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. Author manuscript; available in PMC: 2016 Oct 1.
Published in final edited form as: Epilepsia. 2015 Aug 20;56(10):1542–1550. doi: 10.1111/epi.13113

Genetic Causal Attribution of Epilepsy and its Implications for Felt Stigma

Maya Sabatello 3, Jo C Phelan 8, Dale C Hesdorffer 1,6, Sara Shostak 9, Jeff Goldsmith 7, Shawn T Sorge 1, Melodie R Winawer 1,2, Wendy K Chung 4,5, Ruth Ottman 1,2,6,10
PMCID: PMC4593736  NIHMSID: NIHMS708616  PMID: 26290354

Summary

Objective

Research in other disorders suggests that genetic causal attribution of epilepsy might be associated with increased stigma. We investigated this hypothesis in a unique sample of families containing multiple individuals with epilepsy.

Methods

181 people with epilepsy and 178 biological relatives without epilepsy completed a self-administered survey. In people with epilepsy, felt stigma was assessed through the Epilepsy Stigma Scale (ESS), scored 1 to 7 with higher scores indicating more stigma and >4 indicating some felt stigma. Felt stigma related to having epilepsy in the family was assessed through the Family Epilepsy Stigma Scale (FESS), created by replacing “epilepsy” with “epilepsy in my family” in each ESS item. Genetic attribution was assessed through participants’ perceptions of the (1) role of genetics in causing epilepsy in the family, (2) chance they had an epilepsy-related mutation, and (3) (in people with epilepsy) influence of genetics in causing their epilepsy.

Results

Among people with epilepsy, 22% met criteria for felt stigma (ESS score >4). Scores were increased among individuals who were aged ≥60 years, were unemployed, reported epilepsy-related discrimination, or had seizures within the last year or >100 seizures in their lifetime. Adjusting for other variables, ESS scores in people with epilepsy were significantly higher among those who perceived genetics played a “medium” or “big” role in causing epilepsy in the family than in others (3.4 vs. 2.7, p=0.025). Only 4% of relatives without epilepsy had felt stigma. Scores in relatives were unrelated to genetic attribution.

Significance

In these unusual families, predictors of felt stigma in individuals with epilepsy are similar to those in other studies, and stigma levels are low in relatives without epilepsy. Felt stigma may be increased in people with epilepsy who believe epilepsy in the family has a genetic cause, emphasizing the need for sensitive communication about genetics.

Keywords: genetics, epidemiology, survey, genetic attribution, stigma

Throughout history, epilepsy has been viewed as a source of shame and embarrassment, with cultural constructions of persons with epilepsy as violent, antisocial, or “possessed.”1 The consequences have been pervasive prejudice, social exclusion, and discrimination, which are today recognized as major, even predominant, contributors to the burden associated with epilepsy.2–5 Further, felt stigma, defined as perceived stigma (even in the absence of “enacted”6 stigma and discrimination) is common among persons with epilepsy. Some studies have reported that more than half of persons with epilepsy feel stigmatized because of their epilepsy.7,8 People reporting higher levels of stigma report reduced quality of life and increased anxiety, shame, low self-esteem, and depression.9 In addition, levels of felt stigma are associated with lower socioeconomic status, poor general health status, and reduced seizure control.3,6,9–11 Thus, to improve the quality of life of persons with epilepsy, not only improved medical management but also reduced stigma and increased public understanding are critical.

Research into the genetic origins of epilepsy holds great promise for development of new treatment options, and perhaps even methods to prevent onset of epilepsy. The number of genes identified as influencing risk has exploded,12 and genetic testing is increasingly viewed as integral to clinical care.13,14 However, findings in other stigmatized disorders provide cause for concern that the perception that epilepsy is a genetic disorder (“genetic causal attribution”) might be associated with increased stigma.15 Research on this possibility is important because of the strong current emphasis on genetic causes of epilepsy and persisting effects of stigma on individuals with epilepsy.

