epilepsy
Epilepsy & Behavior 85 (2018) 1–6
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Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh
Affiliate stigma and caregiver burden in intractable epilepsy Barbara Hansen a,⁎,1, Magdalena Szaflarski a, E. Martina Bebin b, Jerzy P. Szaflarski b a b
University of Alabama at Birmingham Department of Sociology, Birmingham, AL, USA Department of Neurology, Birmingham, AL, USA
a r t i c l e
i n f o
Article history: Received 23 March 2018 Revised 17 May 2018 Accepted 17 May 2018 Available online xxxx Keywords: Intractable epilepsy Affiliate stigma Caregiver burden Health stigma
a b s t r a c t Intractable epilepsy can be challenging for patients and for their families. Disability rates in patients are high, causing tremendous physical and emotional burden on family caregivers. Additionally, caregivers may experience affiliate stigma, where they perceive and internalize the negative societal views of a condition and exhibit a psychological response. Affiliate stigma has been rarely studied in caregivers of those with intractable epilepsy. This study examined the relationship between affiliate stigma and the levels of burden experienced by caregivers, as well as how these levels may vary between those caring for children and adults. This cross-sectional approach used a self-administered survey offered to caregivers of family members with confirmed diagnoses of intractable epilepsy. We measured burden with the 30-item Carer's Assessment of Difficulties Index (CADI) and affiliate stigma with a six-item scale examining caregivers' perceptions of stigma directed toward themselves and their family members with epilepsy. Four nested ordinary-least-squares regression models were estimated using stigma scale scores to predict levels of perceived burden adjusting for demographic variables. Age of the patient with epilepsy was dichotomized (pediatric/adult) to assess a possible moderating effect of patient's age on the relationship between stigma and caregiver burden. Respondents (N = 136) were predominantly White (83%), female (75%), and married (69%), with an average age of 43 years. Patients with epilepsy were 52% male with ages ranging from 2 to 82 years. Each of the regression models yielded positive associations (p b 0.001) between perceived levels of caregiver burden and affiliate stigma. Additionally, the age of the family member with epilepsy moderated (p b 0.05) the effect, with the relationship stronger for caregivers of adults. In a highly select group of patients with refractory epilepsy recruited mostly from a cannabidiol (CBD) clinic, this study demonstrated that caregivers experience affiliate stigma, which is significantly associated with higher burden levels. Additionally, this study identified specific needs, which when met, may improve caregivers' physical and mental health. © 2018 Elsevier Inc. All rights reserved.
1. Introduction
1.1. Stigma in epilepsy
There is evidence of stigma associated with epilepsy [1–3] and other literature supporting the notion that caregiver burden in epilepsy is widespread [4–6]. However, there is little information on how stigma itself might be associated with caregiver burden for familial caregivers. In addition, different challenges can arise for caregivers of pediatric patients compared with those who look after adult patients [3]. These differences in patient age may impact a relationship between stigma and burden level and should be considered.
Although media representations of epilepsy have changed over the years, they still tend to depict people with epilepsy (PWE) as scary, unpredictable, dangerous, and out-of-control [7]. This does little to inform the general public about the varied nature of this disorder and contributes to the perpetuation of the stigma associated with epilepsy. People with epilepsy are treated differently and are often avoided or excluded from social activities [8]. As epilepsy is frequently associated with mental illness, PWE were, and still are by some, believed to be evil, dangerous, sexually deviant, aggressive, and unreliable [9]. Although public attitudes concerning epilepsy have changed for the better, there are still instances of prejudice and discrimination toward PWE and their families [1,10]. People with epilepsy can experience both enacted and perceived stigma, with the former focused on discriminatory attitudes and behaviors from society [11,12] and the latter on the internalization of
⁎ Corresponding author at: Department of Sociology, Human Services and Criminal Justice, HSU Box 7761, Arkadelphia, AR 71999, USA. E-mail address: [email protected] (B. Hansen). 1 Current address: Henderson State University Department of Sociology, Human Services, and Criminal Justice, Arkadelphia, AR, USA.
https://doi.org/10.1016/j.yebeh.2018.05.028 1525-5050/© 2018 Elsevier Inc. All rights reserved.
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B. Hansen et al. / Epilepsy & Behavior 85 (2018) 1–6
those negative views [11–13]. Family stigma occurs when relatives of a PWE experience prejudice and discrimination simply by being related to someone with epilepsy [11]. Familial caregivers of those with intractable epilepsy are also susceptible to affiliate stigma. This occurs when family members not only perceive discriminatory attitudes and behaviors from others but also, in turn, internalize those negative views of epilepsy which can result in negative psychological responses such as increased anxiety, depression, and stress [14–16]. Prior research in affiliate stigma has typically been focused on caregiver quality of life (QOL) and almost exclusively in relation to caring for a family member with autism, a developmental disability, or a mental illness [15–19]. The unique experiences of caring for someone with a highly stigmatizing health condition such as epilepsy begs further investigation. It is possible that caregivers of individuals with intractable epilepsy may respond with higher stress-related feelings of caregiver burden. 1.2. Caregiving burden in intractable epilepsy Providing care for a family member with intractable epilepsy can impose a financial, physical, social, and psychological burden on a caregiver, possibly because of the unstructured and unsupported nature of the arrangement [6,20]. Approximately 30% of PWE have intractable epilepsy [21] that adversely affects their physical and mental health and interferes with their normal social functioning [22]. In a 2005 analysis, over 85% of PWE with intractable epilepsy reported some form of disability [23]. Care needs in epilepsy can be as simple as providing transportation or can include more complex activities such as feeding, bathing, and changing diapers for those with debilitating brain injuries. With the majority of this care being provided by family members, the level of burden on caregivers can be great [3]. Prior research typically examined epilepsy stigma as it was perceived by the adolescent or adult PWE rather than the familial caregiver. The studies that did focus on caregivers typically looked at parents of children and how the parents' perception of stigma translated into increased senses of stigma by the child [1,2,24,25]. Stigma, occasionally, was measured as a component of overall burden [10,19] but was not independently measured or differentiated nor was it considered as a possible contributor to caregiver burden levels. In addition, there were no studies identified that examined burden experienced by caregivers of both pediatric and adult populations, and there were very few focusing on caregivers of adults [6]. Because there are so few studies examining the burden associated with caring for adult PWE, this study was of particular importance. Filling this gap will provide a broader understanding of the lived experience of caring for family members with intractable epilepsy.
confidentiality would be maintained to the extent allowed by law and that participation would in no way affect their relationship with the institution. All respondents signed the informed consent before completing the survey. 2.2. Sample Participants aged 18 years and older were recruited from the general epilepsy pediatric and adult clinics and from a State of Alabamasponsored cannabidiol (CBD) study [26,27] from June 2016 to November 2016. Inclusion criteria were met if participants were familial caregivers of patients with a diagnosis of intractable epilepsy, confirmed by the patient's neurologist. Respondents were formally asked if they were a primary caregiver to a family member with intractable epilepsy with the first survey question. Neurologists in general clinics in multiple sites identified eligible current patients, and the questionnaire was offered to their caregivers during a clinic visit. Epilepsy was confirmed by previous electroencephalogram (EEG) or video-electroencephalogram (VEEG) results and was considered intractable if the patient had experienced recurrent seizures and had a history of four or more antiepileptic medication attempts that either produced intolerable side effects or failed to control seizures [21,22]. Because of the recruitment strategies, the number of participants who declined to participate is unknown. Caregivers of patients with comorbid conditions such as intellectual disability and psychiatric comorbidities such as major depression were not excluded, but we did not collect these data because of the limited study scope and resources. Including familial caregivers of participants in the CBD study provided a unique recruitment opportunity as the study included both children and adults, required a confirmed diagnosis of intractable epilepsy, and levels of caregiver burden were expected to be high [26]. 2.3. Procedure Permission was sought from the neurologists in the general clinics as well as the principal investigators on the CBD study before approaching the family members. Participants read and signed the IRB-approved informed consent before completing the questionnaire. The selfadministered questionnaire included two short demographic sections, one for the PWE and another for the familial caregiver [Appendix A]. Caregiver burden was measured using the Carer's Assessment of Difficulties Index (CADI) instrument [28] while perceived stigma was measured with a 6-item affiliate stigma scale adapted for use in this study [29]. Open-ended questions asked what services the family might find helpful.
