- Email: [email protected]
Quality of life outcome is associated with cessation rather than reduction of psychogenic nonepileptic seizures
Epilepsy & Behavior Epilepsy & Behavior 3 (2002) 455–459 www.academicpress.com
Quality of life outcome is associated with cessation rather than reduction of psychogenic nonepileptic seizures Mark Quigg,a,* Robert F. Armstrong,a Elana Farace,b and Nathan B. Fountaina a
Department of Neurology and F.E. Dreifuss Comprehensive Epilepsy Program, University of Virginia, Box 800394, Health Sciences Center, Charlottesville, VA 22908, USA b Division of Neuropsychology, Department of Neurosurgery, University of Virginia, Charlottesville, VA 22908, USA Received 7 June 2002; received in revised form 19 August 2002; accepted 21 August 2002
Abstract The outcome of psychogenic nonepileptic seizures (NES) is usually judged by recurrence of spells, but functional outcome or quality of life (QOL) is less well described. We tested the hypothesis that a decrease in NES recurrence yields corresponding improvement in QOL. Patients with NES were diagnosed with continuous video-EEG. We determined spell rate and QOL through a telephone interview at least six months after diagnosis. Thirty subjects consented to a follow-up interview (mean 17:4 1:5 months between diagnosis and interview). The rate of NES per week decreased significantly, and 10/30 (33%) had complete resolution. QOL, measured by the QOLIE-10 scale, did not improve proportionately with reduction in NES. However, subjects who reported a cessation of NES noted a significantly better total QOLIE-10 score (20:7 2:2) than those with continuing NES (27:4 1:6, P ¼ 0:02 by unpaired t test). Cessation rather than reduction of NES is associated with better QOL outcome. Ó 2002 Elsevier Science (USA). All rights reserved. Keywords: Pseudoseizure; Quality of life; Epilepsy; Intensive monitoring
1. Introduction Psychogenic nonepileptic seizures (NES) are nonepileptic, paroxysmal alterations of behavior or experience that mimic epileptic seizures but are not associated with electrographic ictal activity. As with epileptic seizures, outcome in NES also has been judged primarily in terms of spell rate [1–3]. In the case of epileptic seizures, the traditional measure of seizure rate has been supplemented by determinations of functional outcome. Quality of life (QOL) measurements, such as the Quality of Life in Epilepsy-89 [4] were developed to help measure functional outcome in epilepsy. Variants of these comprehensive scales have been evaluated for efficient screening of QOL outcome. The Quality of Life in Epilepsy-10 (QOLIE-10) is a subset of the QOLIE-89 that has been shown to correlate with the severity of epileptic seizures [5]. The QOLIE-10 contains questions regarding *
Corresponding author. Fax: 1-434-982-1726. E-mail address: [email protected] (M. Quigg).
driving, employment, independence, safety, and medication side-effects, issues that patients themselves consider important in seizure outcome [6]. As in the case of epileptic seizures, the outcome of NES may not be best measured by spell rate alone. For example, one study found that functional status did not predictably improve following diagnosis of NES [7]. To evaluate the hypothesis that improvements in spell rate lead to functional improvements as measured by QOL indices, we contacted patients with NES at least six months after diagnosis to determine outcome. To determine QOL, we used the QOLIE-10 because of its brevity and its correlation with spell rate in patients with epileptic seizures [5]. 2. Methods 2.1. Subject selection The protocol was approved by the University of Virginia Human Investigation Committee. To create the
1525-5050/02/$ - see front matter Ó 2002 Elsevier Science (USA). All rights reserved. PII: S 1 5 2 5 - 5 0 5 0 ( 0 2 ) 0 0 5 2 4 - 3
456
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
initial sample, we screened consecutive records from the University of Virginia F.E. Dreifuss Epilepsy Unit from a 30-month period. All patients with EEG-confirmed NES with an interval of at least six months between diagnosis and follow-up interview were eligible for inclusion. We defined NES as a spell marked by alteration or loss of consciousness that was accompanied by normal waking cerebral activities on continuous video EEG (CV-EEG). Patients with spells that did not include alteration or loss of consciousness were excluded. This definition of NES was designed to exclude patients with epileptic seizures whose possible epileptic auras or simple partial seizures could be misdiagnosed as NES. Some patients with epileptic seizures could be included if they also had only NES during CV-EEG monitoring as defined above. 2.2. Outcome assessment Patients meeting the inclusion criteria were contacted by telephone and interviewed with the use of a standard questionnaire. NES outcome was determined by the patientÕs best estimate of weekly NES rate at the time of admission (past) and at the time of interview (current). A subject was considered spell-free if they noted no events for the four weeks preceding the interview. In data analysis, we evaluated outcome by current NES rate and its interval change. QOL was assessed with the QOLIE-10 scale. The QOLIE-10 [5] consists of 10 clinician-administered questions scaled from 1 (best) to 5 (worst) regarding (1) energy, (2) mood, (3) driving status, (4) memory, (5) employment status, (6) social relationships, (7) physical and (8) mental symptoms and side effects, (9) fear of impending event, and (10) overall quality of life. The current QOL was determined from the sum of the individual 1–5 point scales to arrive at a total QOLIE-10 score ranging from 10–50 with the lower score designating favorable QOL. Interval change in QOL was determined by asking each patient whether each QOLIE-10 scale had changed since admission and assigning categorical variables for worsening, no change, or improvement. Functional outcome was also assessed by determining past and current disability status.