In the context of psychiatric disorders, studies of the general public have found associations between genetic attribution and increased associative stigma (i.e., stigma directed to family members of those with the disorder),16,17 desire for social distance, and perception of dangerousness.18–20 Findings have been consistent with expectations from genetic essentialism, which posits that viewing disorders as genetic makes them appear more fundamental to the person, exacerbating the sense of difference and making the disorders appear more severe and persistent.21 One study found that persons with serious psychiatric conditions who endorsed a genetic causal model were more likely to hold stigmatizing reactions, including stronger implicit guilt for their conditions and greater explicit fear towards other persons with psychiatric conditions.22 Another study of families with multiple individuals with bipolar disorder found that genetic attributions were associated with felt stigma among family members who did not have psychiatric conditions.23

Research suggests that the relations of genetic causal attribution to stigma are shaped by the social and medical context of different disorders,24 and these relations have not been well studied in epilepsy. In our previous qualitative study, individuals in families containing multiple individuals with epilepsy expressed concern that public views of epilepsy as a genetic disorder might lead to increased stigma and discrimination.25 Similarly, in another recent study of the experience of receiving results from genetic research on epilepsy, participants reported that stigma negatively impacted family communication.26

Given the findings in other stigmatized disorders, we would predict that levels of felt stigma would be increased among persons with epilepsy who attribute their condition to a genetic cause. Here, we investigated this hypothesis in a survey of the members of families containing multiple individuals with epilepsy who previously participated in genetic research. We assessed levels of felt stigma in individuals with epilepsy and their biological relatives without epilepsy, and examined associations with measures of genetic causal attribution, demographic variables, and (in persons with epilepsy) measures of epilepsy severity and reported history of discrimination (“enacted stigma”).6

Methods

Study sample

This study is part of a larger investigation of psychosocial outcomes and their relations with genetic attributions and actual genetic test results in families containing multiple individuals with epilepsy.27 The families were originally recruited to participate in the Epilepsy Family Study of Columbia University (EFSCU), a long-term investigation of the genetic influences on epilepsy that began in the mid-1980s as a familial aggregation study and evolved into a genetic linkage study.28 Families were eligible for the genetic linkage study if they contained either a sibling pair or ≥3 individuals with epilepsy of unknown cause. Recruitment was carried out throughout the U.S., by soliciting physician referrals and self-referrals in response to advertisements through the Epilepsy Foundation and a study web site. The families were ascertained from 1993–2006, and have been re-contacted periodically through newsletters and invitations to participate in additional studies.

Study design and data collection

Individuals were eligible for the current study if they previously participated in our genetic research either by being interviewed or donating a blood sample, were currently alive and aged 18–79 years, were able to complete a self-administered questionnaire in English, and had not previously refused contact for future research. Among 1274 participants in our previous studies, 345 were excluded because they did not meet these criteria, and 140 others were excluded from the current analysis because they were married-in to the families, leaving 789 individuals. These eligible individuals were asked to complete a self-administered questionnaire of approximately 30 minutes, either online through Survey Monkey (www.surveymonkey.com) or on paper. The Columbia University Medical Center Institutional Review Board approved the research protocols for the study.

As of October 21, 2014, we reached 564 (71%) eligible individuals by telephone to invite participation; 484 (86%) of those reached agreed to participate; and 359 (74%) of those who agreed completed the survey. The overall participation rate was 46% (359/789). Survey participation rates increased with advancing age (<40 years 35%, 40–59 years 46%, ≥60 years 57%, p < 0.001), and were higher in women than in men (50% vs. 40%) (p = 0.002), in college graduates compared to non-graduates (52% vs. 44%, p = 0.051), and in individuals with epilepsy than in those without epilepsy (51% vs. 42%, p = 0.007). Among individuals who completed the survey, 94% were white, non-Hispanic, 61% were women, 53% were college graduates, and 50% self-reported epilepsy. The mean age of all participants was 52 years (SEM 0.75). Survey participants were distributed in 104 families averaging 3.5 (range 1–18) participants per family and 4.2 (range 2–17) individuals with epilepsy per family.

Epilepsy history

For analyses related to stigma, we used self-reported data on epilepsy history rather than diagnoses from our original genetic study28 because an individual’s perception of whether he or she had epilepsy is most relevant for consideration of felt stigma, and in some cases, a long time interval (up to 20 years) had elapsed since our previous assessment. Individuals were classified as having epilepsy if they responded “yes” to either of two survey questions. The first question asked “Which of your biological relatives have had epilepsy or a seizure disorder?” followed by a list of relative types with “Yourself” at the top. The second question asked “Have you ever been told that you had epilepsy or a seizure disorder?” and was included in a later section of the survey that addressed respondents’ seizure histories in more detail. Among 178 individuals who self-reported epilepsy, seven appeared to have new onset epilepsy since our previous assessment, and nine others were not classified as having epilepsy in our genetic study because they had only febrile or acute symptomatic seizures or nonepileptic events. Conversely, five of the 181 individuals who did not self-report epilepsy had previously been classified as having epilepsy.