1.3. Study aims 2.4. Measurement To our knowledge, no study has investigated whether variations in caregiver burden and affiliate stigma occur between pediatric and adult patient populations. The purpose of this study was to examine the relationship between caregiver's perceived level of stigma and perception of burden and to assess what role patient's age might play in this relationship. 2. Materials and methods 2.1. Study design and informed consent procedure This study used a cross-sectional quantitative approach (structured survey) with a few open-ended questions. The self-administered survey was offered to caregivers of family members with confirmed diagnoses of intractable epilepsy. Ethics approval for the study protocol and consent forms were granted by the Institutional Review Board (IRB) of the University of Alabama at Birmingham through an expedited review for human subject research. Participants were assured that
2.4.1. Dependent variable The CADI was designed to assess caregiver perceptions of the difficulties associated with caregiving across a stress continuum and is appropriate for caregivers of all ages of impaired family members [28]. As a whole, the CADI has high internal consistency, α = 0.92 [28]. A unidimensional index measure, CADI was intended for use as an assessment of individual challenges of caregiving and has been used in the UK and across Europe as a 3-part family assessment instrument by social workers. Principal component analyses (PCA) have been conducted for the CADI in mixed caregiving groups and caregivers of people with dementia, but not specifically for caregivers of people with intractable epilepsy [30]. The original index includes 30 individual items that measure overall scores of burden with the option to divide into subscales or individual items when a caregivers' needs were being considered and social care plans drawn up to provide assistance to families. The assessment instrument was developed with an appreciation of the nature of disability and was designed
B. Hansen et al. / Epilepsy & Behavior 85 (2018) 1–6
to capture contributors to stress experienced by caregivers within a framework that considers financial, physical, social, and emotional factors [28]. For use in this study, three modifications were made to the index to reflect American spelling and vernacular. Each of the 30 items had four possible responses for each statement, each with a value of 0–3. Responses included: “This statement does not apply to me” (value = 0), and “This statement applies to me and I find it: not stressful, stressful, or very stressful” (values 1–3, respectively). Possible index scores ranged from 0 to 90, with higher scores indicating greater perceived overall burden. Including the response “This statement does not apply to me” was an integral component of the measure as it assessed how the respondent felt about each statement as it applied to them personally within the assessment framework. Answering that a particular statement did not apply to the respondent did not mean that the question did not ever apply to the respondent, per se, but was a level of feeling toward the statement in a caregiving capacity. No responses were excluded in the calculations of total CADI scores. 2.4.2. Independent variables As there was no validated scale to measure affiliate stigma, we adapted and refined the 3-item scale used by researchers examining family or “courtesy” stigma experienced by mothers of children with epilepsy in Zambia [29]. Their 3-item scale included three statements: “Because of my child's epilepsy: 1) some people are uncomfortable with me/my child, 2) some people would prefer to avoid me/my child, and 3) some people treat me/my child like an inferior person.” Each affirmative answer received a score ranging from 0 to 3. Our adapted scale separated these into six statements in order to differentiate between the stigma familial caregivers perceived directed toward them from the stigma directed toward their family members, then combined the two into a total stigma score as a way to operationalize affiliate stigma. Wording was changed from “my child” to “my family member” in order to accommodate all ages of PWE and familial relationships other than parent/child. The total affiliate stigma scores had a possible range of 0–6. Higher scores were reflective of greater feelings of stigmatization. Because one of the aims of this study was to examine variations in burden and stigma levels by age, age was recoded into a dichotomous age-category variable. The decision was made to differentiate between children and adults using the age of 20 years because special needs children in the State of Alabama are allowed to attend public schools and receive services through this age. Upon turning 21 years old, they are no longer eligible to receive education services through the public school system [31]. 2.5. Analysis All analyses were performed using Stata 13.1 [32]. A power analysis was conducted to determine the minimum sample size required for this study's analytical strategy which determined a minimum sample size of 76 respondents. Descriptive analyses were calculated to report demographic data and a Cronbach's alpha score (α) calculated for the total CADI score. Pearson's correlations, with two-tailed significance tests, were performed to identify significant relationships between the stigma perceived toward the PWE, stigma toward the caregiver, and the continuous age variable. Four-nested ordinary-least-squares (OLS) regression models were estimated with an F-test to determine which model had the best fit. Collinearity between independent variables was assessed with a variance inflation factor (VIF) score. Two-tailed t-tests were conducted with significance set at α = 0.05. In Model 1, the estimated model used the CADI total score variable as the dependent variable with the total stigma score as the main independent variable. In Model 2, the regression model from above was again estimated, this time adding in the categorical age variable (pediatric/ adult). Model 3 was then estimated controlling for the following
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demographic variables: caregiver age (continuous), number of people in the household (continuous), race of caregiver (dichotomized White/non-White), whether or not their family member was in the CBD study (dichotomized), caregiver income level (continuous), and whether or not the caregiver had at least a bachelor's degree (dichotomized). In order to normalize the distribution, the log of annual income was used because the distribution of the income variable as it is with actual values was positively skewed. Finally, in Model 4, the interaction term between the total stigma score and age category was added. Individuals with incomplete questionnaires were excluded in these analyses. Bivariate analyses showed no relationships of the control variable caregiver sex to total CADI score, total stigma score, stigma toward the PWE, or stigma toward the caregiver, and, thus, it was not included in the multivariable analysis. 3. Results Of 154 returned surveys, 136 were included in the analyses. Of the 18 surveys not included in the analyses, three were returned without completed signatures on the informed consent, 14 surveys were incomplete, and 1 survey was complete but the respondent indicated he was not a primary caregiver of a family member with intractable epilepsy. Demographic characteristics are listed in Table 1. Overall, the caregivers in the study were predominantly White, female, and married, with at least some education past high school. Results for the CADI analyses are shown in Table 2. The Cronbach's alpha score was 0.95 indicating high reliability and responses to each of the 30 items are included across the stress continuum. The item most frequently experienced by respondents was “Caregiving is physically tiring” at 86% with 65% of those affected finding it stressful or very stressful. Another 75% of respondents reported their sleep was affected with over 30% of them saying they found it very stressful. The 6 items in the affiliate stigma scale were analyzed individually and collectively (Table 3). Almost three quarters of caregivers felt that people were uncomfortable being around their family member, and half believed others avoided and treated them in an inferior manner. The majority of caregivers felt stigma directed toward themselves in some form. Correlations were performed to assess possible significant assocations between age of PWE, perceived stigma of the caregiver, and perceived stigma toward the PWE. Perceived stigma of the caregiver had weak negative relationships with caregiver age (− 0.18) and PWE age (−0.17), both significant at p b 0.05. Four-nested OLS regression models were estimated with total CADI scores as the dependent variable. Results are presented in Table 4. When considered as the only independent variable, affiliate stigma explained 24% of the variation in caregiver burden scores. The effect of perceived stigma on caregiver burden was not ameliorated by whether the PWE was an adult or a child when only looking at these two independent variables. Instead, there was a slight suppression Table 1 Demographic characteristics of respondents (N = 136) and patients with epilepsy.a Variable Respondents Mean age (years) % male % White % married % bachelor's degree or higher Mean annual income category Patients with epilepsy Mean age (years) % male % adult (ages 21 and up) % in CBD study a
Mean or %
SD
Range
48 25.0 83.1 69.1 32.4 $40K–$50K
13
22 to 83
23 52.2 44.9 60.3
17
b$5K to N$200K
Respondents are familial caregivers of patients with epilepsy.
2 to 82
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B. Hansen et al. / Epilepsy & Behavior 85 (2018) 1–6
Table 2 Carer's Assessment of Difficulty Index (CADI) item frequencies (N = 136).
Table 4 Four-nested OLS regression models predicting total CADI scores, (N = 136).
Individual item frequencies
A
B
C
D
I can feel helpless/not in control of the situation. My emotional well-being suffers. It can put a strain on family relationships. I can't relax because of worry about caring. I don't have enough private time for myself. I can't devote enough time to other family members. I feel angry about the situation. I feel guilty about the situation. It is physically tiring. The person I care for is immobile/has problems getting about. The person I care for doesn't always help as much as they could. My physical health has suffered. The person I care for needs a lot of help with personal care. My sleep is affected. The person I care for is incontinent. It restricts my social life/outside interests. I can't have a break or take a holiday. I can't see friends as often as I'd like. Professional workers don't appreciate the problems caregivers face. I don't get enough help from the health and social services. It causes financial difficulties. My standard of living has fallen. Relatives don't keep in touch as often as I'd like. Some family members don't help as much as they could. The person I care for sometimes manipulates me. The person I care for can demand too much of me. I no longer have a meaningful relationship with the person I care for. The person I care for doesn't always appreciate what I do. The behavior of the person I care for is a problem. There is no satisfaction to be gained from caring.