3. Results 3.1. Subject characteristics Fifty-one patients met initial criteria. During the telephone interview, seven subjects with severe mental retardation were judged too cognitively impaired to participate. One subject refused to be interviewed.
Another subject had died from unknown causes. Twelve subjects were lost to follow-up. The remaining 30 subjects (59% of the total sample) consented to the telephone interview. A comparison of the demographics (e.g., age, disability status, and gender) between patients available for follow-up and those who were not did not show any significant differences between groups. Thus, the current sample is likely to represent the overall population of NES patients at our institution, with the exclusion of severely mentally retarded patients. The mean interval standard error between admission and interview was 17:4 1:5 months (range 6–36 months). Only two of the thirty subjects (7%) had a coexistent diagnosis of epilepsy in addition to NES confirmed by CV-EEG. Ten of the 30 subjects had psychopathology assessments using the Minnesota Multiphasic Personality Inventory-2. Subjects were not administered the MMPI-2 if they did not have a reading level above 8th grade and/or were not able to sustain the 2–3 h of testing. All subjects had at least one scale with an abnormal score (t score P70, corresponding to two standard deviations from the mean, which is interpreted as being clinically significant compared to the normal population). Nearly all subjects (9/10) had abnormal Scales 1 and 3, consistent with a somatization disorder. Posttraumatic stress disorder scales (PK-PTSD or PS-PTSD) were P70 in 5/10 subjects. The majority of patients (18/ 30) underwent formal psychiatric consultation during admission. Six patients had Axis I diagnoses of conversion, four of depression, two with dissociation, and one each with panic or somatization disorder. Four received no Axis I diagnoses and none were given Axis II diagnoses. Only three of these 18 patients continued with psychiatric care after discharge, largely due to financial limitations and poor mental health care availability in this largely rural patient population. 3.2. NES rate and QOL outcome The subjectsÕ recollections of their past NES rate at the time of admission highly correlated with the corresponding estimate in the hospital chart (PearsonÕs r ¼ 0:61; P ¼ 0:0007), indicating a high reliability for self-reporting. The 30 subjects, as a group, enjoyed a significant decrease in the occurrence of NES. The current weekly rate of NES was 4:8 2:5, significantly reduced from the past rate of 23:7 8:0 per week (P ¼ 0:01 by paired t test). Many subjects (27/30) noted at least a >50% reduction. One-third (10/30) said that their NES had completely resolved. Two subjects had comorbid epileptic seizures and NES diagnosed by video EEG. These two subjects experienced worsening in the rate of NES after tapering of one of their multiple anticonvulsant medications. None of the 28 subjects with only NES experienced worsening of spell rate.
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
The current mean QOLIE-10 score was 25:2 1:4 (range 10–40), with lower numbers designating favorable outcome. Current QOLIE-10 subscales and their interval improvement from admission for all 30 subjects are shown in Fig. 1. Subscales with the worst scores were employment status, driving status, and energy, and those with the best were mental and physical symptoms. Measured by the number of responses of worsening (n ¼ 47, 16% of 300 possible responses), no change (n ¼ 120, 40%), or improvement (n ¼ 133, 44%) within each QOLIE-10 subscale, there was no clear interval improvement from admission to interview in QOL. Energy, mood, and fear of impending seizures were the only subscales to improve in over half the subjects. Driving and employment underwent the least improvement. 3.3. Effects of NES rate on QOL Despite overall improvements in NES rate, improvement in QOL indices did not correspond to reduction in NES rate. Neither the current NES rate (PearsonÕs r ¼ 0:17, P ¼ 0:93) nor its interval change (r ¼ 0:19, P ¼ 0:31) predicted the current total QOLIE-10 score. Similarly, neither the current NES rate (P ¼ 0:07) nor change in NES rate (P ¼ 0:65) were as-
457
Fig. 2. Mean and standard error of total QOLIE-10 scores in subjects with psychogenic nonepileptic seizures (NES) divided into those whose spells ceased at least six months after diagnosis and those whose NES continued.
