Maladaptive emotional regulation in patients diagnosed with psychogenic non-epileptic seizures (PNES) compared with healthy volunteers

Journal Pre-proof Maladaptive emotional regulation in patients diagnosed with psychogenic non-epileptic seizures (PNES) compared with healthy volunteers ´ ´ Lucia Hreˇskova, ´ Zdenek ˇ Vojtech, ˇ ´ Lenka Kramsk a, David Kramsk´ y, Lorna Myers

PII:

S1059-1311(20)30050-9

DOI:

https://doi.org/10.1016/j.seizure.2020.02.009

Reference:

YSEIZ 3662

To appear in:

Seizure: European Journal of Epilepsy

Received Date:

19 December 2019

Revised Date:

8 February 2020

Accepted Date:

12 February 2020

´ ˇ Z, Kramsk´ ´ Please cite this article as: Kramsk a´ L, Hreˇskova´ L, Vojtech y D, Myers L, Maladaptive emotional regulation in patients diagnosed with psychogenic non-epileptic seizures (PNES) compared with healthy volunteers, Seizure: European Journal of Epilepsy (2020), doi: https://doi.org/10.1016/j.seizure.2020.02.009

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Maladaptive emotional regulation in patients diagnosed with psychogenic non-epileptic seizures (PNES) compared with healthy volunteers

1Department 2Epilepsy 3Charles

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Lenka Krámská1,2,3*, Lucia Hrešková1, Zdeněk Vojtěch2, David Krámský3, Lorna Myers4

of Clinical Psychology, Na Homolce Hospital, Prague, Czech Republic

Center, Na Homolce Hospital, Prague, Czech Republic

University in Prague, Czech Republic

Regional Epilepsy Group, New York, United States of America

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4Northeast

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Supported by Ministry of Health of the Czech Republic, grant MH CZ and DRO (NHH, 193001)

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IG193001

*Corresponding author: Lenka Krámská, Ph.D.; Department of Clinical Psychology, Epilepsy

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Center, Na Homolce Hospital, [email protected]; tel.+420 257272593

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Highlights This study compares Czech patients with PNES to healthy controls on emotional regulation Patients with PNES had significant emotion regulation impairments compared to controls. Patients with PNES exhibited a lack of awareness or inattention to emotional responses. These findings are significant for the design of treatment approaches for those with PNES.

ABSTRACT Purpose: The term PNES refers to a conversion disorder that mimics epileptic seizures but has a psychological etiology. Recent studies report that in patients with PNES, there is reduced understanding of emotions, impulse control difficulties, and limited access to emotional regulation strategies. The aim of this study was to compare patients diagnosed with PNES with healthy volunteers on the presence of maladaptive emotional regulation. Method: Patients (N=64 F:M 52:12; mean age 35.5 years; duration  2 years) were assessed while inpatients at the Epilepsy Center, Na Homolce Hospital, Prague. PNES diagnosis was based on normal EEG findings, habitual seizure capture, suggestive seizure provocation,

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neuropsychological assessment and patients´ history. The clinical sample was compared with healthy volunteers (N=64 F:M 52:12; mean age 35.8 years). The presence of maladaptive emotional regulation was assessed through the Czech research version of the ASQ and DERS. Results: Compared with a healthy sample, patients with PNES had greater emotion regulation

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impairments across nearly all dimensions of the DERS - Nonacceptance=17.0 (M=14.5), p=0.006, Goals=16.0 (M=11.5), p<0.001, Impulse control=13.8 (M=11.5), p=0.005,

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Awareness=16.4 (M=15.3), p=0.183, Strategies=17.9 (M=13.0), p<0.001, Clarity=11.8 (M=9.5), p<0.001 and the total score of the DERS = 92.9 (M=75.3), p<0.001. Similar results were found

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in ASQ questionnaire scales - Concealing=24.5 (M=21.0), p=0.002, and Adjusting=20.9 (M=22.8), p=0.076 but not on the Tolerating Scale=14.5 (M=14.7), p=0.873. Conclusion: Our results highlight that maladaptive emotional regulation is a key psychological

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mechanism in PNES. Emotional dysregulation may represent an important target when designing psychoeducational and psychotherapeutic approaches for patients with PNES.

