Anda di halaman 1dari 5

G Model

YSEIZ-2409; No. of Pages 4


Seizure xxx (2014) xxxxxx

Contents lists available at ScienceDirect

Seizure
journal homepage: www.elsevier.com/locate/yseiz

Co-morbidities and outcome of childhood psychogenic non-epileptic


seizuresAn observational study
Vikram Singh Rawat a, Vikas Dhiman b,c, Sanjib Sinha c,*, Kommu John Vijay Sagar d,
Harish Thippeswamy a, Santosh Kumar Chaturvedi a, Shoba Srinath d,
Parthasarthy Satishchandra c
a

Department of Psychiatry, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India
Department of Clinical Neurosciences, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India
Department of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India
d
Department of Child and Adolescent Psychiatry, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India
b
c

A R T I C L E I N F O

A B S T R A C T

Article history:
Received 29 May 2014
Received in revised form 24 September 2014
Accepted 26 September 2014

Purpose: To assess the psychiatric diagnoses and outcome in children with psychogenic non-epileptic
seizures (PNES).
Methodology: This hospital based observational study was performed on 44 children aged <16 years,
who suspected to have psychogenic non-epileptic seizures based on video-EEG, from August 2005 to
August 2012. The parameters noted were the psychiatric diagnosis, co-morbidities, management
assessment and interventions (pharmacological and psychosocial), number and duration of follow-up
visits, symptoms at follow-up, functioning as reflected by involvement in the social and scholastic work.
Results: All forty four children completed the evaluation. Thirty four children were diagnosed as having
PNES and the underlying psychiatric diagnosis was conversion disorder (n = 34, 77.3%). Co-morbid
psychiatric disorders were present in 17 children (50%). The common co-morbidities were intellectual
disability (n = 8, 23.5%), specific learning disorder (n = 5, 14.7%), and depression (n = 5, 14.7%). Co-morbid
epilepsy was present in 8 (23.5%) children and family history of epilepsy was present in 10 (29.4%) cases.
About 17 of 34 (50.0%) patients had a minimum follow-up of 6 months (13.9 ! 4.8 months). Twenty six
children (76.5%) remained symptom free at the follow-up of 9.8 ! 7 months. The remaining 10 children
(22.7%) had non-epileptic seizures with underlying diagnosis of Attention Deficit Hyperactivity Disorder
(ADHD), gratification disorder and other physiological conditions.
Conclusions: Conversion disorder is a common diagnosis underlying psychogenic non-epileptic seizures.
Outcome was good in 76.5% children with PNES. A multidisciplinary approach is needed in the diagnosis
and management of PNES.
! 2014 British Epilepsy Association. Published by Elsevier Ltd. All rights reserved.

Keywords:
Non-epileptic
Outcome assessment seizures
Pediatric
Psychogenic

1. Introduction
Psychogenic non-epileptic seizures (PNES) are involuntary time
limited events that may manifest as motor, sensory or behavioral
phenomena resembling true seizures.13 Clinically, when a nonepileptic event is suspected, it is prudent to reassess the clinical
history and evaluate for the possible causes of the paroxysmal
events like psychiatric problems, migraine, syncope, periodic

* Corresponding author at: Department of Neurology, National Institute of


Mental Health and Neurosciences (NIMHANS), 560029, Karnataka, India.
Tel.: +91 8026995150; fax: +91 80 26564830.
E-mail address: sanjib_sinha2004@yahoo.co.in (S. Sinha).

paralysis, etc.3,4 Psychogenic non-epileptic seizures are a common


morbidity in the pediatric population referred to epilepsy clinics
and are often misdiagnosed with epilepsy.5 Around 2040% of
children evaluated in epilepsy clinics have been reported to have
PNES.69 It is often seen as a co-morbid condition in the patients
with epilepsy, depression and other psychiatric disorders.10,11
Psychogenic non-epileptic seizures, if suspected, need evaluation by a mental health professional for the diagnostic clarification
and appropriate management.12,13 Various psychological factors
like anxiety or stress, physical abuse, significant bereavement,
family dysfunction, relationship problems, depression, sexual
abuse have been identified as important factors in children with
PNES.14 The prognosis of PNES in children is found to be more
favorable15,16 than in the adults although differing rates have been

http://dx.doi.org/10.1016/j.seizure.2014.09.011
1059-1311/! 2014 British Epilepsy Association. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Rawat VS, et al. Co-morbidities and outcome of childhood psychogenic non-epileptic seizuresAn
observational study. Seizure: Eur J Epilepsy (2014), http://dx.doi.org/10.1016/j.seizure.2014.09.011