sociated with worsened, unchanged, or improved overall QOL when compared by ANOVA. Because patients with epileptic seizures may not enjoy functional benefit until seizures completely remit [8], subsequent analyses compared those subjects who were completely free of NES (n ¼ 10=30) to those with continuing NES (n ¼ 20=30). Patients with resolved NES had significantly lower current QOLIE-10 scores than those with continuing NES (Fig. 2). Resolution of NES, however, was not associated with perceived changes in QOL in the interval between admission and interview; the proportion of subjects with resolved NES who noted an overall improvement in QOL (5/10 ¼ 50%) was not significantly different from that noted by those with continuing NES (9/20 ¼ 45%, P ¼ 0:99 by FisherÕs test). Table 1 shows evaluations of other patient variables by NES outcome. Older age and positive disability status at admission were associated with recurring NES. Whereas none of the patients with resolved NES received disability at admission, 44% of patients with recurrent NES did (P ¼ 0:01). In fact, the proportion of patients currently receiving disability (18/30 subjects) significantly worsened in the interval (admission proportion ¼ 10/30, P ¼ 0:02 by McNemarÕs test).
4. Conclusion
Fig. 1. (A) Mean scores and standard errors of a quality-of-life assessment survey (QOLIE-10). (B) Percentage of subjects noting improvements in QOLIE-10 subscales in the interval between diagnosis and follow-up interview.
The main finding of this study of NES outcome is that a complete cessation of NES is required to demonstrate a significantly higher QOL. Neither the rate nor the interval change in the rate of NES following diagnosis was proportional to QOL indices or their interval change. This study suggests that the goal in treatment of NES should be remission of NES rather than a reduction. This study has several limitations. First, the initial sample was determined from retrospective review of appropriate subjects. Some patients from the initial
458
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
Table 1 Effect of resolution of psychogenic nonepileptic seizures Variable
Spell free
Not spell free
n Admission age (years) Sex (female) Race (Caucasian) Duration of symptoms (years) Abnormal neurological exam or cognition (yes) Interictal epileptiform discharges (present) Understood diagnosis (yes) Psychiatric treatment (yes) Therapist/psychologist (yes) Past education level ( P high school/GED) Past employment (part or full time) Past disability status (receiving disability) Current education level ( P high school/GED) Current employment (part or full time) Current disability status (receiving disability)
10 23:4 2:4 8 (80%) 8 (80%) 2:1 2:9 2 (20%) 2 (20%) 8 (80%) 6 (60%) 3 (30%) 5 (50%) 2 (20%) 0 (0%) 8 (80%) 3 (30%) 2 (20%)
20 38:0 2:9 15 (75%) 19 (95%) 6:8 1:8 8 (40%) 7 (35%) 11 (55%) 11 (55%) 11 (55%) 11 (55%) 2 (10%) 10 (50%) 11 (55%) 1 (5%) 16 (80%)
p-value
0.0032 0.99 0.25 0.13 0.42 0.67 0.25 0.99 0.26 0.99 0.57 0.01 0.25 0.09 0.004
Continuous variables are expressed in mean SE with p value derived from unpaired t tests; nominative variables are expressed in number (percentage within group) with p values derived from FisherÕs exact tests (* ¼ significant).
sample were lost to follow-up or did not wish to participate; these subjects may have outcomes different from those patients available for our follow-up interview. However, a comparison of demographics between subjects available for follow-up and those from whom data were not collected did not show a significant difference, except that patients with severe mental retardation were not included in the current study group. Second, the resulting sample size did not support the use of multivariate statistics, and factors such as age or disability benefits may play important roles in QOL outcome. Finally, self-reported rates of spell occurrence may be prone to error. However, our preliminary studies, as well as our experience in the evaluation of patients who have NES, indicated that compliance with potentially more accurate methods such as spell diaries was poor. Because self-reported spell rates correlated with self-reported rates at the time of diagnostic evaluation, we believe that self-report is a reasonably accurate method of spell determination in this patient population. Nevertheless, NES patients available for follow-up interview appear similar to those in earlier studies. Incidence of comorbidity of epileptic seizures and NES in other studies ranges from 6–73% [3,9–11]. The incidence of seizure resolution—one third of the sample—lay within the 25–40% reported in other recent studies [1–3,7]. Parity of outcome with previous studies suggests that our criterion of NES includes a representative sample of NES patients. We note that the more subjective of the QOLIE-10 subscales, such as mood, fear of impending seizures, and energy were the most reported as improved in the interval between diagnosis and interview. The more objective subscales, such as driving or employment,
showed the least improvement. Furthermore, the number of subjects on disability increased after diagnosis, and positive disability status at admission predicted its presence at follow-up. These factors associated with the failure to attain financial and social independence suggest that secondary gain may be a significant barrier to clinical and occupational improvement. We believe that the dissociation between subjective and objective components of QOL may originate from interactions with the underlying psychiatric disorders that render achievement of more objective successes difficult [12]. Although comparisons of QOL outcome between patients with intractable epilepsy and NES find similar impairments between groups, noting that NES and epileptic seizures can equally lead to loss of driving privileges and to impaired social and occupational functioning [13], the etiology of these impairments are very different between these groups. Although in the past NES patients were thought solely to suffer from conversion disorder, in several careful psychodiagnostic studies the NES patient population has been found to include other, often comorbid, psychiatric diagnoses, particularly panic disorder [14], somatoform disorders, dissociative disorders, affective disorders, and personality disorders [15]. Trauma, particularly from sexual or physical abuse or other significant stressors, occurs frequently in NES patients [15,16]. The high rate of trauma in this population may explain the comorbidity of psychiatric diagnoses in this population. The spells themselves may be a defense mechanism or a dissociative response to a traumatic event [17]. Unfortunately, data on recent trauma and stressors was not uniformly available in the present study, but this may be an explanation for the four NES patients with no formal Axis I diagnosis.