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Key words: Psychogenic non-epileptic seizures, emotional regulation, DERS, ASQ.

1. Introduction Psychogenic nonepileptic seizures (PNES) are involuntary experiential and behavioral responses to internal or external triggers that superficially resemble epileptic seizures but that are not associated with abnormal electroencephalographic electrical activity [1]. PNES are classified in the Diagnostic and Statistical Manual of Mental Disorder Fifth Edition (DSM-5) as a conversion disorder (or functional neurological symptom disorder) in the category of Somatic Symptom and Related Disorders [2]. Although the incidence of PNES in the general population is low with an approximate

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33 per every 100,000, compared to 780 per every 100,000 for epileptic seizures, PNES are a significant health problem generating high costs to the patient, the health system, and society. In fact, the health-related economic impact of PNES may be similar to the that of epilepsy) [3]. Most studies of patients with PNES in comparison to patients with epileptic seizures and

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healthy samples have found a higher prevalence of abuse, trauma, and life stresses in those with PNES [4]. Clear trends include the association of PNES with significantly more negative life

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events, a tendency to perceive events as substantially more stressful, significantly more dysfunctional cognitions and ruminations about stress, more stress-related disease, more

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reported social pressures, more health-anxiety (in males) and less perceived parental care (insecure or disorganized attachment). The most significant associated between PNES and trauma appears to be a family environment that fosters attachment trauma and produces

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tendencies toward somatization [5].

Among several etiological models for conversion disorder of paroxysmal type, one model conceives of PNES as deriving from two main categories of psychological conflicts:

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posttraumatic and developmental [6]. Posttraumatic PNES emerge as a maladaptive response to prior psychological or physical trauma(s). In this scenario, PNES reflects an automatic “cutoff

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phenomenon” in response to cued or spontaneous intrusions into the consciousness of unspeakable memories. On the other hand, developmental PNES is thought to develop in response to struggles coping with complex life tasks and milestones along the patient’s continuum of psychosocial development, which can occur in an environment of emotional deprivation. Having endured these difficult circumstances, many patients with PNES demonstrate an avoidant style of coping characterized with denial/avoidance of perceived threat and reluctance to confront problems directly (i.e., focusing on the physicality of

symptoms rather than emotional components) [6]. Emotional regulation is a mechanism that allows the person to respond to internal and external experiences with a range of emotions that are socially acceptable and effective. Through emotion regulation, automatic and effortful processes are employed to alter aspects of emotional experience or behavior. Emotion management is implied, if not stated explicitly as a contributing factor, in most theories of PNESs and most theories of conversion or dissociation [7]. Emotional dysregulation plays a role in various mental disorders and psychopathology. Limited findings exist on how patients with PNES process and understand emotional information. Increasingly, there are cross cultural studies of PNES emerging that are contributing to the

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field’s understanding of how intricately a person's cultural background and socially reinforced modes of expression influence body conception and corporeal manifestations. The present study is unique in that it is the first to examine emotional regulation profiles and affective responses to emotional situations in persons with PNES and healthy volunteers from the Czech

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would be higher than those of healthy subjects.

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Republic. It was hypothesized that the ASQ and DERS scores of patients diagnosed with PNES

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2. Materials and methods 2.1. Study design and target population

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A comprehensive neuropsychological evaluation was conducted on 64 patients (N=64 F:M 52:12; mean age 35.5 years, MIN:MAX 18:71) diagnosed with PNES during 2017-2018 who underwent video EEG monitoring at Epilepsy Center, Na Homolce Hospital in Prague. PNES diagnosis was based on normal EEG findings, habitual seizure capture, suggestive seizure provocation, patients´ history, and neuropsychological assessment. The clinical sample was compared to healthy volunteers (N=64 F:M 52:12; mean age 35.8 years, SD MIN:MAX 19:71), matched according to gender, age, and education. Volunteers were recruited through occasional voluntary selection for the purposes of this study. They signed informed consent.

Neurological examination All patients were admitted to an epilepsy monitoring unit where a detailed medical history was obtained, and neurological examination by a certified epileptologist (Z.V.) was performed.