Measures of felt stigma

To assess felt stigma in people with epilepsy, we used the Epilepsy Stigma Scale (ESS), a previously validated 10-item scale that assesses respondents’ belief that epilepsy is negative and interferes with relationships (Table 1).9 Each item is scored from 1=“strongly disagree” to 7=“strongly agree,” with 4=“neither agree nor disagree.” To assess felt stigma related to having epilepsy in the family (in both individuals with epilepsy and family members without epilepsy), we modified the ESS to create a “Family Epilepsy Stigma Scale” (FESS) by replacing “epilepsy” with “epilepsy in my family” in each of the 10 items (Table 1). For each scale, an individual’s score was taken as the mean of all 10 items.

Table 1.

The Epilepsy Stigma Scale (ESS) and Family Epilepsy Stigma Scale (FESS): mean scores for overall scales, subscales, and individual items

Total Scale, Subscales, and Individual Itemsd People with epilepsy: ESS People with epilepsy: FESS Relatives without epilepsy: FESS p-valuec
Na x̄ SEMb Na χ̄ SEMb Na x̄ SEMb
Total Scale 154 2.9 0.13 174 2.7 0.12 165 1.9 0.09 <0.001
Self Subscale 154 2.7 0.14 174 2.4 0.12 165 1.5 0.08 <0.001
Others Subscale 154 3.0 0.13 174 2.9 0.12 165 2.1 0.10 <0.001
Individual items:
1. People who know that I have [epilepsy/[epilepsy in my family] treat me differently 160 3.0 0.15 176 2.7 0.15 170 2.2 0.15 0.086
2. It really doesn’t matter what I say to people about [my epilepsy/having epilepsy in my family], they usually have their minds made up 160 3.3 0.16 176 3.6 0.16 171 3.4 0.13 NS
3. I always have to prove myself because I have [epilepsy/ epilepsy in my family] 162 2.7 0.15 177 2.6 0.15 174 1.7 0.11 <0.001
4. Because of [my epilepsy/epilepsy in my family], I have problems developing intimate relationships 161 2.4 0.15 176 2.2 0.14 174 1.5 0.10 <0.001
5. In many people’s minds, having [epilepsy/epilepsy in my family] attaches a stigma or label to me 160 3.6 0.18 176 3.1 0.16 174 2.1 0.15 <0.001
6. I feel different from other adults because [of my epilepsy/I have epilepsy in my family] 161 3.2 0.16 176 2.9 0.16 176 1.9 0.14 <0.001
7. I feel embarrassed about [my epilepsy/having epilepsy in my family] 160 2.9 0.18 176 2.2 0.14 176 1.5 0.09 <0.001
8. I feel ashamed to tell others about [my epilepsy/having epilepsy in my family] 160 2.7 0.17 174 2.2 0.16 176 1.5 0.09 <0.001
9. I feel others are uncomfortable with me because [of my epilepsy/I have epilepsy in my family] 161 2.9 0.14 176 2.7 0.14 174 1.6 0.10 <0.001
10. I feel others would prefer not to be with me because [of my epilepsy/I have epilepsy in my family] 160 2.3 0.13 176 2.2 0.14 173 1.4 0.08 <0.001
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a

Ns vary slightly because of missing data. Ns for total scale and subscales restricted to individuals with non-missing values for all items.

b

SEM, standard error of the mean.

c

p-value from GEE model comparing FESS scores in individuals with epilepsy and relatives without epilepsy, after Bonferroni correction for 13 tests.

d

Self subscale: Items 4, 6, 7, 8; Others subscale: remaining items. In each item, alternative wording for ESS vs. FESS shown in brackets, with FESS wording italicized.