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
The most effective treatment for patients with NES is controversial. At first glance our data suggests that traditional approaches such as a psychiatric evaluation was not associated with cessation of NES. A clear presentation of findings of NES and the subjectÕs acceptance of them, the standard first step thought best to lead to cessation of NES [18], also did not appear effective. Our data is insufficient to determine if the relatively low number of patients who continued with mental health care reflects an underutilization of available community resources or the poor availability of appropriate mental health care, a distinct possibility in our rural-based catchment area. We agree with previous investigators who conclude that continued reassurance and input of the neurologist, in concert with mental health workers, may be the best way to approach NES patients with chronic psychiatric disorders [3]. Indeed, NES should be considered a symptom of an underlying psychological or psychiatric disorder or recent trauma rather than a disorder unto itself. Our findings of worsened spells following diagnosis in patients with comorbid epileptic seizures and NES emphasizes that the epileptologistÕs job may not be done following the discovery and explanation of NES in this particular group. Prospective studies of pharmacological and pyschotherapeutic treatments are required to further delineate treatment approaches. Acknowledgment Our thanks to our extern Christina Ayers for her dedicated help. References [1] Lempert T, Schmidt D. Natural history and outcome of psychogenic seizures: a clinical study in 50 patients. J Neurol 1990;237:35–8.
459
[2] Meierkord H, Will B, Fish D, Shorvon S. The clinical features and prognosis of pseudoseizures diagnosed using video-EEG telemetry. Neurology 1991;41:1643–6. [3] Kanner AM, Parra J, Frey M, Stebbins G, Pierre-Louis S, Iriarte J. Psychiatric and neurologic predictors of psychogenic pseudoseizure outcome. Neurology 1999;53:933–8. [4] Devinsky O, Vickrey BG, Cramer J, et al. Development of the quality of life in epilepsy inventory. Epilepsia 1995;36:1089–104. [5] Cramer J, Perrine K, Devinsky O, Meador K. A brief questionnaire to screen for quality of life in epilepsy: the QOLIE-10. Epilepsia 1996;37:577–82. [6] Gilliam F, Kuzniecky R, Faught E, Black L, Carpenter C, Schrodt R. Patient validated content of epilepsy-specific quality of life measurement. Epilepsia 1997;38:233–6. [7] Walczak T, Papacostas S, Williams DT, Scheuer M, Lebowitz N, Notarfrancesco A. Outcome after diagnosis of psychogenic nonepileptic seizures. Epilepsia 1995;36:1131–7. [8] Sperling M, Saykin A, Roberts F, French J, OÕConnor M. Occupational outcome after temporal lobectomy for refractory epilepsy. Neurology 1995;45:970–7. [9] Devinsky O, Thacker K. Nonepileptic seizures. Neurol Clin 1995;13:299–319. [10] Chabolla D, Knahn L, So E, Rummans T. Psychogenic nonepileptic seizures. Mayo Clin Proc 1996;71:493–500. [11] Lesser R. Psychogenic seizures. Neurology 1996;46:1499–507. [12] Krumholz A, Niedermeyer E. Psychogenic seizures: a clinical study with follow-up data. Neurology 1983;33:35–8. [13] Brier JI, Fuchs KL, Brookshire BL, et al. Quality of life perception in patients with intractable epilepsy or pseudoseizures. Arch Neurol 1998;55:660–5. [14] Snyder SL, Rosenbaum DH, Rowan AJ, Strain JJ. SCID diagnosis of panic disorder in nonepileptic events. J Neuropsychiatry 1994;6:261–6. [15] Bowmand ES, Markand ON. Psychodynamics and psychiatric diagnoses and pseudoseizure subjects. Am J Psychiatry 1996;153:57–63. [16] Drake ME, Pakalnis A, Phillips BB. Neuropsychological and psychiatric correlates of intractable pseudoseizures. Seizure 1992;1:11–3. [17] Jawad SSM, Jamil N, Clarke EJ, Lewis A, Whitecross S, Richens A. Psychiatric morbidity and psychodynamics of patients with convulsive pseudoseizures. Seizure 1995;4:201–6. [18] Betts T. Conversion disorders. In: Engel JJ, Pedley TA, editors. Epilepsy: a comprehensive textbook. Philadelphia: LippincottRaven; 1997. p. 2757–66.