During hospitalization, patients underwent long-term video-EEG monitoring, routine laboratory tests, and high-resolution magnetic resonance imaging. The length of stay depended on the time during which spontaneous diagnostic PNES was recorded. In patients in whom no seizure was captured, suggestive induction of their habitual seizure was performed at the end of their stay (usually a week). Neuropsychological examination Cognitive performance, personality assessment, mood (not included in this study) were tested with a battery of standardized neuropsychological tests (RBANS, MMPI-II, BDI-II, BAI, PBI, ROR)

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and were performed by a licensed clinical neuropsychologist (L.K.). The study focused on the presence of maladaptive emotional regulation using the Czech research version [8] of Affective Style Questionnaire (ASQ) and Difficulties in Emotion Regulation Scale (DERS). Patients were tested during two sessions and tests were administered in the same order. Each session took 2

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hours on average.

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2.2. Psychometric measures

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Affective Style Questionnaire (ASQ) is a 20-item instrument to measure individual differences in emotion regulation. Affective style is an individual variable that refers to tendencies for regulating emotions. The ASQ identifies three general strategies to manage emotions: some

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strategies are aimed at re-adjusting affect to adapt successfully to situational demands; other strategies are intended to conceal or suppress affect; and a third approach is to tolerate and accept emotions, including unwanted and aversive reactions. A factor analysis supported the

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following three factors: habitual attempts to conceal or suppress affect (Concealing subscale; 8 items), a general ability to manage, adjust, and work with emotions as needed (Adjusting

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subscale; 7 items), and an accepting and tolerant attitude toward emotions (Tolerating subscale; 5 items). The scale has exhibited satisfactory internal consistency. Findings were cross-validated and results of psychometric analyses were replicated [9]. The Difficulties in Emotion Regulation Scale (DERS) is a widely used self-report measure of subjective emotional management ability (Gratz and Roemer, 2004). The DERS is a 36-item selfreport measure scored on a 5-point Likert-type scale. Respondents indicate the frequency with which statements such as “I am clear about my feelings” apply to themselves. The items from

the DERS reflect difficulties within the following dimensions of emotion regulation: “(a) awareness and understanding of emotions; (b) acceptance of emotions; (c) the ability to engage in goal-directed behavior, and refrain from impulsive behavior, when experiencing negative emotions; and (d) access to emotion regulation strategies perceived as effective”. The DERS has strong internal consistency (α = .93). All the DERS subscales (computed from the six factors obtained in the factor analysis) also have adequate internal consistency, with Cronbach’s α>.80 for each subscale [10].

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2.3. Data analysis Statistical data analysis was performed in the SPSS statistical program from IBM, version 20.

For the assessment of normality, the Kolmogorov-Smirnov test was used, which is sensitive to any violation of a hypothesis of normality. To analyze the differences in scales distribution in

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both groups, the Welch modification of the t-test was used, which is sensitive to a possible different variation in both groups. This parametric test is adequate given the sufficient range of

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both samples - the adequacy is guaranteed by the central limit theorem.

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2.4. Ethics

All data collection, storage and processing were done in compliance with the Helsinki Declaration. All patients signed informed consent and study was approved by the Ethical

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Committee of the Na Homolce Hospital. Study was supported by Ministry of Health of the Czech Republic, grant MH CZ and DRO (NHH, 193001) IG193001. The authors declare that

3. Results

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there is no conflict of interest.

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There were 64 patients with PNES (52 F: 12 M) with 12.5 mean years of education

(SD=1.9, MED=13.0). Nineteen percent of patients were students, 46.8% of patients were unemployed with social support, 4.7% were on the maternity leave, 1.6% of patients were retired and 28% of patients were actively working. The percent of single patients was 56.3%, 32.8% were married, 9.4% were divorced, and 1.6% lived with partner. Healthy volunteers involved 52 Females and 12 Males with 12.9 years of education (SD=1.7, MED=13.0). There were 3.1% students, 90.6 % were employed, 3.1% were on the

maternity leave, 1.6% had partial social support and 1.6% were retired. The percent of single individuals was 26.6%, 57.8 % were married, 4.7% were divorced, 3.1% were widowed and 7.8 % lived with a partner. With regard to the psychometric measures of emotional regulation, compared to healthy volunteers, patients with PNES had greater emotion regulation difficulties across almost all dimensions of the ASQ and DERS questionnaires. Results of these psychological instruments are presented in Table 1 and 2.