21 37 28 26 36 37 69 66 19 67 58 66 36 34 71 30 48 48 53
35 21 28 36 31 33 33 34 36 25 31 23 39 37 26 35 32 31 34
52 63 51 45 41 45 20 14 41 17 32 28 31 34 21 37 35 41 31
28 15 29 29 28 21 14 22 40 27 15 19 30 31 18 34 21 16 18
57 37 69 70 52 93 74 115
22 29 25 26 25 14 25 12
30 34 17 20 28 20 26 6
27 36 25 20 31 9 11 3
81 34 15 6 82 19 22 13 110 15 7 4
A = This statement does not apply to me. B = This statement applies to me and I find it: not stressful. C = This statement applies to me and I find it: stressful. D = This statement applies to me and I find it: very stressful.
effect, with the inclusion of age strengthening the relationship rather than mediating. When the age category was added to the model, the coefficient for stigma increased with both of the independent variables statistically significant (stigma, p b 0.001; age, p b 0.05.) After controlling for demographic variables in Model 3, the relationship between affiliate stigma and caregiver burden remained significant. Post hoc Wald test results indicated that compared with caregivers of children, caregivers of adults experienced significantly higher levels of burden. Age of the family member with epilepsy had a moderating effect on the relationship between total perceived stigma and burden levels, as shown in Model 4. Not only was the final model highly significant, the model as a whole also accounted for 14% more of the variation in burden scores than the first model where stigma was the sole independent variable. 4. Discussion In this study, the percentage of caregivers reporting perceived stigma is higher than those in other studies that show just over 50% Table 3 Frequency distribution for affiliate stigma scale items (N = 136). Individual items
People are uncomfortable around me People avoid me People treat me like an inferior person People are uncomfortable around my family member People avoid my family member People treat my family member like an inferior person a
Response categories were “yes” and “no”.
“Yes” responsesa f
%
69 48 30 101 68 57
50.7 35.3 22.1 74.3 50.0 47.1
Predictor
Total stigma Adult PWEa Caregiver age Family size White In CBDb study Income (log) ≥Bachelor deg Stigma ∗ age Constant F (df) R2 Nested F
Model 1
Model 2
Model 3
Model 4
β
SE
β
SE
β
SE
β
SE
4.85⁎⁎⁎
0.69
5.10⁎⁎⁎ 6.37⁎
0.69 3.01
4.66⁎⁎⁎ 7.77⁎ −0.07 0.74 −4.37 7.71⁎
0.69 3.65 0.15 1.04 4.12 3.09 1.63 3.48
3.50⁎⁎⁎ 0.56 −0.09 0.70 −4.32 8.13⁎⁎
0.90 5.06 0.15 0.99 4.07 3.06 1.61 3.44 1.35 18.72
−3.07 4.58 20.08 2.43 49.93⁎⁎⁎ (1, 134) 0.27 –
16.53 2.93 27.84⁎⁎⁎ (2, 133) 0.30 4.46⁎
48.81 18.78 8.95⁎⁎⁎ (8, 127) 0.36 2.17
−3.16 4.92 2.74⁎ 53.82 8.61⁎⁎⁎ (9, 126) 0.38 4.12⁎
⁎ p b 0.05. ⁎⁎ p b 0.01. ⁎⁎⁎ p b 0.001. a People with epilepsy. b Cannabidiol.
feeling stigmatized [1,2]. In reviewing the results from each of the individual stigma items, we noted that almost 75% reported that people are uncomfortable around their family member, and 51% said that others are uncomfortable around them. Thus, caregivers appeared to be acutely aware that they and their family members bear a stigma. This may be because of the intractable nature of seizures in this study and the difficulty in concealing the condition from others. One of the primary components of perceived stigma is the shame the affected person feels from the negative attitudes they perceive and the subsequent attempts to hide the stigmatizing condition as a way to manage their “spoiled” identity [11–13]. Epilepsy is typically considered a concealable stigmatized identity [33] but in the case of intractable epilepsy, this can be more difficult to achieve. This level of discomfort could lead caregivers to use avoidance and social isolation as ways of coping with the stress of anticipated stigma. For example, several respondents commented that they purposely do not expose their family members to the outside world in order to protect them from rude looks and comments. One mother said of her 22-year-old daughter, “She is always so happy. She doesn't know how mean and awful people can get and I'm not about to let her find out.” Another mother said, “We just stay home; don't go anywhere. We used to go to church but he got to where he would scream and yell and people would act like they wanted us to leave, like it was my fault or something.” These instances of isolating or hiding in an attempt to manage the stigma they expected to receive are similar with impression management techniques in other health conditions such as morbid obesity [34], HIV [35], and leprosy [36]. In these health conditions though, the affected person decides to hide from public view while in intractable epilepsy, the person making that decision to isolate is often a wellmeaning family member. There is evidence of stigma coaching leading to the concealment of an epilepsy diagnosis [23], but this can be difficult or nearly impossible with intractable epilepsy as seizures are more frequent and unpredictable. More than one family caregiver echoed one father who said, “Usually it's just easier to stay in when he's having a bad day. No one will stare at us at home.” The CADI is an excellent instrument for measuring the different domains of caregiving. The thirty items in the index cover an exhaustive list of activities, feelings, and situations that add to the stress in caring for someone with intractable epilepsy. Most burden studies in epilepsy use either a qualitative methodology with interviews or focus groups [2,20] or a checklist of health-related QOL issues [5,6], and the fact that stigma is not addressed in the CADI is a distinct advantage. Using this instrument makes it simpler to differentiate stigma from caregiving.
B. Hansen et al. / Epilepsy & Behavior 85 (2018) 1–6
The acknowledgment that caregiving is physically tiring was the item most reported as applicable to respondents. This is understandable because intractable epilepsy is frequently associated with intellectual and physical disabilities rendering some patients unable to independently perform activities of daily life such as walking, bathing, feeding oneself, and using the toilet [20]. As PWE grow out of childhood and into adulthood, they become larger and heavier, making it more difficult for caregivers to perform these activities. In addition, sleep is affected for many caregivers in this study similar with results from other studies. Reduced hours of sleep and fitful sleep due to worry about seizures are common occurrences [3,20]. One mother in this study reported that when her child is having a difficult day with seizures, she sleeps in a sleeping bag on the floor next to her daughter's bed in case she needs her. The CADI, in allowing respondents to choose the option “This statement does not apply to me,” offered us a chance to examine the levels of stress associated with each item relative to other caregivers experiencing the stressor. There were a few respondents who this category on every item in the index indicating no burden at all. One mother remarked, “[Son's name] is a gift from God. I was chosen to take care of him and it wouldn't do for me to act like that was a problem.” Another parent remarked, “It would be nice to not worry about buying the new wheelchair but God will provide.” While religiosity was not measured in this study, there was a strong thread of religious faith running through the spoken and written comments made by those surveyed. Results from this study showed that there is a positive relationship between affiliate stigma and perceived burden among caregivers in intractable epilepsy. Because previous studies did not typically differentiate between stigma and burden, it has been unclear how much of an effect perceived stigma might have on burden. This is the first study to show a very strong and statistically significant association. Other studies also did not include caregivers of both children and adults. Unfortunately, this aspect is overlooked in most studies of caregiving in epilepsy. In addition, the association between stigma and caregiver burden was stronger among caregivers of adult PWE compared with those looking after pediatric PWE, and that was after factoring in the age, race, family size, household income, and education level of the caregivers. It is surprising that the number of people in a household was not significantly associated with caregiver burden scores. Intuitively, it may seem that if there are more people in a home to take care of, the burden load would be greater for a primary caregiver or perhaps smaller families might not have others to share in their tasks and experience greater burden, but this did not seem to be the case. One interesting finding was that when the PWE were in the CBD study, their caregivers experienced more total burden. One possible explanation for this might be the high frequency of visits the PWE make to the research clinic for the study. Until patients are stable on a dosage of CBD, they are required to attend clinic every 2 weeks (uab.edu/cbd). For patients who are combative or nonambulatory or require significant assistance with personal care, this can be challenging for caregivers, especially those who drive for several hours. Because many of the caregivers were recruited from the CBD study, it is possible that the caregivers were experiencing high levels of stress while completing the survey. There is also a possibility that seizure frequencies and severity may vary between those in the CBD study and those not participating, but these data were not specifically collected. Some caregivers who are against using marijuana for medical (or any) purposes might have not considered participating in the study. These variations remain unclear and might be examined in future studies.