We noted that four of the 10 items in the original ESS and FESS (items 4, 6, 7, and 8; Table 1) relate to individuals’ internal feelings and the other six to their interpretation of how others feel about them. To capture possible differences in responses to these items, we created two subscales of the ESS and FESS: the “Self” subscale, defined as the mean value of items 4, 6, 7, and 8, and the “Others” subscale, defined as the mean of the remaining items. All of the scales had high reliability, as assessed by Cronbach’s alpha (ESS: Total 0.93, Self 0.86, Others 0.91; FESS: Total 0.92, Self 0.86, Others 0.88).

We examined several potential predictors of felt stigma: epilepsy history, age (<40, 40–59, ≥60 years), sex, education (college graduate vs. less), employment status (employed, unemployed, retired), number of family members with epilepsy (<4 vs. ≥4 as reported in the survey), and genetic causal attribution of epilepsy (assessed by questions described below). In people with epilepsy, we also examined the possible effects of epilepsy “severity” as reflected by survey questions about time since last seizure (<1 year vs. ≥1 year), and lifetime number of seizures (≤20, 21–100, >100). We also examined the relationship of ESS scores in people with epilepsy to reports of experiences of discrimination. To assess discrimination, participants were requested to respond true, false, or uncertain to six items about specific ways in which they could have been discriminated against (Table 4).

Table 4.

Self-reported discrimination and relationship to ESS scores in individuals with epilepsy

Discrimination items Nb Unadjusted Adjusteda
mean ESS SEMc p-valued mean ESS SEMc p-valued
I have been denied a job or promotion because of my epilepsy
 True 28 3.7 0.30 0.020 3.6 0.30 0.193
 False or uncertain 125 2.7 0.14 referent 2.9 0.14 referent
I have been refused housing because of my epilepsy
 True 2 2.9 0.60 NS 2.2 0.83 NS
 False or uncertain 150 2.9 0.13 referent 3.0 0.12 referent
I have been rejected from a social or work group because of my epilepsy
 True 12 4.4 0.51 0.017 4.2 0.51 0.065
 False or uncertain 139 2.8 0.13 referent 2.9 0.12 referent
I have been removed or rejected from a school, daycare, or other program because of my epilepsy
 True 8 3.9 0.58 0.486 3.7 0.53 NS
 False or uncertain 145 2.8 0.13 referent 3.0 0.13 referent
I have been harassed or victimized during a seizure.
 True 15 3.9 0.38 0.063 3.7 0.38 0.263
 False or uncertain 138 2.8 0.13 referent 2.9 0.12 referent
People have discriminated against me in other ways because of my epilepsy
 True 20 4.1 0.35 0.002 4.0 0.35 0.008
 False or uncertain 123 2.7 0.14 referent 2.9 0.12 referent
Any item
 True 45 3.6 0.21 <0.001 3.6 0.21 0.005
 False or uncertain 108 2.6 0.15 referent 2.7 0.14 referent
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a

Adjusted for age (<40, 40–59, ≥60 years) and employment (employed, unemployed, retired).

b

Ns vary slightly because of missing data

c

SEM, standard error of the mean

d

p-value from GEE model, after Bonferroni correction for 7 tests

Measures of Genetic Causal Attribution

Assessment of genetic attribution was based on three survey questions. The first question was focused on the family level, and asked, “In your opinion, how big a role has genetics had in causing the epilepsy in your family?” with possible responses: none, small, medium, or big. The second question focused on the individual level, and asked, “In your opinion, what do you think the chances are that you have a change or mutation in a gene that affects risk for epilepsy?” with possible responses: no, small, moderate, high chance, or don’t know. The third question (for individuals with epilepsy only) also focused on the individual level, and asked, “How much do you think each of the following influenced your risk of developing epilepsy? (Please answer what you think caused your epilepsy in the first place, rather than what you think might trigger your seizures.),” followed by a list of 14 items with “genetics or inheritance” in the list. Response options were: no influence, some influence, or strong influence.

Statistical analysis

We used paired t-tests and Pearson’s correlation analysis for within-individual comparisons of ESS and FESS scores in individuals with epilepsy, and scores from the Self and Others subscales of the ESS and FESS. To assess differences in FESS scores between individuals with epilepsy and relatives without epilepsy, we used generalized linear models with generalized estimating equations (GEE) to account for non-independence resulting from the inclusion of multiple individuals per family. We also used GEE models to examine the relationships of ESS and FESS scores to demographic and seizure severity variables, genetic attribution measures, and reported discrimination. To control for potential confounding in the analyses of these predictors, we carried out multivariate analyses using GEE models. Analyses of predictors were carried out separately within individuals with epilepsy and relatives without epilepsy, using as the outcome ESS scores in individuals with epilepsy and FESS scores in relatives without epilepsy.