Quality of life outcome is associated with cessation rather than reduction of psychogenic nonepileptic seizures Mark Quigg,a,* Robert F. Armstrong,a Elana Farace,b and Nathan B. Fountaina a
Department of Neurology and F.E. Dreifuss Comprehensive Epilepsy Program, University of Virginia, Box 800394, Health Sciences Center, Charlottesville, VA 22908, USA b Division of Neuropsychology, Department of Neurosurgery, University of Virginia, Charlottesville, VA 22908, USA Received 7 June 2002; received in revised form 19 August 2002; accepted 21 August 2002
Abstract The outcome of psychogenic nonepileptic seizures (NES) is usually judged by recurrence of spells, but functional outcome or quality of life (QOL) is less well described. We tested the hypothesis that a decrease in NES recurrence yields corresponding improvement in QOL. Patients with NES were diagnosed with continuous video-EEG. We determined spell rate and QOL through a telephone interview at least six months after diagnosis. Thirty subjects consented to a follow-up interview (mean 17:4 1:5 months between diagnosis and interview). The rate of NES per week decreased significantly, and 10/30 (33%) had complete resolution. QOL, measured by the QOLIE-10 scale, did not improve proportionately with reduction in NES. However, subjects who reported a cessation of NES noted a significantly better total QOLIE-10 score (20:7 2:2) than those with continuing NES (27:4 1:6, P ¼ 0:02 by unpaired t test). Cessation rather than reduction of NES is associated with better QOL outcome. Ó 2002 Elsevier Science (USA). All rights reserved. Keywords: Pseudoseizure; Quality of life; Epilepsy; Intensive monitoring
1. Introduction Psychogenic nonepileptic seizures (NES) are nonepileptic, paroxysmal alterations of behavior or experience that mimic epileptic seizures but are not associated with electrographic ictal activity. As with epileptic seizures, outcome in NES also has been judged primarily in terms of spell rate [1–3]. In the case of epileptic seizures, the traditional measure of seizure rate has been supplemented by determinations of functional outcome. Quality of life (QOL) measurements, such as the Quality of Life in Epilepsy-89 [4] were developed to help measure functional outcome in epilepsy. Variants of these comprehensive scales have been evaluated for efficient screening of QOL outcome. The Quality of Life in Epilepsy-10 (QOLIE-10) is a subset of the QOLIE-89 that has been shown to correlate with the severity of epileptic seizures [5]. The QOLIE-10 contains questions regarding *
Corresponding author. Fax: 1-434-982-1726. E-mail address: [email protected] (M. Quigg).
driving, employment, independence, safety, and medication side-effects, issues that patients themselves consider important in seizure outcome [6]. As in the case of epileptic seizures, the outcome of NES may not be best measured by spell rate alone. For example, one study found that functional status did not predictably improve following diagnosis of NES [7]. To evaluate the hypothesis that improvements in spell rate lead to functional improvements as measured by QOL indices, we contacted patients with NES at least six months after diagnosis to determine outcome. To determine QOL, we used the QOLIE-10 because of its brevity and its correlation with spell rate in patients with epileptic seizures [5]. 2. Methods 2.1. Subject selection The protocol was approved by the University of Virginia Human Investigation Committee. To create the
1525-5050/02/$ - see front matter Ó 2002 Elsevier Science (USA). All rights reserved. PII: S 1 5 2 5 - 5 0 5 0 ( 0 2 ) 0 0 5 2 4 - 3
456
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
initial sample, we screened consecutive records from the University of Virginia F.E. Dreifuss Epilepsy Unit from a 30-month period. All patients with EEG-confirmed NES with an interval of at least six months between diagnosis and follow-up interview were eligible for inclusion. We defined NES as a spell marked by alteration or loss of consciousness that was accompanied by normal waking cerebral activities on continuous video EEG (CV-EEG). Patients with spells that did not include alteration or loss of consciousness were excluded. This definition of NES was designed to exclude patients with epileptic seizures whose possible epileptic auras or simple partial seizures could be misdiagnosed as NES. Some patients with epileptic seizures could be included if they also had only NES during CV-EEG monitoring as defined above. 2.2. Outcome assessment Patients meeting the inclusion criteria were contacted by telephone and interviewed with the use of a standard questionnaire. NES outcome was determined by the patientÕs best estimate of weekly NES rate at the time of admission (past) and at the time of interview (current). A subject was considered spell-free if they noted no events for the four weeks preceding the interview. In data analysis, we evaluated outcome by current NES rate and its interval change. QOL was assessed with the QOLIE-10 scale. The QOLIE-10 [5] consists of 10 clinician-administered questions scaled from 1 (best) to 5 (worst) regarding (1) energy, (2) mood, (3) driving status, (4) memory, (5) employment status, (6) social relationships, (7) physical and (8) mental symptoms and side effects, (9) fear of impending event, and (10) overall quality of life. The current QOL was determined from the sum of the individual 1–5 point scales to arrive at a total QOLIE-10 score ranging from 10–50 with the lower score designating favorable QOL. Interval change in QOL was determined by asking each patient whether each QOLIE-10 scale had changed since admission and assigning categorical variables for worsening, no change, or improvement. Functional outcome was also assessed by determining past and current disability status.