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is a need several caregivers expressed. Other items related to medical office environment were dim lighting, quiet areas to wait with few environmental triggers, and family style restrooms that accommodate large wheelchairs. Access to social services that are available in their area would be extremely helpful. Many caregivers noted that they have no idea if any services were available. However, they would find a list useful. Most neurologists are affiliated with hospitals which have on-staff medical social workers who could connect caregivers with agencies. Understanding the stress that caregivers of highly dependent patients are under may influence scheduling in order to reduce the time patients spend in waiting rooms, which is another need mentioned. The number one need stated by respondents in this study was respite care. Some even elaborated that they did not want babysitting services but trained professionals who were not afraid of epilepsy. Several caregivers explained that although there were a few day programs available in their counties for intellectually disabled citizens, almost none accepted adults with epilepsy, presumably because of fear of seizures. Some caregivers reported not having seen a movie in years because there was no one with whom to leave their child. The second most frequently cited need was a support group where caregivers of PWE could get together to talk and share experiences or resources. One respondent explained that even an online group would be helpful. Another frequently mentioned need was a website for caregivers that included links to access services and social agencies with help for families taking care of chronically ill relatives. As more research in affiliate stigma and caregiver burden in intractable epilepsy is conducted, more evidence will be available to inform grant proposals for services and implement policies to assist families. 4.2. Limitations Limitations of the study need to be discussed. With the use of a convenience sample and the study's cross-sectional nature, random sampling was not possible. A longitudinal study would have been more robust, as it would examine the dynamic variations in perceived levels of stigma and caregiver burden over time, but this was not possible within our study's constraints. Data concerning the severity and types of seizures patients experienced were not captured. This information would have been helpful in examining the differences between the group in the CBD study and those not enrolled. It is possible that those in the study suffered more debilitating epilepsy leading to higher levels of burden. Caregivers of patients with comorbid conditions such as intellectual disability and psychiatric comorbidities such as major depression were not excluded and this needs to be addressed in future studies. It is also possible that the group not enrolled in the CBD study were ineligible because of coexisting health or genetic issues. Future studies should include gathering seizure types, frequencies, and comorbid conditions. Recruitment should focus more outside a CBD study in order for results to be more generalizable. Another limitation is the fact that the survey was self-administered, which may introduce bias. A question might also be added to the survey examining the familial relationship of the caregiver to the PWE, i.e., parent, sibling, spouse, and child. There was also little variation in race and ethnicity with respondents being mostly White with no Hispanic or Latino participants. Using data from an original survey is also a limitation in that the survey has not been validated from repeated use in other studies. 4.3. Implications for further research
4.1. Clinical repercussions and suggested solutions Neurologists who treat PWE might use these results to assist in identifying concerns of caregivers. With limited time spent during office visits, the needs of caregivers might be overlooked. Adequate space in waiting rooms and examination rooms for wheelchairs and caregivers
This study is an important step in the research into stigma and caregiver burden in intractable epilepsy. As one of the first studies to provide evidence of affiliate stigma among caregivers in intractable epilepsy, it adds to the literature from which to expand future research. Repeating this study will provide opportunities for validation of the survey
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instrument. Other studies might utilize a mixed-methods approach, using the instrument as a quantitative measurement of affiliate stigma and caregiver burden and incorporate a few in-depth interviews or focus groups to flesh out the perceptions of stigma and to discuss the specific aspects of burden that are troubling. Particular emphasis in future studies might examine the psychological responses in affiliate stigma exhibited by caregivers, such as isolating behaviors and other coping mechanisms. Before this study, affiliate stigma in intractable epilepsy had not been examined. In fact, it has not been examined in many health-related conditions. Viewing epilepsy through this lens can provide motivation to study affiliate stigma in other disorders. Results from this study also add to the literature on caregiving in chronic illness and the stress process. Being able to control for comorbidities would be advantageous in that we could differentiate between the burden and stigma due to the epilepsy and those due to the comorbidity. We urge future studies to close this gap. 5. Conclusions Despite its limitations, this study extends past literature by providing a conceptual framework for studying affiliate stigma in intractable epilepsy. Differentiating the perception of stigma from the perception of burden, examining caregivers for both children and adults, and assessing caregiving activities across a stress continuum are unique to this study. In addition, using stigma as a possible explanation for perceived burden levels expands on the research into affiliate stigma. Further research is needed in examining the intersection of affiliate stigma and caregiver burden. Results from this study and respondents' comments may be incorporated into grant proposals exploring how best to meet the clinical and service needs in this population. Supplementary data to this article can be found online at https://doi. org/10.1016/j.yebeh.2018.05.028. Conflicts of interest There are no funding sources to report and no conflicts of interests for any of the authors. Acknowledgments This work was part of the doctoral thesis requirement of the first author. The authors wish to thank Mike Nolan for his insight into using the CADI as an assessment tool and for providing a copy of the original CADI handbook, Assessing the Needs of Family Carers: A Guide for Practitioners (1998), as this book was out of print and could no longer be purchased. Further appreciation goes to Pongkiat Kankirawatana, MD, for allowing us the opportunity to offer the survey to parents of his pediatric patients. References [1] Jacoby A, Austin JK. Social stigma for adults and children with epilepsy. Epilepsia 2007;48(Supplement 9):6–9. [2] VanStraten AF, Ng YT. What is the worst part of having epilepsy? A children's and parents' perspective. Pediatr Neurol 2012;47:431–5. [3] Gibson PA. Lennox–Gastaut syndrome: impact on the caregivers and families of patients. J Multidiscip Healthc 2014;7:441–8. [4] Nolan M, Grant G. Regular respite: an evaluation of a hospital rota bed scheme for elderly people from the Age Concern Institute of Gerontology. London: Ace Books; 1992. [5] Van Andel J, Zijlmans M, Fischer K, Leijten FSS. Quality of life of caregivers of patients with intractable epilepsy. Epilepsia 2009;50(5):1289–300.
[6] Karakis I, Cole AJ, Montouris GD, San Luciano M, Meador KJ, Piperidou C. Caregiver burden in epilepsy: determinants and impact. Epilepsy Res Treat 2014;2014:808421, 9 (https://www.hindawi.com/journals/ert/2014/808421/ [Accessed 10 March 2018]) https://doi.org/10.1155/2014/808421. [7] Kerson JF, Schwaber-Kerson T, Kerson LA. The depiction of seizures in film. Epilepsia 1999;40(8):1163–7. [8] Dilorio C, Kobau RE, Holden W, Berkowitz JM, Kamin SL, Antonak RF, et al. Developing a measure to assess attitudes toward epilepsy in the US population. Epilepsy Behav 2004;5(6):965–75. [9] Prioreschi PA. History of medicine: primitive and ancient Medicine. 2nd edition. Omaha, NE: Horatius Press; 1996. [10] Jacoby A. Stigma, epilepsy, and quality of life. Epilepsy Behav 2002;3:S10–20. [11] Goffman E. Stigma: notes on managing a spoiled identity. New York: Simon and Schuster; 1963. [12] Link BG, Phelan JC. Conceptualizing stigma. Annu Rev Sociol 2001;27:363–85. [13] Scambler G. Health-related stigma. Sociol Health Illn 2009;31(3):441–55. [14] Rodenburg R, Meijer AMM, Dekovic M, Aldenkamp AP. Parents of children with enduring epilepsy: predictors of parenting stress and parenting. Epilepsy Behav 2007; 11:197–207. [15] Mak WWS, Cheung RYM. Affiliate stigma among caregivers of people with intellectual disability or mental illness. J Appl Res Intellect Disabil 2008;21:532–45. [16] Mak WSS, Cheung RYM. Psychological distress and subjective burden of caregivers of people with mental illness: the role of affiliate stigma and face concern. J Comm Ment Health 2012;48:270–4. [17] Mak WSS, Kwok YTY. Internalization of stigma for parents of children with autism spectrum disorder in Hong Kong. Soc Sci Med 2010;70:2045–51. [18] Chiu MYL, Yang X, Wong HT, Li JH, Li J. Caregiving of children with intellectual disabilities in China: an examination of affiliate stigma and the cultural thesis. J Intellect Disabil Res 2013;57(12):1117–29. [19] Werner S, Shulman C. Subjective well-being among family caregivers of individuals with developmental disabilities: the role of affiliate stigma and psychosocial moderating variables. Res Dev Disabil 2013;34:4103–14. [20] Smith G, Wagner J, Andrews J, Austin J, Mueller M, Carter E, et al. Caregiving in pediatric epilepsy: results of focus groups and implications for research and practice. Epilepsy Behav 2014;34:34–41. [21] Kwan P, Brodie MJ. Early identification of refractory epilepsy. N Engl J Med 