To assess the proportion of individuals who experienced stigma, we defined “any stigma” as a mean score on the ESS or FESS greater than 4. This was based on the interpretation of 4 as “neither agree nor disagree,” suggesting that a mean score above this value indicated some agreement with the items, on average.

Results

Among people with epilepsy, overall mean scores were 2.9 on the ESS and 2.7 on the FESS. Fewer people with epilepsy completed the ESS than the FESS, because some participants skipped the questions in the epilepsy section of the survey (which contained the ESS questions and appeared later in the survey), despite having self-reported epilepsy. In paired analyses, ESS and FESS scores were highly correlated (r=0.80), but were significantly higher on the ESS than the FESS (p=0.013). Values on the Self and Others subscales were also significantly correlated for both the ESS (r=0.81) and FESS (r=0.80). However, for both scales, scores were significantly lower on the Self subscale than the Others subscale (Table 1: ESS 2.7 vs. 3.0, FESS 2.4 vs. 2.9, paired t-test p<0.001 for both scales).

Among people with epilepsy, mean values for individual questions in the ESS ranged from 2.3 to 3.6, with the highest value for item 5 (“In many people’s minds, having epilepsy attaches a stigma or label to me”) and the lowest value for item 10 (“I feel others would prefer not to be with me because of my epilepsy” (Table 1). For every item except #2 (“It really doesn’t matter what I say to people about my epilepsy, they usually have their minds made up”), the means were slightly higher on the ESS than the FESS.

Among relatives without epilepsy, the overall mean FESS score was 1.9, which was significantly lower than the mean FESS score in people with epilepsy (p<0.001). As in people with epilepsy, scores on the Self and Others subscales were significantly correlated (r=0.76), but were significantly lower on the Self than the Others subscale (1.5 vs. 2.1, p<0.001, paired t-test). Mean values for all but two individual items (#1 and #2) were also significantly lower in relatives without epilepsy than in individuals with epilepsy, after Bonferroni correction for 13 tests (Table 1).

Twenty-two percent of people with epilepsy met our criterion for any stigma (Score >4 on ESS: 34/154 [22.1%], FESS: 28/174 [21.8%]). However, only 4.2% (7/165) of relatives without epilepsy met this criterion, based on a score >4 on the FESS.

Predictors of felt stigma

In people with epilepsy, ESS scores were significantly higher in individuals aged ≥60 years than in those aged <40, but were similar in those aged 40–59 and <40 years (Table 2). Employment status was a strong predictor of ESS scores, with significantly lower scores in employed individuals than in either unemployed or retired individuals. Mean scores were also significantly higher in individuals who reported a history of >100 seizures in their lifetime or who had seizures within the past year than in others. ESS scores were not associated with sex, education, or number of relatives with epilepsy. Among the three measures of genetic attribution, only one was associated with ESS scores in people with epilepsy: scores were higher in individuals with epilepsy who responded that genetics played a “medium” or “big” role in causing epilepsy in their families than in others (p=0.041, Table 2).

Table 2.