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4. Discussion To the best of our knowledge this is the first study that compares Czech patients with PNES to Czech healthy controls on measures of emotional regulation. Findings revealed that on a group level, there was a higher level of emotional dysregulation in patients diagnosed with

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PNES compared to healthy probands on two different questionnaires (ASQ and DERS).

Studies describing the regulation of emotions confirm an increased tendency to control

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emotional expression in patients diagnosed with PNES compared to a control group of healthy volunteers [11]. Patients diagnosed with PNES often suppress or hide their emotional reactions,

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not only to others but also even to themselves. This maladaptive affective strategy is theorized to dissociate strong emotions from the conscious part of the human psyche for defensive purposes. Managing emotions in this manner can result in passive or aggressive forms of self-

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communication and with others, further increasing PNES frequency and emotional distress [12]. In the present study with individuals from the Czech Republic, patients diagnosed with PNES scored significantly higher in the ASQ subscale Concealing compared to healthy subjects.

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This replicates other English-speaking studies that have found patients with PNES tend to suppress emotions [11].

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Furthermore, the present patient sample scored lower than healthy probands on the

Adjusting subscale of the ASQ, which is characterized by modulation of emotional experience and manifestations in accordance with the qualities of a particular context. The ability to effectively modulate emotional experiences requires a capacity for introspection, selfknowledge, and kindness to oneself because only such an attitude makes it possible to look at the experience from a different perspective. Previous research has shown that although most PNES patients are aware of stressful situations in their lives, difficulties recognizing the specifics

of their emotions may represent an obstacle in realizing that these stressors affect their seizures [12]. However, in the ASQ Tolerating subscale, which includes aspects of accepting strong emotional experience and sensitivity to emotional awareness [13, 14], patients with PNES scored lower than healthy probands but this difference was not significant. Patients diagnosed with PNES scored in the Nonacceptance subscale of the DERS higher than healthy probands. Studies [14] confirm the tendency of patients diagnosed with PNES to have a negative secondary assessment of primary emotional experiences. The defense process of dissociation tends to proceed unconsciously and chronically, even under conditions where

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human survival is not in real danger [15]. It is theorized that patients diagnosed with PNES can tend to confuse common stress with life-threatening circumstances and their subsequent reactions further confuse themselves and the people around them. This can result in the repeated occurrence of dissociative seizures in all sorts of situations that are, interpreted as

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perilous, on a subconscious level.

In the Goals subscale of the DERS, PNES patients scored significantly higher than healthy

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subjects. This subscale measures difficulty concentrating and/or accomplishing tasks when experiencing negative emotions. In PNES, patients frequently have other psychiatric

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comorbidities including affective and anxious disorders which could clearly impact their cognitive efficiency.

It is believed that patients diagnosed with PNES have difficulty maintaining impulse

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control while experiencing negative affections. Studies focused on the psychotherapeutic treatment of patients diagnosed with PNES [16, 17] emphasize the importance of mediating

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patient's insight into how negative conditions can be converted to productive outcomes, which in turn helps prevent seizures or other negative symptoms (such as headaches). Although

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patients diagnosed with PNES in the present study scored higher on the DERS Impulse Control subscale than healthy subjects, this difference was non-significant. In the DERS Awareness subscale, the patient sample scored higher than healthy

probands. This subscale reflects a lack of awareness or inattention to emotional responses. This is consistent with other studies [18, 19] that have reported that the identification and expression of emotional experiences are impaired in patients diagnosed with PNES as compared to a healthy population. Emotional unawareness or alexithymia results in the patient

finding him/herself facing difficulties without an emotional compass which can obviously further complicate their situation. In the DERS Strategies subscale, the PNES patients in our sample had a significantly higher score than healthy subjects. Clinicians hypothesize that available regulatory mechanisms for downregulating negative affect are often weak in profiles of patients diagnosed with PNES. Patients diagnosed with PNES tend to experience helplessness, reduced self-esteem, and confidence in their abilities [20]. One final finding in this study was that the patients diagnosed with PNES scored significantly higher scores than healthy subjects on the DERS – Clarity subscale. This is

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consistent with reports that patients diagnosed with PNES who report a lower level of difficulty regulating emotions may also exhibit emotional avoidance [21]. Yet again, navigating life without an emotional compass is highly problematic.