G Model

YSEIZ-2409; No. of Pages 4


2

V.S. Rawat et al. / Seizure xxx (2014) xxxxxx

reported.1719 The outcome in pediatric population varies, possibly


owing to differences in the diagnostic guidelines and psychological, social and cultural factors across different regions.
Hence, the aim of this study was to analyze the underlying
psychiatric diagnosis in children with psychogenic non-epileptic
seizures and to assess the outcome of their illness on follow-up.
2. Patients and methods
This is a retrospective hospital-based observational study,
which was carried out in the Departments of Child and Adolescent
Psychiatry, Neurology and Psychiatry at the National Institute of
Mental Health and Neurosciences (NIMHANS), a tertiary care
center in south India for neuropsychiatric disorders. All the
patients younger than 16 years, suspected to have PNES on videoEEG monitoring from August 2005 to August 2012 were included in
this study. Patients were admitted in the epilepsy-monitoring unit
(EMU) either to characterize the undiagnosed event (epileptic vs.
NES) or to characterize the semiology of the ictus as a part of the
evaluation of drug resistant epilepsy.
The video-EEG data of 44 children, who were suspected to have
PNES on VEEG, were retrieved from the server maintaining the
records and were reviewed by the two epileptologists (SS, PS)
independently. The differences in the interpretation of PNES
attacks between the two reviewers were sorted out after the
discussion. The criteria used to diagnose the NES were (a) at least
one typical attack should have been recorded on the VEEG, (b) no
EEG changes should be noticed during the event, (c) no post ictal
slowing on EEG, and (d) no evidence of any neurological condition
responsible for the events.20 Co-existent epilepsy was not an
exclusion criterion. At least one of the two expert child and
adolescent psychiatrists in the child and adolescent psychiatry
department (KJVS and SS) evaluated all 44 children either on
inpatient or outpatient basis. The clinical information was
obtained from the child and corroborated with the parent/
guardian. The diagnosis of PNES was made after a detailed
evaluation while assessing the psychiatric morbidity based on
DSM-5 criteria. The diagnosis of epilepsy in children with definite
evidence of PNES was based on both the clinical description of the
event and sometimes associated epileptiform discharges recorded
on the EEG.
The medical records of these children were reviewed in detail to
assess the nature of the illness, past history of seizures, associated
psychiatric co-morbidity, family history of psychiatric disorders or
epilepsy, final diagnosis and interventions done (pharmacological
or psychosocial). The outcome parameters noted were the number
of follow-up visits, duration of follow-up, response to the
psychotropic and/or psychosocial intervention, presence of symptoms at follow-up, return to the premorbid functional status as per
self/parental report, level of functioning as reflected by the social
involvement and scholastic work.
Written informed consent was taken from the parents/legal
guardians before the video-EEG recording. The data was tabulated
in a spreadsheet and descriptive analysis approach was used. The
means, standard deviations (SD) and medians were calculated. All
analyses were performed using R software (version 3.0.2).
3. Results
Out of a total of 1281 video-EEGs (adult: 763, children: 518)
performed during this period, 139 (10.8%) patients were suspected
to have NES based on the VEEG monitoring. Forty four of the 139
(31.7%) subjects were children below 16 years of age. These 44 four
children along with parent/caretaker completed the psychiatric
evaluation. Thirty four among 44 children were diagnosed as PNES.