Predictors of felt stigma in individuals with epilepsy

Predictors Nb Unadjusted Adjusteda
mean ESS SEMc p-valued mean ESS SEMc p-valued
Age (years)
 <40 41 2.7 0.22 referent 2.6 0.27 referent
 40–59 67 2.7 0.17 0.847 2.9 0.22 0.124
 ≥60 46 3.3 0.24 0.037 3.6 0.30 0.013
Sex
 Women 96 2.8 0.16 0.711 2.8 0.19 0.102
 Men 58 2.9 0.20 referent 3.2 0.21 referent
Education
 < College graduate 73 3.0 0.20 referent 3.0 0.19 referent
 College graduate 80 2.8 0.17 0.607 3.1 0.20 0.666
Employed
 Employed 80 2.5 0.17 referent 2.8 0.25 referent
 Unemployed 45 3.4 0.23 0.002 3.6 0.29 0.009
 Retired 27 3.2 0.22 0.010 2.7 0.33 0.862
Number of relatives with epilepsy
 ≥4 75 2.9 0.18 0.751 2.9 0.23 0.339
 <4 74 2.8 0.18 referent 3.2 0.17 referent
Chance you have a change or mutation in a gene that affects risk for epilepsy
 Moderate/high 88 2.9 0.17 0.578 3.0 0.22 0.799
 None/small/DK 65 2.8 0.20 referent 3.1 0.20 referent
Role of genetics in causing epilepsy in your family
 Medium/big 122 3.0 0.15 0.041 3.4 0.17 0.025
 No/small 31 2.5 0.21 referent 2.7 0.27 referent
Influence of genetics in causing your epilepsy
 strong 89 2.9 0.18 0.993 3.1 0.21 0.805
 weak/none 58 2.9 0.18 referent 3.0 0.20 referent
Lifetime number of seizures
 ≤ 20 70 2.6 0.18 referent 2.7 0.22 referent
 21–100 29 3.2 0.32 0.088 3.4 0.27 0.020
 >100 31 3.5 0.28 0.007 3.3 0.27 0.054
 DK/missing 24 2.5 0.21 0.680 2.7 0.30 0.921
Time since last seizure
 ≥1 years 96 2.6 0.14 referent 2.6 0.17 referent
 <1 year 39 3.5 0.24 0.002 3.2 0.23 0.044
 unknown 14 3.2 0.43 0.159 3.3 0.32 0.020
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a

Adjusted for all variables in table.

b

Ns vary slightly because of missing data

c

SEM, standard error of the mean

d

p-value from GEE model

To evaluate and control for potential confounding, we performed a multivariate analysis of ESS scores in individuals with epilepsy, including all variables in the model (Table 2). The results confirmed significantly higher scores in individuals aged ≥60 years than in those aged <40 years, and in unemployed vs. employed individuals. As expected, the difference between retired and employed individuals disappeared after controlling for age. The higher scores in individuals with >100 seizures or seizures during the last year persisted, though p-values increased somewhat compared with the bivariate analysis. The p-value for “role of genetics in causing epilepsy in your family” declined to 0.025 (vs. 0.041 in the bivariate analysis). On further analysis, we found that this decline was explained by negative confounding with employment. Individuals who responded there was a “medium/big” role of genetics in causing epilepsy in the family were more likely than others to be employed; hence adjustment for employment resulted in higher ESS scores and a lower p-value.

In relatives without epilepsy, FESS scores were significantly higher in unemployed than employed individuals, but were not associated with any other demographic variable or either measure of genetic attribution (Table 3).

Table 3.

Predictors of felt stigma related to having epilepsy in the family in biological relatives without epilepsy

Predictors Nb Unadjusted Adjusteda
mean FESS SEMc p-valued mean FESS SEMc p-valued
Age (years)
 <40 38 1.9 0.17 referent 2.0 0.21 referent
 40–59 75 1.9 0.12 0.968 2.0 0.17 0.720
 ≥60 52 1.9 0.18 0.929 2.3 0.26 0.378
Sex
 Women 102 1.9 0.12 0.532 2.0 0.17 0.451
 Men 63 2.0 0.13 referent 2.2 0.17 referent
Education
 < College graduate 73 1.9 0.13 0.614 2.1 0.14 referent
 College graduate 91 1.9 0.11 referent 2.1 0.20 0.708
Employed
 Employed 113 1.8 0.08 referent 1.9 0.12 referent
 Unemployed 20 2.4 0.28 0.024 2.7 0.35 0.013
 Retired 29 1.8 0.22 0.893 1.7 0.29 0.601
Number of relatives with epilepsy
 ≥4 61 1.9 0.12 0.915 2.1 0.18 0.617
 <4 97 1.9 0.13 referent 2.2 0.17 referent
Chance you have a change or mutation in a gene that affects risk for epilepsy
 Moderate/high 62 2.1 0.16 0.088 2.2 0.19 0.291
 None/small/DK 102 1.8 0.11 referent 2.0 0.16 referent
Role of genetics in causing epilepsy in your family
 Medium/big 111 2.0 0.11 0.544 2.1 0.15 0.856
 No/small 47 1.9 0.17 referent 2.1 0.22 referent
Open in a new tab
a

Adjusted for all variables in table.

b

Ns vary slightly because of missing data

c

SEM, standard error of the mean

d

p-value from GEE model

Stigma and discrimination

Among people with epilepsy, 29% (45/153) responded “true” to any discrimination item (Table 4). The types of discrimination most frequently reported were denial of a job or promotion because of epilepsy (18.3%), being harassed or victimized during a seizure (9.8%), and rejection from social or work group because of epilepsy (7.9%). In addition, 14% of individuals reported that people had discriminated against them in other ways, providing descriptions relating to health insurance coverage, job-related discrimination, and forms of social exclusion (“I was not allowed to play sports,” “people avoided me”).