3. Results 3.1. Subject characteristics Fifty-one patients met initial criteria. During the telephone interview, seven subjects with severe mental retardation were judged too cognitively impaired to participate. One subject refused to be interviewed.
Another subject had died from unknown causes. Twelve subjects were lost to follow-up. The remaining 30 subjects (59% of the total sample) consented to the telephone interview. A comparison of the demographics (e.g., age, disability status, and gender) between patients available for follow-up and those who were not did not show any significant differences between groups. Thus, the current sample is likely to represent the overall population of NES patients at our institution, with the exclusion of severely mentally retarded patients. The mean interval standard error between admission and interview was 17:4 1:5 months (range 6–36 months). Only two of the thirty subjects (7%) had a coexistent diagnosis of epilepsy in addition to NES confirmed by CV-EEG. Ten of the 30 subjects had psychopathology assessments using the Minnesota Multiphasic Personality Inventory-2. Subjects were not administered the MMPI-2 if they did not have a reading level above 8th grade and/or were not able to sustain the 2–3 h of testing. All subjects had at least one scale with an abnormal score (t score P70, corresponding to two standard deviations from the mean, which is interpreted as being clinically significant compared to the normal population). Nearly all subjects (9/10) had abnormal Scales 1 and 3, consistent with a somatization disorder. Posttraumatic stress disorder scales (PK-PTSD or PS-PTSD) were P70 in 5/10 subjects. The majority of patients (18/ 30) underwent formal psychiatric consultation during admission. Six patients had Axis I diagnoses of conversion, four of depression, two with dissociation, and one each with panic or somatization disorder. Four received no Axis I diagnoses and none were given Axis II diagnoses. Only three of these 18 patients continued with psychiatric care after discharge, largely due to financial limitations and poor mental health care availability in this largely rural patient population. 3.2. NES rate and QOL outcome The subjectsÕ recollections of their past NES rate at the time of admission highly correlated with the corresponding estimate in the hospital chart (PearsonÕs r ¼ 0:61; P ¼ 0:0007), indicating a high reliability for self-reporting. The 30 subjects, as a group, enjoyed a significant decrease in the occurrence of NES. The current weekly rate of NES was 4:8 2:5, significantly reduced from the past rate of 23:7 8:0 per week (P ¼ 0:01 by paired t test). Many subjects (27/30) noted at least a >50% reduction. One-third (10/30) said that their NES had completely resolved. Two subjects had comorbid epileptic seizures and NES diagnosed by video EEG. These two subjects experienced worsening in the rate of NES after tapering of one of their multiple anticonvulsant medications. None of the 28 subjects with only NES experienced worsening of spell rate.
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
The current mean QOLIE-10 score was 25:2 1:4 (range 10–40), with lower numbers designating favorable outcome. Current QOLIE-10 subscales and their interval improvement from admission for all 30 subjects are shown in Fig. 1. Subscales with the worst scores were employment status, driving status, and energy, and those with the best were mental and physical symptoms. Measured by the number of responses of worsening (n ¼ 47, 16% of 300 possible responses), no change (n ¼ 120, 40%), or improvement (n ¼ 133, 44%) within each QOLIE-10 subscale, there was no clear interval improvement from admission to interview in QOL. Energy, mood, and fear of impending seizures were the only subscales to improve in over half the subjects. Driving and employment underwent the least improvement. 3.3. Effects of NES rate on QOL Despite overall improvements in NES rate, improvement in QOL indices did not correspond to reduction in NES rate. Neither the current NES rate (PearsonÕs r ¼ 0:17, P ¼ 0:93) nor its interval change (r ¼ 0:19, P ¼ 0:31) predicted the current total QOLIE-10 score. Similarly, neither the current NES rate (P ¼ 0:07) nor change in NES rate (P ¼ 0:65) were as-
457
Fig. 2. Mean and standard error of total QOLIE-10 scores in subjects with psychogenic nonepileptic seizures (NES) divided into those whose spells ceased at least six months after diagnosis and those whose NES continued.
sociated with worsened, unchanged, or improved overall QOL when compared by ANOVA. Because patients with epileptic seizures may not enjoy functional benefit until seizures completely remit [8], subsequent analyses compared those subjects who were completely free of NES (n ¼ 10=30) to those with continuing NES (n ¼ 20=30). Patients with resolved NES had significantly lower current QOLIE-10 scores than those with continuing NES (Fig. 2). Resolution of NES, however, was not associated with perceived changes in QOL in the interval between admission and interview; the proportion of subjects with resolved NES who noted an overall improvement in QOL (5/10 ¼ 50%) was not significantly different from that noted by those with continuing NES (9/20 ¼ 45%, P ¼ 0:99 by FisherÕs test). Table 1 shows evaluations of other patient variables by NES outcome. Older age and positive disability status at admission were associated with recurring NES. Whereas none of the patients with resolved NES received disability at admission, 44% of patients with recurrent NES did (P ¼ 0:01). In fact, the proportion of patients currently receiving disability (18/30 subjects) significantly worsened in the interval (admission proportion ¼ 10/30, P ¼ 0:02 by McNemarÕs test).