The prevalence of PNES in children who underwent evaluation


with video-EEG was found to be 6.6% (34/518) in the present study.
3.1. Clinical and demographic details
The age at onset of PNES was 9 ! 3.7 years (range: 516 years;
median: 8.0 years), age at diagnosis was 12 ! 3.0 years (range: 818
years; median: 12.0 years). The mean frequency of the PNES attacks
was 69.3 ! 77.2 per month, ranging from 2 per month to as high as
300 per month (median: 30.0 per month). The mean duration of the
illness before the diagnosis of PNES was 0.83 ! 1.2 years (range: 16
years; median: 1.5 years).
3.2. Psychiatric diagnosis and co-morbidity
Thirty four children were diagnosed as having PNES and the
most common underlying psychiatric diagnosis was conversion
disorder (34/44; 77.3%). Stressors delineated among the 34 children diagnosed to have conversion disorder, most often related to
the scholastic difficulties (17/34; 50.0%) followed by the interpersonal relationship problems (9/34; 26.5%) and the familial/
parental stressors (8/34; 23.5%). More than one psychiatric
diagnosis was noted in 17 children (17/34, 50%). These were
intellectual disability in 8/34 (23.5%), depression in 5/34 (14.7%),
and specific learning disorder in 5/34 (14.7%) children. In total,
there were 13 (38.2%) children with developmental disability
either in the form of intellectual disability or specific learning
disorder.
The remaining 10 children (10/34, 22.7%) had non-epileptic
seizures with underlying diagnosis of Attention Deficit Hyperactivity Disorder (ADHD) (n = 3), gratification disorder (n = 2) and
other conditions like specific learning disorder, night terrors,
hypersomnolence, cold precipitating dyskinesia, isovaleric aciduria seen in one patient each.
4. Interventions
Psychosocial interventions included working with the family
and child. Reassurance along with acknowledging of the symptoms
and educating the parents with regard to the nature of the illness
and ways of handling the paroxysmal attacks was the most
common psychosocial intervention done in these children (34/34,
100%). Individual therapy, involving cognitive behavioral therapy
(CBT)/psychotherapy was used in 58.8% (20/34). Psychotropic
medications were used in 18 children (18/34, 52.9%). The most
common medication class used was selective serotonin reuptake
inhibitors (SSRI) (14/34; 41.2%), fluoxetine (n = 5/34; 14.7%),
escitalopram (n = 5/34; 14.7%), sertraline (n = 3/34; 8.8%) and
fluvoxamine (n = 1/34, 2.9%). Clonazepam was the most common
non-SSRI drug prescribed (7/34, 20.6%). Combined intervention
involving both pharmacological and psychosocial interventions
and was used in 18 children (18/34, 52.9%).
4.1. Follow-up and outcome
The mean follow-up was 10.1 ! 6.8 months. Twenty six (76.5%)
children did not have PNES attacks at the follow-up. Five (14.7%)
children had experienced reduced number of attacks while 3 (8.8%)
children continued to have attacks at the same or increased frequency
at the mean follow-up. Improvement was seen in the functional
status among 19 (19/26, 73.1%) children, who had no recurrence of
PNES after intervention.
Among children who underwent individual therapy, 12/20
(60.0%) responded well, 4/20 (20.0%) had partial response with
reduced frequency of attacks and 4/20 (20.0%) children had
persistent attacks with the same or increased frequency. Among

Please cite this article in press as: Rawat VS, et al. Co-morbidities and outcome of childhood psychogenic non-epileptic seizuresAn
observational study. Seizure: Eur J Epilepsy (2014), http://dx.doi.org/10.1016/j.seizure.2014.09.011

G Model

YSEIZ-2409; No. of Pages 4


V.S. Rawat et al. / Seizure xxx (2014) xxxxxx

children in combined intervention, 16 (16/18, 88.9%) had


improvement either with partial or complete resolution of the
attacks whereas among sixteen children, who received only
psychotherapy, 10 (10/16, 62.5%) children showed improvement.

frequency of attacks and one child had minimal improvement in


this group of the patients.