2000; 342:314–9. [22] Devinsky O. Patients with refractory seizures. N Engl J Med 1999;340:1565–70. [23] Center for Disease Control. Prevalence of epilepsy and health-related quality of life and disability among adults with epilepsy — South Carolina, 2003 and 2004. MMWR Morb Mortal Wkly Rep 2005;54:1080–2. [24] Schneider J, Conrad P. In the closet with illness: epilepsy, stigma potential and information control. Soc Probl 1980;28:32–44. [25] Scambler G, Hopkins A. Being epileptic: coming to terms with stigma. Sociol Health Illn 1986;8(1):26–43. [26] Szaflarski M, Hansen B, Bebin EM, Szaflarski JP. Social correlates of health status, quality of life, and mood states in patients treated with cannabidiol for epilepsy. Epilepsy Behav 2017;70(Pt. B):36–369. [27] Szaflarski JP, Bebin EM. Cannabis, cannabidiol, and epilepsy — from receptors to clinical response. Epilepsy Behav 2014;41:277–82. [28] Nolan M, Grant G, Keady J. Assessing the needs of family carers: a guide for practitioners. Brighton, UK: Pavilion; 1998. [29] Elafros M, Sakubita-Simasiku C, Atadzhanov M, Haworth A, Chomba E, Birbeck GL. Stigma and psychiatric morbidity among mothers of children with epilepsy in Zambia. J Int Health 2013;5:288–94. [30] Charlesworth GM, Tzimoula XM, Newman SP. Carer's assessment of difficulties index (CADI): psychometric properties for use with carers of people with dementia. Aging Ment Health 2007;11(2):218–25. [31] Alabama Administrative Code. 290-8-9.04(3). Rules of the Alabama state board of education state department of education chapter 290-8-9 special education services, Supplement 13–3. https://www.alsde.edu/sec/ses/Policy/AAC%20290-8-9%2011-42013.pdf; 2013. [Accessed 10 March 2018]. [32] StataCorp. Stata statistical software: release 13. College Station, TX: StataCorp LP; 2013. [33] Benson A, O'Toole S, Lambert V, Gallagher P, Shahwan A, Austin JK. To tell or not to tell: a systematic review of the disclosure practices of children living with epilepsy and their parents. Epilepsy Behav 2015;51:73–95. [34] Hansen B, Dye MH. Damned if you do, damned if you don't: the stigma of weight loss surgery. Deviant Behav 2016;39(2):137–47. [35] Siegel K, Lune H, Meyer IH. Stigma management among gay/bisexual men with HIV/AIDS. Qual Sociol 1998;21(1):3–24. [36] Barrett R. Self-mortification and the stigma of leprosy in northern India. Med Anthropol Q 2005;19(2):216–30.
Contents lists available at ScienceDirect
Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh
Affiliate stigma and caregiver burden in intractable epilepsy Barbara Hansen a,⁎,1, Magdalena Szaflarski a, E. Martina Bebin b, Jerzy P. Szaflarski b a b
University of Alabama at Birmingham Department of Sociology, Birmingham, AL, USA Department of Neurology, Birmingham, AL, USA
a r t i c l e
i n f o
Article history: Received 23 March 2018 Revised 17 May 2018 Accepted 17 May 2018 Available online xxxx Keywords: Intractable epilepsy Affiliate stigma Caregiver burden Health stigma
a b s t r a c t Intractable epilepsy can be challenging for patients and for their families. Disability rates in patients are high, causing tremendous physical and emotional burden on family caregivers. Additionally, caregivers may experience affiliate stigma, where they perceive and internalize the negative societal views of a condition and exhibit a psychological response. Affiliate stigma has been rarely studied in caregivers of those with intractable epilepsy. This study examined the relationship between affiliate stigma and the levels of burden experienced by caregivers, as well as how these levels may vary between those caring for children and adults. This cross-sectional approach used a self-administered survey offered to caregivers of family members with confirmed diagnoses of intractable epilepsy. We measured burden with the 30-item Carer's Assessment of Difficulties Index (CADI) and affiliate stigma with a six-item scale examining caregivers' perceptions of stigma directed toward themselves and their family members with epilepsy. Four nested ordinary-least-squares regression models were estimated using stigma scale scores to predict levels of perceived burden adjusting for demographic variables. Age of the patient with epilepsy was dichotomized (pediatric/adult) to assess a possible moderating effect of patient's age on the relationship between stigma and caregiver burden. Respondents (N = 136) were predominantly White (83%), female (75%), and married (69%), with an average age of 43 years. Patients with epilepsy were 52% male with ages ranging from 2 to 82 years. Each of the regression models yielded positive associations (p b 0.001) between perceived levels of caregiver burden and affiliate stigma. Additionally, the age of the family member with epilepsy moderated (p b 0.05) the effect, with the relationship stronger for caregivers of adults. In a highly select group of patients with refractory epilepsy recruited mostly from a cannabidiol (CBD) clinic, this study demonstrated that caregivers experience affiliate stigma, which is significantly associated with higher burden levels. Additionally, this study identified specific needs, which when met, may improve caregivers' physical and mental health. © 2018 Elsevier Inc. All rights reserved.
1. Introduction
1.1. Stigma in epilepsy
There is evidence of stigma associated with epilepsy [1–3] and other literature supporting the notion that caregiver burden in epilepsy is widespread [4–6]. However, there is little information on how stigma itself might be associated with caregiver burden for familial caregivers. In addition, different challenges can arise for caregivers of pediatric patients compared with those who look after adult patients [3]. These differences in patient age may impact a relationship between stigma and burden level and should be considered.
Although media representations of epilepsy have changed over the years, they still tend to depict people with epilepsy (PWE) as scary, unpredictable, dangerous, and out-of-control [7]. This does little to inform the general public about the varied nature of this disorder and contributes to the perpetuation of the stigma associated with epilepsy. People with epilepsy are treated differently and are often avoided or excluded from social activities [8]. As epilepsy is frequently associated with mental illness, PWE were, and still are by some, believed to be evil, dangerous, sexually deviant, aggressive, and unreliable [9]. Although public attitudes concerning epilepsy have changed for the better, there are still instances of prejudice and discrimination toward PWE and their families [1,10]. People with epilepsy can experience both enacted and perceived stigma, with the former focused on discriminatory attitudes and behaviors from society [11,12] and the latter on the internalization of
⁎ Corresponding author at: Department of Sociology, Human Services and Criminal Justice, HSU Box 7761, Arkadelphia, AR 71999, USA. E-mail address: [email protected] (B. Hansen). 1 Current address: Henderson State University Department of Sociology, Human Services, and Criminal Justice, Arkadelphia, AR, USA.
https://doi.org/10.1016/j.yebeh.2018.05.028 1525-5050/© 2018 Elsevier Inc. All rights reserved.
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B. Hansen et al. / Epilepsy & Behavior 85 (2018) 1–6
those negative views [11–13]. Family stigma occurs when relatives of a PWE experience prejudice and discrimination simply by being related to someone with epilepsy [11]. Familial caregivers of those with intractable epilepsy are also susceptible to affiliate stigma. This occurs when family members not only perceive discriminatory attitudes and behaviors from others but also, in turn, internalize those negative views of epilepsy which can result in negative psychological responses such as increased anxiety, depression, and stress [14–16]. Prior research in affiliate stigma has typically been focused on caregiver quality of life (QOL) and almost exclusively in relation to caring for a family member with autism, a developmental disability, or a mental illness [15–19]. The unique experiences of caring for someone with a highly stigmatizing health condition such as epilepsy begs further investigation. It is possible that caregivers of individuals with intractable epilepsy may respond with higher stress-related feelings of caregiver burden. 1.2. Caregiving burden in intractable epilepsy Providing care for a family member with intractable epilepsy can impose a financial, physical, social, and psychological burden on a caregiver, possibly because of the unstructured and unsupported nature of the arrangement [6,20]. Approximately 30% of PWE have intractable epilepsy [21] that adversely affects their physical and mental health and interferes with their normal social functioning [22]. In a 2005 analysis, over 85% of PWE with intractable epilepsy reported some form of disability [23]. Care needs in epilepsy can be as simple as providing transportation or can include more complex activities such as feeding, bathing, and changing diapers for those with debilitating brain injuries. With the majority of this care being provided by family members, the level of burden on caregivers can be great [3]. Prior research typically examined epilepsy stigma as it was perceived by the adolescent or adult PWE rather than the familial caregiver. The studies that did focus on caregivers typically looked at parents of children and how the parents' perception of stigma translated into increased senses of stigma by the child [1,2,24,25]. Stigma, occasionally, was measured as a component of overall burden [10,19] but was not independently measured or differentiated nor was it considered as a possible contributor to caregiver burden levels. In addition, there were no studies identified that examined burden experienced by caregivers of both pediatric and adult populations, and there were very few focusing on caregivers of adults [6]. Because there are so few studies examining the burden associated with caring for adult PWE, this study was of particular importance. Filling this gap will provide a broader understanding of the lived experience of caring for family members with intractable epilepsy.