4. Conclusion
Fig. 1. (A) Mean scores and standard errors of a quality-of-life assessment survey (QOLIE-10). (B) Percentage of subjects noting improvements in QOLIE-10 subscales in the interval between diagnosis and follow-up interview.
The main finding of this study of NES outcome is that a complete cessation of NES is required to demonstrate a significantly higher QOL. Neither the rate nor the interval change in the rate of NES following diagnosis was proportional to QOL indices or their interval change. This study suggests that the goal in treatment of NES should be remission of NES rather than a reduction. This study has several limitations. First, the initial sample was determined from retrospective review of appropriate subjects. Some patients from the initial
458
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
Table 1 Effect of resolution of psychogenic nonepileptic seizures Variable
Spell free
Not spell free
n Admission age (years) Sex (female) Race (Caucasian) Duration of symptoms (years) Abnormal neurological exam or cognition (yes) Interictal epileptiform discharges (present) Understood diagnosis (yes) Psychiatric treatment (yes) Therapist/psychologist (yes) Past education level ( P high school/GED) Past employment (part or full time) Past disability status (receiving disability) Current education level ( P high school/GED) Current employment (part or full time) Current disability status (receiving disability)
10 23:4 2:4 8 (80%) 8 (80%) 2:1 2:9 2 (20%) 2 (20%) 8 (80%) 6 (60%) 3 (30%) 5 (50%) 2 (20%) 0 (0%) 8 (80%) 3 (30%) 2 (20%)
20 38:0 2:9 15 (75%) 19 (95%) 6:8 1:8 8 (40%) 7 (35%) 11 (55%) 11 (55%) 11 (55%) 11 (55%) 2 (10%) 10 (50%) 11 (55%) 1 (5%) 16 (80%)
p-value
0.0032 0.99 0.25 0.13 0.42 0.67 0.25 0.99 0.26 0.99 0.57 0.01 0.25 0.09 0.004
Continuous variables are expressed in mean SE with p value derived from unpaired t tests; nominative variables are expressed in number (percentage within group) with p values derived from FisherÕs exact tests (* ¼ significant).
sample were lost to follow-up or did not wish to participate; these subjects may have outcomes different from those patients available for our follow-up interview. However, a comparison of demographics between subjects available for follow-up and those from whom data were not collected did not show a significant difference, except that patients with severe mental retardation were not included in the current study group. Second, the resulting sample size did not support the use of multivariate statistics, and factors such as age or disability benefits may play important roles in QOL outcome. Finally, self-reported rates of spell occurrence may be prone to error. However, our preliminary studies, as well as our experience in the evaluation of patients who have NES, indicated that compliance with potentially more accurate methods such as spell diaries was poor. Because self-reported spell rates correlated with self-reported rates at the time of diagnostic evaluation, we believe that self-report is a reasonably accurate method of spell determination in this patient population. Nevertheless, NES patients available for follow-up interview appear similar to those in earlier studies. Incidence of comorbidity of epileptic seizures and NES in other studies ranges from 6–73% [3,9–11]. The incidence of seizure resolution—one third of the sample—lay within the 25–40% reported in other recent studies [1–3,7]. Parity of outcome with previous studies suggests that our criterion of NES includes a representative sample of NES patients. We note that the more subjective of the QOLIE-10 subscales, such as mood, fear of impending seizures, and energy were the most reported as improved in the interval between diagnosis and interview. The more objective subscales, such as driving or employment,
showed the least improvement. Furthermore, the number of subjects on disability increased after diagnosis, and positive disability status at admission predicted its presence at follow-up. These factors associated with the failure to attain financial and social independence suggest that secondary gain may be a significant barrier to clinical and occupational improvement. We believe that the dissociation between subjective and objective components of QOL may originate from interactions with the underlying psychiatric disorders that render achievement of more objective successes difficult [12]. Although comparisons of QOL outcome between patients with intractable epilepsy and NES find similar impairments between groups, noting that NES and epileptic seizures can equally lead to loss of driving privileges and to impaired social and occupational functioning [13], the etiology of these impairments are very different between these groups. Although in the past NES patients were thought solely to suffer from conversion disorder, in several careful psychodiagnostic studies the NES patient population has been found to include other, often comorbid, psychiatric diagnoses, particularly panic disorder [14], somatoform disorders, dissociative disorders, affective disorders, and personality disorders [15]. Trauma, particularly from sexual or physical abuse or other significant stressors, occurs frequently in NES patients [15,16]. The high rate of trauma in this population may explain the comorbidity of psychiatric diagnoses in this population. The spells themselves may be a defense mechanism or a dissociative response to a traumatic event [17]. Unfortunately, data on recent trauma and stressors was not uniformly available in the present study, but this may be an explanation for the four NES patients with no formal Axis I diagnosis.