4.2. Anti-epileptic drug usage

The aim of this study was to assess the psychiatric diagnoses


and the outcome of childhood psychogenic non-epileptic seizures.
The prevalence of PNES in adults ranges from 2 to 33 per 100,000.21
In two Indian studies, the prevalence of PNES of 4% was noted in a
child psychiatry center and 10% among the children admitted for
the evaluation of refractory epilepsy.22 In the present study, the
prevalence of PNES in children was around 6.6%, which is
remarkably close to the prevalence reported by Kotagal et al.
(62/883, 7%).11 There was no gender differences in the present
study as against a female predominance reported in the previously
(76% and 61% respectively).19,22
A previous study analyzed the outcome of PNES in 167 adults
over a span of 10 years based on questionnaire, which showed that
71.2% of the patients in their cohort continued to have PNES.18 An
Indian study looked into the outcome of 17 children with PNES
after 36 months of the diagnosis, where 82.4% of the children
either recovered fully or had more than 50% reduction in the
symptoms.19 The present study analyzed 34 children aged less
than 16 years with diagnosis of PNES confirmed on VEEG, we found
76.5% of the children were free of the symptoms at the end of the
follow-up of 10.1 ! 6.8 months. Scholastic difficulties being the most
common stressor, was identified in 50% of children, which is lower
than that reported in a previous study (82%).22
Patients with PNES are usually misdiagnosed as epilepsy and
started on AEDs. Various studies have reported variable rates of comorbid epilepsy and PNES ranging from 18% to 31%.23,24 In this
study, 16% of children had comorbid epilepsy, which is slightly
lower than the reported range. Previous studies reported that 35%
of children with PNES were unnecessarily taking AEDs.23 In the
present study, 82% children were taking AEDs prior to the
evaluation, which reduced to 16% after appropriate evaluation
and intervention. An unfavorable prognosis for the children who
have co-morbid epilepsy has been reported (54.0% in co-morbid
group vs. 71.0% in only PNES group)15 which is comparable to the
present study (50.0% in co-morbid vs. 70% in PNES only group).
In the current study, conversion disorder was the psychiatric
diagnosis in the children with PNES, which is similar to various
previous reports.24,25 Reassurance along with educating the parents
or family about the disorder is the mainstay of the treatment.26 The
main stay of intervention used in PNES is psychological intervention,
which was used in 65% of the children in the present study. The role
of psychosocial interventions is invaluable in managing the
conversion disorder and other co-morbid psychiatric disorders in
the pediatric age group. In the present study, 76.5% of children
became asymptomatic, which is comparable to the previous studies,
which have reported a good outcome (80%) in children at 36
months follow-up and at the discharge from the hospital.19,22,27
indicating that the conversion disorder as PNES has good prognosis
as compared to the other psychiatric diagnosis.
Physiological causes, like gratification disorder, ADHD, sleep
disorders and some metabolic disorders like isovaleric aciduria
may also appear as PNES at the first visit, as reported in 10 children
in the present study. Careful clinical history and investigations can
help in diagnosing such disorders.
The limitation of this study is its retrospective design, small
sample size and rather short follow-up. The diagnosis of PNES in
children is very important for proper management. A comprehensive management involving psychosocial interventions and in
some children add-on psychotropic medication results in cessation
of psychogenic non-epileptic attacks. Hence, accurate diagnosis of
PNES at proper time is crucial for the development of the child.