ESS scores were higher among people with epilepsy who reported any type of discrimination than among those who did not (3.6 vs. 2.6) and this difference persisted after adjustment for age and employment status (Table 4). The mean ESS score was highest among individuals who reported having been rejected from a social or work group: 4.4. The proportion of individuals who met the criterion for any stigma (ESS score >4) was 40% (18/45) among individuals reporting any form of discrimination, compared with 15% (16/108) among those not reporting discrimination.

Discussion

This study extends the literature on epilepsy-related stigma in three ways: by exploring the impact on felt stigma of attribution of epilepsy to a genetic cause; by assessing felt stigma in both people with epilepsy and their relatives without epilepsy; and by distinguishing between subtypes of felt stigma (assessed by the Self vs. Others subscales).

Overall, 22% of people with epilepsy in our study met the criterion for any felt stigma (score >4), whereas only 4% of relatives without epilepsy did. Stigma scores in both people with epilepsy and their relatives were significantly lower on the Self subscale than the Others subscale. One useful categorization of stigma distinguishes among internalized stigma (feelings, thoughts, and beliefs within the self); interpersonal stigma (feelings of isolation and exclusionary behaviors in interaction with others), and institutional stigma (indirect, impersonal, expression in policy-level decision-making).29 In this categorization, our observation of lower stigma scores on the Self vs. Other subscales may reflect less internalized than interpersonal stigma among study participants.

Most research on the effects of geneticization on family members of people with stigmatized disorders examines views of the public toward the relatives.16,17 In contrast, we examined felt stigma from the point of view of the family members themselves. We are aware of only one other study that has addressed this question in epilepsy: a study of mothers of children with epilepsy in Zambia, which found that 20% of the mothers felt stigmatized because of the child’s epilepsy.30 In the markedly different context of our study, the results are reassuring: levels of stigma were low among relatives without epilepsy.

Consistent with previous studies,9,31 we found that employment status and severity of epilepsy (more than 100 lifetime seizures or seizures within the last year) were strongly associated with felt stigma in people with epilepsy. Neither sex, education, nor number of relatives with epilepsy was significantly associated with felt stigma in either people with epilepsy or relatives without epilepsy (Tables 2 and 3).

Felt stigma was strongly associated with reported experiences of discrimination (“enacted stigma” or “institutional stigma”). Because experiences of discrimination were self-reported, the causal direction of this association is unclear. Experiences of discrimination may have led to feelings of stigmatization in some participants, or alternatively, participants who felt stigmatized may have interpreted some experiences as discrimination that could have been interpreted differently. However, contrary to the historical distinction between “felt” and “enacted” stigma,5 this strong association between felt stigma and reported discrimination supports recent reconceptualizations of stigma as including and overlapping with discrimination.32 These findings are also in line with the notion of stigma as moral experience that reflects what is “most at stake for people in a local world.”33

Although 29% of our participants responded “true” to any discrimination item, most discrimination items were reported by fewer than 10% of participants (Table 4). This may reflect increasing social acceptance of people with epilepsy,4 which would be consistent with our finding of higher stigma scores in individuals aged ≥60 years than in younger individuals.

Federal laws prohibiting discrimination on the basis of disabilities may have had some impact in reducing reports of discrimination in our sample. For example, the low frequency of reported rejection from a school, daycare, or other program because of epilepsy (5.2%), may be related to the Individuals with Disabilities Education Act (2004). On the other hand, the most frequently reported type of discrimination was denial of job or promotion because of epilepsy (18.3%), and some participants described “other ways” of being discriminated against that included health insurance and employment-based discrimination. Although some incidents of discrimination in healthcare may have occurred before the adoption of the Patient Protection and Affordable Care Act (2010), almost 25 years have passed since the adoption of the Americans with Disabilities Act (ADA) in 1990, aimed at improving employment opportunities for persons with disabilities. Thus, our findings reinforce the conclusions of other studies that stigma and discrimination in employment settings persist for people with epilepsy4,34 and hint at the limits of laws in transforming social perceptions about epilepsy.