M. Quigg et al. / Epilepsy & Behavior 3 (2002) 455–459
The most effective treatment for patients with NES is controversial. At first glance our data suggests that traditional approaches such as a psychiatric evaluation was not associated with cessation of NES. A clear presentation of findings of NES and the subjectÕs acceptance of them, the standard first step thought best to lead to cessation of NES [18], also did not appear effective. Our data is insufficient to determine if the relatively low number of patients who continued with mental health care reflects an underutilization of available community resources or the poor availability of appropriate mental health care, a distinct possibility in our rural-based catchment area. We agree with previous investigators who conclude that continued reassurance and input of the neurologist, in concert with mental health workers, may be the best way to approach NES patients with chronic psychiatric disorders [3]. Indeed, NES should be considered a symptom of an underlying psychological or psychiatric disorder or recent trauma rather than a disorder unto itself. Our findings of worsened spells following diagnosis in patients with comorbid epileptic seizures and NES emphasizes that the epileptologistÕs job may not be done following the discovery and explanation of NES in this particular group. Prospective studies of pharmacological and pyschotherapeutic treatments are required to further delineate treatment approaches. Acknowledgment Our thanks to our extern Christina Ayers for her dedicated help. References [1] Lempert T, Schmidt D. Natural history and outcome of psychogenic seizures: a clinical study in 50 patients. J Neurol 1990;237:35–8.
459
[2] Meierkord H, Will B, Fish D, Shorvon S. The clinical features and prognosis of pseudoseizures diagnosed using video-EEG telemetry. Neurology 1991;41:1643–6. [3] Kanner AM, Parra J, Frey M, Stebbins G, Pierre-Louis S, Iriarte J. Psychiatric and neurologic predictors of psychogenic pseudoseizure outcome. Neurology 1999;53:933–8. [4] Devinsky O, Vickrey BG, Cramer J, et al. Development of the quality of life in epilepsy inventory. Epilepsia 1995;36:1089–104. [5] Cramer J, Perrine K, Devinsky O, Meador K. A brief questionnaire to screen for quality of life in epilepsy: the QOLIE-10. Epilepsia 1996;37:577–82. [6] Gilliam F, Kuzniecky R, Faught E, Black L, Carpenter C, Schrodt R. Patient validated content of epilepsy-specific quality of life measurement. Epilepsia 1997;38:233–6. [7] Walczak T, Papacostas S, Williams DT, Scheuer M, Lebowitz N, Notarfrancesco A. Outcome after diagnosis of psychogenic nonepileptic seizures. Epilepsia 1995;36:1131–7. [8] Sperling M, Saykin A, Roberts F, French J, OÕConnor M. Occupational outcome after temporal lobectomy for refractory epilepsy. Neurology 1995;45:970–7. [9] Devinsky O, Thacker K. Nonepileptic seizures. Neurol Clin 1995;13:299–319. [10] Chabolla D, Knahn L, So E, Rummans T. Psychogenic nonepileptic seizures. Mayo Clin Proc 1996;71:493–500. [11] Lesser R. Psychogenic seizures. Neurology 1996;46:1499–507. [12] Krumholz A, Niedermeyer E. Psychogenic seizures: a clinical study with follow-up data. Neurology 1983;33:35–8. [13] Brier JI, Fuchs KL, Brookshire BL, et al. Quality of life perception in patients with intractable epilepsy or pseudoseizures. Arch Neurol 1998;55:660–5. [14] Snyder SL, Rosenbaum DH, Rowan AJ, Strain JJ. SCID diagnosis of panic disorder in nonepileptic events. J Neuropsychiatry 1994;6:261–6. [15] Bowmand ES, Markand ON. Psychodynamics and psychiatric diagnoses and pseudoseizure subjects. Am J Psychiatry 1996;153:57–63. [16] Drake ME, Pakalnis A, Phillips BB. Neuropsychological and psychiatric correlates of intractable pseudoseizures. Seizure 1992;1:11–3. [17] Jawad SSM, Jamil N, Clarke EJ, Lewis A, Whitecross S, Richens A. Psychiatric morbidity and psychodynamics of patients with convulsive pseudoseizures. Seizure 1995;4:201–6. [18] Betts T. Conversion disorders. In: Engel JJ, Pedley TA, editors. Epilepsy: a comprehensive textbook. Philadelphia: LippincottRaven; 1997. p. 2757–66.