Twenty nine (85.3%) children were receiving anti-epileptic


drugs (AEDs) before the VEEG recording. Eighteen (52.9%) children
were receiving >1 AEDs while 11 children (32.4%) were on single
AED at the time of the initial evaluation.
4.3. Co-morbid epilepsy
Eight (8/34, 23.5%, M:F = 5:3) children were diagnosed to have
both psychogenic non-epileptic seizures and epilepsy. The mean
age at onset for epilepsy and PNES was 7.6 ! 3.2 years and
12 ! 3.5 years respectively. They were followed up for 10 ! 6 months
(range: 018 months). The commonest seizure type was complex
partial (n = 4, 50.0%) followed by generalized (n = 3, 37.5%) and simple
partial (n = 1, 12.5%). EEG abnormalities were noted in all 8 children.
Five children (62.5%) were on two AEDs, 2 (25.0%) were on three AEDs
while 1 (12.5%) child was on four AEDs. The abnormalities noted on
the brain MRI in this cohort of patient were: mesial temporal sclerosis
(MTS): 2 children; focal lesions: 3 children (right inferior frontal gyrus
gliosis, right inferior frontal gyrus cortical dysplasia, right frontal ring
enhancing lesion), while MRI was normal in rest of 3 children
(Tables 1 and 2).
All the 8 children with combined epilepsy with PNES had
underlying conversion disorder. Psychological assessments were
carried out in four children (50.0%). One child (12.5%) was found to
have low intelligence (mild intellectual disability) while 3 children
(37.5%) had co-morbid psychiatric disorder, e.g. ADHD, intellectual
disability, specific learning disorder, depression and adjustment
disorder, which could be responsible for their PNES. Four children
had no recurrence of the PNES attacks, while three had reduced

Table 1
Socio-demographic details of children with PNES.
Patient-related characteristics in PNESs (n = 34)

N (%)

Education
Preschool or primary school
High school
Pre-university
Positive history of anxiety disorder
Positive history of mood disorder
Positive history of self-harm
Positive family history of seizures
Positive family history of psychiatric disorders
Co-existent neurological deficit
Coexistent systemic complaint
Headache

19
13
2
4
4
4
7
3
4
9
5

(55.9)
(38.2)
(5.9)
(11.8)
(11.8)
(11.8)
(20.6)
(8.8)
(11.8)
(26.5)
(14.7)

Table 2
Details of psychiatric diagnosis among children.
Diagnosis (total = 44)
PNES
Conversion disorder
Other diagnosis
Attention Deficit Hyperactivity Disorder (ADHD)
Gratification disorder
Specific learning disorder
Night terrors
Hypersomnolence
Cold precipitating dyskinesia
Isovaleric aciduria

N (%)
34 (77.3)
3
2
1
1
1
1
1

(6.8)
(4.5)
(2.3)
(2.3)
(2.3)
(2.3)
(2.3)

5. Discussion

Please cite this article in press as: Rawat VS, et al. Co-morbidities and outcome of childhood psychogenic non-epileptic seizuresAn
observational study. Seizure: Eur J Epilepsy (2014), http://dx.doi.org/10.1016/j.seizure.2014.09.011

G Model

YSEIZ-2409; No. of Pages 4


4

V.S. Rawat et al. / Seizure xxx (2014) xxxxxx

Funding
None.
Conflict of interest
None declared.
Acknowledgements
We acknowledge the contributions of the staffs of the epilepsymonitoring unit, Department of Neurology. Last but not the least,
we are grateful to the subjects for allowing us to carry out such
study.
References
1. Bodde NM, Brooks JL, Baker GA, Boon PA, Hendriksen JG, Aldenkamp AP.
Psychogenic non-epileptic seizures diagnostic issues: a critical review. Clin
Neurol Neurosurg 2009;111:19.
2. Reuber M. Psychogenic nonepileptic seizures: diagnosis, aetiology, treatment
and prognosis. Schweiz Arch Neurol Psychiatr 2005;156:4757.
3. Gates JR. Non-epileptic seizures classification co-existence with epilepsy:
diagnosis, therapeutic approaches and consensus. Epilepsy Behav 2002;3:
2833.
4. Rao TS. Paroxysmal non-epileptic seizures in children: recognition and approach to diagnosis. Adv Clin Neurosci Rehabil 2012;12:1720.
5. Beach R, Reading R. The importance of acknowledging clinical uncertainty in the
diagnosis of epilepsy and non-epileptic events. Arch Dis Child 2005;90:121222.
6. Desai P, Talwar D. Non-epileptic events in normal and neurologically handicapped children: a video-EEG study. Pediatr Neurol 1992;8:1279.
7. Bye AM, Nunan J. Video EEG analysis of non-ictal events in children. Clin Exp
Neurol 1992;29:928.
8. Bye AM, Kok DJ, Ferenschild FT, Vles JS. Paroxysmal non-epileptic events in
children: a retrospective study over a period of 10 years. J Paediatr Child Health
2000;36:2448.
9. Uldall P, Alving J, Hansen LK, Kibaek M, Buchholt J. The misdiagnosis of epilepsy
in children admitted to a tertiary epilepsy centre with paroxysmal events. Arch
Dis Child 2006;9:21921.