confidentiality would be maintained to the extent allowed by law and that participation would in no way affect their relationship with the institution. All respondents signed the informed consent before completing the survey. 2.2. Sample Participants aged 18 years and older were recruited from the general epilepsy pediatric and adult clinics and from a State of Alabamasponsored cannabidiol (CBD) study [26,27] from June 2016 to November 2016. Inclusion criteria were met if participants were familial caregivers of patients with a diagnosis of intractable epilepsy, confirmed by the patient's neurologist. Respondents were formally asked if they were a primary caregiver to a family member with intractable epilepsy with the first survey question. Neurologists in general clinics in multiple sites identified eligible current patients, and the questionnaire was offered to their caregivers during a clinic visit. Epilepsy was confirmed by previous electroencephalogram (EEG) or video-electroencephalogram (VEEG) results and was considered intractable if the patient had experienced recurrent seizures and had a history of four or more antiepileptic medication attempts that either produced intolerable side effects or failed to control seizures [21,22]. Because of the recruitment strategies, the number of participants who declined to participate is unknown. Caregivers of patients with comorbid conditions such as intellectual disability and psychiatric comorbidities such as major depression were not excluded, but we did not collect these data because of the limited study scope and resources. Including familial caregivers of participants in the CBD study provided a unique recruitment opportunity as the study included both children and adults, required a confirmed diagnosis of intractable epilepsy, and levels of caregiver burden were expected to be high [26]. 2.3. Procedure Permission was sought from the neurologists in the general clinics as well as the principal investigators on the CBD study before approaching the family members. Participants read and signed the IRB-approved informed consent before completing the questionnaire. The selfadministered questionnaire included two short demographic sections, one for the PWE and another for the familial caregiver [Appendix A]. Caregiver burden was measured using the Carer's Assessment of Difficulties Index (CADI) instrument [28] while perceived stigma was measured with a 6-item affiliate stigma scale adapted for use in this study [29]. Open-ended questions asked what services the family might find helpful.
1.3. Study aims 2.4. Measurement To our knowledge, no study has investigated whether variations in caregiver burden and affiliate stigma occur between pediatric and adult patient populations. The purpose of this study was to examine the relationship between caregiver's perceived level of stigma and perception of burden and to assess what role patient's age might play in this relationship. 2. Materials and methods 2.1. Study design and informed consent procedure This study used a cross-sectional quantitative approach (structured survey) with a few open-ended questions. The self-administered survey was offered to caregivers of family members with confirmed diagnoses of intractable epilepsy. Ethics approval for the study protocol and consent forms were granted by the Institutional Review Board (IRB) of the University of Alabama at Birmingham through an expedited review for human subject research. Participants were assured that
2.4.1. Dependent variable The CADI was designed to assess caregiver perceptions of the difficulties associated with caregiving across a stress continuum and is appropriate for caregivers of all ages of impaired family members [28]. As a whole, the CADI has high internal consistency, α = 0.92 [28]. A unidimensional index measure, CADI was intended for use as an assessment of individual challenges of caregiving and has been used in the UK and across Europe as a 3-part family assessment instrument by social workers. Principal component analyses (PCA) have been conducted for the CADI in mixed caregiving groups and caregivers of people with dementia, but not specifically for caregivers of people with intractable epilepsy [30]. The original index includes 30 individual items that measure overall scores of burden with the option to divide into subscales or individual items when a caregivers' needs were being considered and social care plans drawn up to provide assistance to families. The assessment instrument was developed with an appreciation of the nature of disability and was designed
B. Hansen et al. / Epilepsy & Behavior 85 (2018) 1–6
to capture contributors to stress experienced by caregivers within a framework that considers financial, physical, social, and emotional factors [28]. For use in this study, three modifications were made to the index to reflect American spelling and vernacular. Each of the 30 items had four possible responses for each statement, each with a value of 0–3. Responses included: “This statement does not apply to me” (value = 0), and “This statement applies to me and I find it: not stressful, stressful, or very stressful” (values 1–3, respectively). Possible index scores ranged from 0 to 90, with higher scores indicating greater perceived overall burden. Including the response “This statement does not apply to me” was an integral component of the measure as it assessed how the respondent felt about each statement as it applied to them personally within the assessment framework. Answering that a particular statement did not apply to the respondent did not mean that the question did not ever apply to the respondent, per se, but was a level of feeling toward the statement in a caregiving capacity. No responses were excluded in the calculations of total CADI scores. 2.4.2. Independent variables As there was no validated scale to measure affiliate stigma, we adapted and refined the 3-item scale used by researchers examining family or “courtesy” stigma experienced by mothers of children with epilepsy in Zambia [29]. Their 3-item scale included three statements: “Because of my child's epilepsy: 1) some people are uncomfortable with me/my child, 2) some people would prefer to avoid me/my child, and 3) some people treat me/my child like an inferior person.” Each affirmative answer received a score ranging from 0 to 3. Our adapted scale separated these into six statements in order to differentiate between the stigma familial caregivers perceived directed toward them from the stigma directed toward their family members, then combined the two into a total stigma score as a way to operationalize affiliate stigma. Wording was changed from “my child” to “my family member” in order to accommodate all ages of PWE and familial relationships other than parent/child. The total affiliate stigma scores had a possible range of 0–6. Higher scores were reflective of greater feelings of stigmatization. Because one of the aims of this study was to examine variations in burden and stigma levels by age, age was recoded into a dichotomous age-category variable. The decision was made to differentiate between children and adults using the age of 20 years because special needs children in the State of Alabama are allowed to attend public schools and receive services through this age. Upon turning 21 years old, they are no longer eligible to receive education services through the public school system [31]. 2.5. Analysis All analyses were performed using Stata 13.1 [32]. A power analysis was conducted to determine the minimum sample size required for this study's analytical strategy which determined a minimum sample size of 76 respondents. Descriptive analyses were calculated to report demographic data and a Cronbach's alpha score (α) calculated for the total CADI score. Pearson's correlations, with two-tailed significance tests, were performed to identify significant relationships between the stigma perceived toward the PWE, stigma toward the caregiver, and the continuous age variable. Four-nested ordinary-least-squares (OLS) regression models were estimated with an F-test to determine which model had the best fit. Collinearity between independent variables was assessed with a variance inflation factor (VIF) score. Two-tailed t-tests were conducted with significance set at α = 0.05. In Model 1, the estimated model used the CADI total score variable as the dependent variable with the total stigma score as the main independent variable. In Model 2, the regression model from above was again estimated, this time adding in the categorical age variable (pediatric/ adult). Model 3 was then estimated controlling for the following
3
demographic variables: caregiver age (continuous), number of people in the household (continuous), race of caregiver (dichotomized White/non-White), whether or not their family member was in the CBD study (dichotomized), caregiver income level (continuous), and whether or not the caregiver had at least a bachelor's degree (dichotomized). In order to normalize the distribution, the log of annual income was used because the distribution of the income variable as it is with actual values was positively skewed. Finally, in Model 4, the interaction term between the total stigma score and age category was added. Individuals with incomplete questionnaires were excluded in these analyses. Bivariate analyses showed no relationships of the control variable caregiver sex to total CADI score, total stigma score, stigma toward the PWE, or stigma toward the caregiver, and, thus, it was not included in the multivariable analysis. 3. Results Of 154 returned surveys, 136 were included in the analyses. Of the 18 surveys not included in the analyses, three were returned without completed signatures on the informed consent, 14 surveys were incomplete, and 1 survey was complete but the respondent indicated he was not a primary caregiver of a family member with intractable epilepsy. Demographic characteristics are listed in Table 1. Overall, the caregivers in the study were predominantly White, female, and married, with at least some education past high school. Results for the CADI analyses are shown in Table 2. The Cronbach's alpha score was 0.95 indicating high reliability and responses to each of the 30 items are included across the stress continuum. The item most frequently experienced by respondents was “Caregiving is physically tiring” at 86% with 65% of those affected finding it stressful or very stressful. Another 75% of respondents reported their sleep was affected with over 30% of them saying they found it very stressful. The 6 items in the affiliate stigma scale were analyzed individually and collectively (Table 3). Almost three quarters of caregivers felt that people were uncomfortable being around their family member, and half believed others avoided and treated them in an inferior manner. The majority of caregivers felt stigma directed toward themselves in some form. Correlations were performed to assess possible significant assocations between age of PWE, perceived stigma of the caregiver, and perceived stigma toward the PWE. Perceived stigma of the caregiver had weak negative relationships with caregiver age (− 0.18) and PWE age (−0.17), both significant at p b 0.05. Four-nested OLS regression models were estimated with total CADI scores as the dependent variable. Results are presented in Table 4. When considered as the only independent variable, affiliate stigma explained 24% of the variation in caregiver burden scores. The effect of perceived stigma on caregiver burden was not ameliorated by whether the PWE was an adult or a child when only looking at these two independent variables. Instead, there was a slight suppression Table 1 Demographic characteristics of respondents (N = 136) and patients with epilepsy.a Variable Respondents Mean age (years) % male % White % married % bachelor's degree or higher Mean annual income category Patients with epilepsy Mean age (years) % male % adult (ages 21 and up) % in CBD study a
Mean or %
SD
Range
48 25.0 83.1 69.1 32.4 $40K–$50K
13
22 to 83
23 52.2 44.9 60.3
17
b$5K to N$200K
Respondents are familial caregivers of patients with epilepsy.