These results build on recent research outcomes [22] that support the importance of

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representing emotional regulation in the psychodiagnostics and psychoeducation process in patients diagnosed with PNES. In other words, PNES may represent a specific idiom of distress

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for persons subjected to distressing social experiences and situations [23] and treatment should assist in interpreting this and also in imparting effective coping strategies.

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This study is not without limitations. To begin with, the sample size is small and the design relied on self-report methods to assess emotional regulation. The latter can be problematic especially since self-reports rely on a certain amount of introspection and self-

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awareness and this particular patient group may have a limited introspective capacity. Another limitation of this study results from the differences in the research sample of patients

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diagnosed with PNES and healthy probands in the variables of work status and marital status. We realize that emotional regulation difficulties are significantly associated with the life context

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in which a person lives. However, unemployment is common in PNES, due to psychiatric morbidity [24] and as such that this sample had these characteristics is realistic. Subsequently, patients diagnosed with PNES experience high levels of perceived stigma (because of not being in employment or education), which negatively affects their health-related quality of life [25].

Future studies focusing on the emotional regulation of patients with PNES should aim to achieve larger clinical samples of patients diagnosed with PNES and perhaps also comparisons between those with PNES and patients with epilepsy, namely temporal epilepsy,

in addition to a group of healthy subjects. An interesting study might also be to compare trauma and other clinical/historical factors with emotional regulation scores.

5. Conclusion Our empirical results support that maladaptive emotional regulation is significantly elevated in those diagnosed with PNES as compared to healthy controls. This suggests that emotional dysregulation represents a key psychological mechanism in dissociative seizures which could be important for designing psycho-educational and psychotherapeutic approaches with this population.

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The psychotherapy intervention in patients diagnosed with PNES (especially cognitive behavioral therapy) is predicated on the assumption that life experiences and trauma in patients with PNES result in maladaptive core beliefs (negative schemas), and as a result, patients may demonstrate cognitive distortions and somatic symptoms [20]. Psychotherapeutic

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treatment should highlight this avoidance behavior and these maintenance factors of PNES. One of the important goals of psychotherapy in PNES patients is to achieve awareness of the

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stressful factors in the patients’ lives, an accurate reading of their internal and external responses, and then, the implementation of effective coping strategies that lead to stress relief.

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As such, emotional regulation is a primary target for treatment in these patients.

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Declaration of interests The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

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Table 1. Comparison of patients with PNES and healthy volunteers in ASQ subscales. PNES (n=64) Healthy volunteers (n=64) p-value M; SD M; SD M = 24.5; M = 21.0; Concealing p = 0.002 SD = 6.9 SD = 5.2 M = 14.5; M = 14.7; Tolerating p = 0.873 SD = 4.2 SD = 3.5 M = 20.9; M = 22.8; Adjusting p = 0.076 SD = 6.6 SD = 5.3 Significant differences (p < 0.01) in both subgroups are outlined in bold print

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ASQ subscales

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Table 2. Comparison of patients with PNES and healthy volunteers in DERS subscales. DERS PNES (n=64) Healthy volunteers (n=64) p-value subscales M; SD M; SD M = 17.0; M = 14.5; Nonacceptance p =0.006 SD = 5.6 SD = 4.1 M = 16.0; M = 11.5; Goals p < 0.001 SD = 5.2 SD = 3.7 M = 13.8; M = 11.5; Impulse control p =0.005 SD = 5.5 SD = 3.8 M = 16.4; M = 15.3; Awareness p = 0.183 SD = 4.7 SD = 4.3 M = 17.9; M = 13.0; Strategies p < 0.001 SD = 7.8 SD = 4.3 M = 11.8; M = 9.5; Clarity p < 0.001 SD = 3.5 SD = 3.1 Total score of M = 92.9; M = 75.3; p < 0.001 the DERS SD = 25.1 SD = 17.5 Significant differences (p < 0.01) in both subgroups are outlined in bold print