One of the three measures of genetic attribution we examined -- the perception that genetics played a “medium” or big” role in causing epilepsy in the family -- was significantly associated with felt stigma in people with epilepsy. This association was strengthened after control for other variables associated with stigma in people with epilepsy, suggesting it was not due to confounding with those variables. Two issues raise questions about this finding, however. First, the results are not consistent across our three measures of genetic attribution. We have not done any cognitive testing of these items, and hence it remains somewhat unclear how they were interpreted by respondents. This question was the only one of the three that focused on genetics at the family (as opposed to individual) level. In the families we studied, felt stigma may be more closely related to family concerns (e.g., worry or guilt about transmission of susceptibility to an offspring) than to the perception of a genetic cause at the individual level. Second, this item was not associated with felt stigma in relatives without epilepsy. Although stigma levels were low in the relatives and associations could differ, it would have been reassuring to observe the same finding in both groups. Nevertheless, this result suggests that our hypothesis that genetic attribution is associated with felt stigma may be correct.

ESS scores in people with epilepsy were lower in our study (mean 2.9; Table 1) than in other studies that have used the ESS, which had means ranging from 3.2 to 4.9.3,9,31,35–37 Several factors may contribute to this difference. First, in other studies most participants had seizures within the past year,9,35,38 whereas in our study only 26% did (Table 1). Second, participants in our study had relatively high socioeconomic status (52% college graduates), which is associated with reduced levels of stigma.3 The mean ESS score in our study was similar to that among participants with higher socioeconomic status in a recent study.3 Third, our sample was predominantly white, non-Hispanic, and higher stigma scores have been found in studies with greater proportions of racial minorities.3,36,38 Finally, participants in our study may be unusually well informed about epilepsy because many were recruited through the Epilepsy Foundation, which strongly advocates for education and support for people with epilepsy and their family members. A recent study found that levels of felt stigma were increased in people with epilepsy who were less informed and less treatment adherent.37

Our study has several limitations. First, we assessed felt stigma in relation to genetic attribution of epilepsy rather than examining participants’ reactions to receiving actual genetic findings. Future studies should examine the positive and negative psychosocial impacts of receiving results from genetic testing,14,15 and data collection to address this question is ongoing in our study. Second, our sample has unique characteristics that reduce generalizability to all people with epilepsy. It is restricted to members of unusual families containing multiple people with epilepsy, and these individuals expressed a strong interest in genetics by virtue of having participated in genetic research. Although such families are uncommon,39 their members are arguably important stakeholders for assessing felt stigma related to genetics, because many perceive (likely correctly) that genetics plays an important role in causing epilepsy in their families. Our sample is also ethnically homogeneous and has an unusually high level of education, which probably led to reduced levels of felt stigma. Finally, many participants in our study do not have severe forms of epilepsy – 45% reported ≤20 seizures in their lifetimes, and 64% had not had seizures within the last year.

Despite these limitations, our findings confirm that stigma remains a concern for people with epilepsy.31 The association of stigma with one measure of genetic attribution in our study emphasizes the need for sensitive communication about genetic influences on epilepsy, particularly in families such as those studied here.14 However, more research is needed on the impact of genetic attribution – and receipt of genetic test results – on felt stigma in the wider population of people with epilepsy and their relatives, as well as on epilepsy-related stigma in the general public.

Key points.

  • In a unique sample of families containing multiple individuals with epilepsy, 22% of people with epilepsy had felt stigma.

  • Only 4% of the biological relatives without epilepsy in these families had felt stigma.

  • In people with epilepsy, felt stigma was associated with age ≥60 years, unemployment, greater epilepsy severity, and reported discrimination.

  • Felt stigma was increased in people with epilepsy who believed that genetics played a medium or big role in causing epilepsy in their families.

Acknowledgments

This research was supported by NIH grants R01 NS078419 and P50 HG007257. We are grateful to the families who generously donated their time to participate in this research.

Footnotes

Disclosure

None of the authors has any conflict of interest to disclose. We confirm that we have read the Journal’s position on issues involved in ethical publication and affirm that this report is consistent with those guidelines.

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