2 to 82
4
B. Hansen et al. / Epilepsy & Behavior 85 (2018) 1–6
Table 2 Carer's Assessment of Difficulty Index (CADI) item frequencies (N = 136).
Table 4 Four-nested OLS regression models predicting total CADI scores, (N = 136).
Individual item frequencies
A
B
C
D
I can feel helpless/not in control of the situation. My emotional well-being suffers. It can put a strain on family relationships. I can't relax because of worry about caring. I don't have enough private time for myself. I can't devote enough time to other family members. I feel angry about the situation. I feel guilty about the situation. It is physically tiring. The person I care for is immobile/has problems getting about. The person I care for doesn't always help as much as they could. My physical health has suffered. The person I care for needs a lot of help with personal care. My sleep is affected. The person I care for is incontinent. It restricts my social life/outside interests. I can't have a break or take a holiday. I can't see friends as often as I'd like. Professional workers don't appreciate the problems caregivers face. I don't get enough help from the health and social services. It causes financial difficulties. My standard of living has fallen. Relatives don't keep in touch as often as I'd like. Some family members don't help as much as they could. The person I care for sometimes manipulates me. The person I care for can demand too much of me. I no longer have a meaningful relationship with the person I care for. The person I care for doesn't always appreciate what I do. The behavior of the person I care for is a problem. There is no satisfaction to be gained from caring.
21 37 28 26 36 37 69 66 19 67 58 66 36 34 71 30 48 48 53
35 21 28 36 31 33 33 34 36 25 31 23 39 37 26 35 32 31 34
52 63 51 45 41 45 20 14 41 17 32 28 31 34 21 37 35 41 31
28 15 29 29 28 21 14 22 40 27 15 19 30 31 18 34 21 16 18
57 37 69 70 52 93 74 115
22 29 25 26 25 14 25 12
30 34 17 20 28 20 26 6
27 36 25 20 31 9 11 3
81 34 15 6 82 19 22 13 110 15 7 4
A = This statement does not apply to me. B = This statement applies to me and I find it: not stressful. C = This statement applies to me and I find it: stressful. D = This statement applies to me and I find it: very stressful.
effect, with the inclusion of age strengthening the relationship rather than mediating. When the age category was added to the model, the coefficient for stigma increased with both of the independent variables statistically significant (stigma, p b 0.001; age, p b 0.05.) After controlling for demographic variables in Model 3, the relationship between affiliate stigma and caregiver burden remained significant. Post hoc Wald test results indicated that compared with caregivers of children, caregivers of adults experienced significantly higher levels of burden. Age of the family member with epilepsy had a moderating effect on the relationship between total perceived stigma and burden levels, as shown in Model 4. Not only was the final model highly significant, the model as a whole also accounted for 14% more of the variation in burden scores than the first model where stigma was the sole independent variable. 4. Discussion In this study, the percentage of caregivers reporting perceived stigma is higher than those in other studies that show just over 50% Table 3 Frequency distribution for affiliate stigma scale items (N = 136). Individual items
People are uncomfortable around me People avoid me People treat me like an inferior person People are uncomfortable around my family member People avoid my family member People treat my family member like an inferior person a
Response categories were “yes” and “no”.
“Yes” responsesa f
%
69 48 30 101 68 57
50.7 35.3 22.1 74.3 50.0 47.1
Predictor
Total stigma Adult PWEa Caregiver age Family size White In CBDb study Income (log) ≥Bachelor deg Stigma ∗ age Constant F (df) R2 Nested F
Model 1
Model 2
Model 3
Model 4
β
SE
β
SE
β
SE
β
SE
4.85⁎⁎⁎
0.69
5.10⁎⁎⁎ 6.37⁎
0.69 3.01
4.66⁎⁎⁎ 7.77⁎ −0.07 0.74 −4.37 7.71⁎
0.69 3.65 0.15 1.04 4.12 3.09 1.63 3.48
3.50⁎⁎⁎ 0.56 −0.09 0.70 −4.32 8.13⁎⁎
0.90 5.06 0.15 0.99 4.07 3.06 1.61 3.44 1.35 18.72
−3.07 4.58 20.08 2.43 49.93⁎⁎⁎ (1, 134) 0.27 –
16.53 2.93 27.84⁎⁎⁎ (2, 133) 0.30 4.46⁎
48.81 18.78 8.95⁎⁎⁎ (8, 127) 0.36 2.17
−3.16 4.92 2.74⁎ 53.82 8.61⁎⁎⁎ (9, 126) 0.38 4.12⁎
⁎ p b 0.05. ⁎⁎ p b 0.01. ⁎⁎⁎ p b 0.001. a People with epilepsy. b Cannabidiol.
feeling stigmatized [1,2]. In reviewing the results from each of the individual stigma items, we noted that almost 75% reported that people are uncomfortable around their family member, and 51% said that others are uncomfortable around them. Thus, caregivers appeared to be acutely aware that they and their family members bear a stigma. This may be because of the intractable nature of seizures in this study and the difficulty in concealing the condition from others. One of the primary components of perceived stigma is the shame the affected person feels from the negative attitudes they perceive and the subsequent attempts to hide the stigmatizing condition as a way to manage their “spoiled” identity [11–13]. Epilepsy is typically considered a concealable stigmatized identity [33] but in the case of intractable epilepsy, this can be more difficult to achieve. This level of discomfort could lead caregivers to use avoidance and social isolation as ways of coping with the stress of anticipated stigma. For example, several respondents commented that they purposely do not expose their family members to the outside world in order to protect them from rude looks and comments. One mother said of her 22-year-old daughter, “She is always so happy. She doesn't know how mean and awful people can get and I'm not about to let her find out.” Another mother said, “We just stay home; don't go anywhere. We used to go to church but he got to where he would scream and yell and people would act like they wanted us to leave, like it was my fault or something.” These instances of isolating or hiding in an attempt to manage the stigma they expected to receive are similar with impression management techniques in other health conditions such as morbid obesity [34], HIV [35], and leprosy [36]. In these health conditions though, the affected person decides to hide from public view while in intractable epilepsy, the person making that decision to isolate is often a wellmeaning family member. There is evidence of stigma coaching leading to the concealment of an epilepsy diagnosis [23], but this can be difficult or nearly impossible with intractable epilepsy as seizures are more frequent and unpredictable. More than one family caregiver echoed one father who said, “Usually it's just easier to stay in when he's having a bad day. No one will stare at us at home.” The CADI is an excellent instrument for measuring the different domains of caregiving. The thirty items in the index cover an exhaustive list of activities, feelings, and situations that add to the stress in caring for someone with intractable epilepsy. Most burden studies in epilepsy use either a qualitative methodology with interviews or focus groups [2,20] or a checklist of health-related QOL issues [5,6], and the fact that stigma is not addressed in the CADI is a distinct advantage. Using this instrument makes it simpler to differentiate stigma from caregiving.