10. Seneviratne U, Briggs B, Lowenstern D, DSouza W. The spectrum of psychogenic


non-epileptic seizures and comorbidities seen in an epilepsy monitoring unit. J
Clin Neurosci 2011;18:3613.
11. Kotagal P, Costa M, Wyllie E, Wolgamuth B. Paroxysmal non-epileptic events in
children and adolescents. Pediatrics 2002;110:e46.
12. Kuyk J, Leijten F, Meinardi H, Spinhoven. Van Dyck R. The diagnosis of psychogenic non-epileptic seizures: a review. Seizure 1997;6:24353.
13. Allen JE, Ferrie CD, Livingston JH, Feltbower RG. Recovery of consciousness after
epileptic seizures in children. Arch Dis Child 2007;92:3942.
14. Moore PM, Baker GA. Non-epileptic attack disorder: a psychological perspective. Seizure 1997;6:42934.
15. Irwin K, Edwards M, Robinson R. Psychogenic non-epileptic seizures: management and prognosis. Arch Dis Child 2000;82:4748.
16. Wyllie E, Friedman D, Rothner AD, Luders H, Dinner D, Morris H, et al.
Psychogenic seizures in children and adolescents: outcome after diagnosis
by ictal video and electroencephalographic recording. Pediatrics 1990;85:
4804.
17. Ettinger AB, Devinsky O, Weisbrot DM, Ramakrishna RK, Goyal A. A comprehensive profile of clinical, psychiatric, and psychosocial characteristics of
patients with psychogenic nonepileptic seizures. Epilepsia 1999;40:12928.
18. Reuber M, Pukrop R, Bauer J, Helmstaedter C, Tessendorf N, Elger CE. Outcome
in psychogenic nonepileptic seizures: 1 to 10-year follow-up in 164 patients.
Ann Neurol 2003;53:30511.
19. Chinta SS, Malhi P, Singhi P, Prabhakar S. Clinical and psychosocial characteristics of children with non-epileptic seizures. Ann Indian Acad Neurol 2008;11:
15963.
20. Seneviratne U, Reutens D, DSouza W. Stereotypy of psychogenic nonepileptic
seizures: insights from video-EEG monitoring. Epilepsia 2010;51:115968.
21. Benbadis SR, Allen Hauser W. An estimate of the prevalence of psychogenic
non-epileptic seizures. Seizure 2000;9:2801.
22. Prabhuswamy M, Jairam R, Srinath S, Girimaji S, Seshadri SP. A systematic chart
review of inpatient population with childhood dissociative disorder. J Indian
Assoc Child Adolesc Ment Health 2006;2:727.
23. Dhiman V, Sinha S, Rawat VS, Vijaysagar KJ, Thippeswamy H, Srinath S, et al.
Children with psychogenic non-epileptic seizures (PNES): a detailed semiologic
analysis and modified new classification. Brain Dev 2014;36:28793.
24. Malhi P, Singhi P. Clinical characteristics and outcome of children and adolescents with conversion disorder. Indian Pediatr 2002;39:74752.
25. Srinath S, Bharat S, Girimaji S, Seshadri S. Characteristics of a child inpatient
population with hysteria in India. J Am Acad Child Adolesc Psychiatry 1993;32:
8225.
26. Leung AK, Robson WL. Childhood masturbation. Clin Pediatr (Phila) 1993;32:
23841.
27. Nechay A, Ross LM, Stephenson JB, ORegan M. Gratification disorder (infantile
masturbation): a review. Arch Dis Child 2000;89:2256.

Please cite this article in press as: Rawat VS, et al. Co-morbidities and outcome of childhood psychogenic non-epileptic seizuresAn
observational study. Seizure: Eur J Epilepsy (2014), http://dx.doi.org/10.1016/j.seizure.2014.09.011