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The acknowledgment that caregiving is physically tiring was the item most reported as applicable to respondents. This is understandable because intractable epilepsy is frequently associated with intellectual and physical disabilities rendering some patients unable to independently perform activities of daily life such as walking, bathing, feeding oneself, and using the toilet [20]. As PWE grow out of childhood and into adulthood, they become larger and heavier, making it more difficult for caregivers to perform these activities. In addition, sleep is affected for many caregivers in this study similar with results from other studies. Reduced hours of sleep and fitful sleep due to worry about seizures are common occurrences [3,20]. One mother in this study reported that when her child is having a difficult day with seizures, she sleeps in a sleeping bag on the floor next to her daughter's bed in case she needs her. The CADI, in allowing respondents to choose the option “This statement does not apply to me,” offered us a chance to examine the levels of stress associated with each item relative to other caregivers experiencing the stressor. There were a few respondents who this category on every item in the index indicating no burden at all. One mother remarked, “[Son's name] is a gift from God. I was chosen to take care of him and it wouldn't do for me to act like that was a problem.” Another parent remarked, “It would be nice to not worry about buying the new wheelchair but God will provide.” While religiosity was not measured in this study, there was a strong thread of religious faith running through the spoken and written comments made by those surveyed. Results from this study showed that there is a positive relationship between affiliate stigma and perceived burden among caregivers in intractable epilepsy. Because previous studies did not typically differentiate between stigma and burden, it has been unclear how much of an effect perceived stigma might have on burden. This is the first study to show a very strong and statistically significant association. Other studies also did not include caregivers of both children and adults. Unfortunately, this aspect is overlooked in most studies of caregiving in epilepsy. In addition, the association between stigma and caregiver burden was stronger among caregivers of adult PWE compared with those looking after pediatric PWE, and that was after factoring in the age, race, family size, household income, and education level of the caregivers. It is surprising that the number of people in a household was not significantly associated with caregiver burden scores. Intuitively, it may seem that if there are more people in a home to take care of, the burden load would be greater for a primary caregiver or perhaps smaller families might not have others to share in their tasks and experience greater burden, but this did not seem to be the case. One interesting finding was that when the PWE were in the CBD study, their caregivers experienced more total burden. One possible explanation for this might be the high frequency of visits the PWE make to the research clinic for the study. Until patients are stable on a dosage of CBD, they are required to attend clinic every 2 weeks (uab.edu/cbd). For patients who are combative or nonambulatory or require significant assistance with personal care, this can be challenging for caregivers, especially those who drive for several hours. Because many of the caregivers were recruited from the CBD study, it is possible that the caregivers were experiencing high levels of stress while completing the survey. There is also a possibility that seizure frequencies and severity may vary between those in the CBD study and those not participating, but these data were not specifically collected. Some caregivers who are against using marijuana for medical (or any) purposes might have not considered participating in the study. These variations remain unclear and might be examined in future studies.
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is a need several caregivers expressed. Other items related to medical office environment were dim lighting, quiet areas to wait with few environmental triggers, and family style restrooms that accommodate large wheelchairs. Access to social services that are available in their area would be extremely helpful. Many caregivers noted that they have no idea if any services were available. However, they would find a list useful. Most neurologists are affiliated with hospitals which have on-staff medical social workers who could connect caregivers with agencies. Understanding the stress that caregivers of highly dependent patients are under may influence scheduling in order to reduce the time patients spend in waiting rooms, which is another need mentioned. The number one need stated by respondents in this study was respite care. Some even elaborated that they did not want babysitting services but trained professionals who were not afraid of epilepsy. Several caregivers explained that although there were a few day programs available in their counties for intellectually disabled citizens, almost none accepted adults with epilepsy, presumably because of fear of seizures. Some caregivers reported not having seen a movie in years because there was no one with whom to leave their child. The second most frequently cited need was a support group where caregivers of PWE could get together to talk and share experiences or resources. One respondent explained that even an online group would be helpful. Another frequently mentioned need was a website for caregivers that included links to access services and social agencies with help for families taking care of chronically ill relatives. As more research in affiliate stigma and caregiver burden in intractable epilepsy is conducted, more evidence will be available to inform grant proposals for services and implement policies to assist families. 4.2. Limitations Limitations of the study need to be discussed. With the use of a convenience sample and the study's cross-sectional nature, random sampling was not possible. A longitudinal study would have been more robust, as it would examine the dynamic variations in perceived levels of stigma and caregiver burden over time, but this was not possible within our study's constraints. Data concerning the severity and types of seizures patients experienced were not captured. This information would have been helpful in examining the differences between the group in the CBD study and those not enrolled. It is possible that those in the study suffered more debilitating epilepsy leading to higher levels of burden. Caregivers of patients with comorbid conditions such as intellectual disability and psychiatric comorbidities such as major depression were not excluded and this needs to be addressed in future studies. It is also possible that the group not enrolled in the CBD study were ineligible because of coexisting health or genetic issues. Future studies should include gathering seizure types, frequencies, and comorbid conditions. Recruitment should focus more outside a CBD study in order for results to be more generalizable. Another limitation is the fact that the survey was self-administered, which may introduce bias. A question might also be added to the survey examining the familial relationship of the caregiver to the PWE, i.e., parent, sibling, spouse, and child. There was also little variation in race and ethnicity with respondents being mostly White with no Hispanic or Latino participants. Using data from an original survey is also a limitation in that the survey has not been validated from repeated use in other studies. 4.3. Implications for further research
4.1. Clinical repercussions and suggested solutions Neurologists who treat PWE might use these results to assist in identifying concerns of caregivers. With limited time spent during office visits, the needs of caregivers might be overlooked. Adequate space in waiting rooms and examination rooms for wheelchairs and caregivers
This study is an important step in the research into stigma and caregiver burden in intractable epilepsy. As one of the first studies to provide evidence of affiliate stigma among caregivers in intractable epilepsy, it adds to the literature from which to expand future research. Repeating this study will provide opportunities for validation of the survey
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instrument. Other studies might utilize a mixed-methods approach, using the instrument as a quantitative measurement of affiliate stigma and caregiver burden and incorporate a few in-depth interviews or focus groups to flesh out the perceptions of stigma and to discuss the specific aspects of burden that are troubling. Particular emphasis in future studies might examine the psychological responses in affiliate stigma exhibited by caregivers, such as isolating behaviors and other coping mechanisms. Before this study, affiliate stigma in intractable epilepsy had not been examined. In fact, it has not been examined in many health-related conditions. Viewing epilepsy through this lens can provide motivation to study affiliate stigma in other disorders. Results from this study also add to the literature on caregiving in chronic illness and the stress process. Being able to control for comorbidities would be advantageous in that we could differentiate between the burden and stigma due to the epilepsy and those due to the comorbidity. We urge future studies to close this gap. 5. Conclusions Despite its limitations, this study extends past literature by providing a conceptual framework for studying affiliate stigma in intractable epilepsy. Differentiating the perception of stigma from the perception of burden, examining caregivers for both children and adults, and assessing caregiving activities across a stress continuum are unique to this study. In addition, using stigma as a possible explanation for perceived burden levels expands on the research into affiliate stigma. Further research is needed in examining the intersection of affiliate stigma and caregiver burden. Results from this study and respondents' comments may be incorporated into grant proposals exploring how best to meet the clinical and service needs in this population. Supplementary data to this article can be found online at https://doi. org/10.1016/j.yebeh.2018.05.028. Conflicts of interest There are no funding sources to report and no conflicts of interests for any of the authors. Acknowledgments This work was part of the doctoral thesis requirement of the first author. The authors wish to thank Mike Nolan for his insight into using the CADI as an assessment tool and for providing a copy of the original CADI handbook, Assessing the Needs of Family Carers: A Guide for Practitioners (1998), as this book was out of print and could no longer be purchased. Further appreciation goes to Pongkiat Kankirawatana, MD, for allowing us the opportunity to offer the survey to parents of his pediatric patients. References [1] Jacoby A, Austin JK. Social stigma for adults and children with epilepsy. Epilepsia 2007;48(Supplement 9):6–9. [2] VanStraten AF, Ng YT. What is the worst part of having epilepsy? A children's and parents' perspective. Pediatr Neurol 2012;47:431–5. [3] Gibson PA. Lennox–Gastaut syndrome: impact on the caregivers and families of patients. J Multidiscip Healthc 2014;7:441–8. [4] Nolan M, Grant G. Regular respite: an evaluation of a hospital rota bed scheme for elderly people from the Age Concern Institute of Gerontology. London: Ace Books; 1992. [5] Van Andel J, Zijlmans M, Fischer K, Leijten FSS. Quality of life of caregivers of patients with intractable epilepsy. Epilepsia 2009;50(5):1289–300.
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