YEBEH-05786; No of Pages 3

Epilepsy & Behavior xxx (2018) xxx–xxx

Contents lists available at ScienceDirect

Epilepsy & Behavior

journal homepage: www.elsevier.com/locate/yebeh

Successful treatment of reflex epilepsy with praxis induction by stimulus

avoidance only
Zhe-Ren Tan, Fa-Fa Tian, Xiao-Yan Long, Chen Zhang, Yan-Yan Feng, Si-Yuan Zhang, Guo-Liang Li ⁎
Department of Neurology, Xiangya Hospital, Central South University, 87 Xiangya Road, Changsha 410008, China

a r t i c l e i n f o a b s t r a c t

Article history: Purpose: Reflex epilepsy is a type of epilepsy with seizures that are consistently triggered by a specific stimulus.
Received 9 January 2018 Zipai is a Chinese ancient card game which has been popular in Southern China for hundreds of years. We sought
Revised 30 April 2018 to report and characterize clinical features of patients with reflex epilepsy evoked by playing Zipai.
Accepted 30 April 2018
Methods: We collected and analyzed clinical data of patients with Zipai-induced epilepsy. Patients were regarded
Available online xxxx
as having Zipai-induced epilepsy if they suffered at least two seizure attack during the course of playing Zipai.
Keywords: Prolonged electroencephalography (EEG) and brain magnetic resonance imaging (MRI) were applied to all
Nonpharmacological conservative treatment patients. All patients were advised to avoid watching and playing Zipai games in daily life, instead of using anti-
Cognitive activity epileptic drugs. The seizure outcome was assessed during outpatient visits and by telephone contact.
Reflex epilepsy Results: Five patients were included in this study. No spontaneous seizures occurred in all five patients. No
Zipai game patients had experienced myoclonic and coexistent absences with generalized tonic–clonic seizures (GTCS).
Praxis induction All patients had normal MRI and prolonged EEG findings. All patients were advised to avoid the Zipai game,
and became seizure-free without medication during the follow-up period (mean 5.4 years, range 3.5–7 years).
Conclusion: Zipai-induced epilepsy may be an unreported subtype form of reflex epilepsy with praxis induction.
Nonpharmacological conservative treatment plays a significant role in the treatment of reflex epilepsy.
© 2018 Elsevier Inc. All rights reserved.

1. Introduction Patients were regarded as having Zipai-induced epilepsy if they suffered

Reflex epilepsy can be reproducibly or immediately triggered by
some well-defined sensory or complex cognitive stimuli [1]. In 1956, a
case of reflex epilepsy induced by reading was reported by Bickford et
al. [2]. An increasing amount of cases on reflex epilepsy induced by
cognitive activities has been reported ever since. The specific aspects of
these cognitive stimuli were noticeably multitudinous, including calcula-
tion, reading, thinking, Mah-Jong, card games, and video games [1,3–8].
Zipai is an ancient Chinese card game which has been popular in
Southern China for hundreds of years. However, Zipai-induced epilepsy
has never been reported in any publication. In this study, we sought to
report and characterize clinical features of patients with reflex epilepsy
evoked by playing Zipai.
at least two seizures during the course of playing Zipai.
Prolonged electroencephalography (EEG) and brain magnetic reso-
nance imaging (MRI) were applied to all patients. Previous studies had
demonstrated that nonpharmacological conservative (NPC) treatment
may be as effective as pharmacotherapy in patients with pure reflex ep-
ilepsy [10], so all patients were advised to avoid watching and playing
Zipai games in daily life instead of using antiepileptic drugs (AEDs).
The seizure outcome was assessed during outpatient visits and by tele-
phone contact. Patients were excluded if they had seizures that occurred
during the course of other occasions rather than watching and playing a
Zipai game. Age, gender, clinical diagnosis, types of seizures, prolonged
EEG, brain MRI, and results of the follow-up study were summarized.

2. Patients and methods 3. Results

We collected and analyzed the clinical data of patients with Zipai-in-
duced epilepsy from November 2012 to December 2015 in the database
of the Department of Neurology, Xiangya Hospital, Central South Uni-
versity, Changsha, China. Seizure pattern was classified according to
the International League Against Epilepsy (ILAE) classification [9].
⁎ Corresponding author.
E-mail address: guoliangli09@126.com (G.-L. Li).
Five patients were included in this study (5 males; average age: 30.8
± 8.98 years, range: 19–44 years). The history of Zipai-induced epilepsy
varied from 2 to 30 years (mean: 13.2 years). No spontaneous seizures
occurred in all five patients, and seizures of all patients were not evoked
by other types of complex cognitive activities such as playing Mah-Jong
and poker. Demographics and clinical characteristics of all cases are
listed in Table 1. No patients had experienced myoclonic and coexistent
absences with generalized tonic–clonic seizures (GTCS).

https://doi.org/10.1016/j.yebeh.2018.04.030
1525-5050/© 2018 Elsevier Inc. All rights reserved.

Please cite this article as: Tan Z-R, et al, Successful treatment of reflex epilepsy with praxis induction by stimulus avoidance only, Epilepsy Behav
(2018), https://doi.org/10.1016/j.yebeh.2018.04.030
2 Z.-R. Tan et al. / Epilepsy & Behavior xxx (2018) xxx–xxx

Table 1
Demographics and clinical characteristics of patients with Zipai-induced epilepsy.

No Age/sex Age at onset Risk FHX Spontaneous Stimuli Seizure latency Description Other specific Seizure frequency
(years) factors seizure after stimuli activation (n/year)

1 58/M 28 NO NO NO Zipai 5–6 h Transient dizziness, jerking of the left limb NO 3–4/year
during the game and evolving into GTCS
2 34/M 32 NO NO NO Zipai 6–7 h Difficulty with concentrating and left NO 3/year
limb numbness followed by GTCS
3 39/M 31 NO NO NO Zipai 4–5 h GTCS NO 1/year
4 39/M 19 NO NO NO Zipai 3–4 h Mental clouding and right forearm clonus Playing 5–6/year
during Zipai game and evolving into GTCS Mah-Jong
5 50/M 44 NO NO NO Zipai 8–12 h GTCS NO 1–2/year

GTCS: generalized tonic–clonic seizure; FHX: family history for epilepsy.

Table 2 Seizure latency varied from 3 to 12 h in these cases, suggesting that

Prolonged EEG, brain MRI, and treatment in five patients with Zipai-induced epilepsy. epileptic seizures triggered by this game require a long-term cognitive
Patients Prolonged EEG Routine brain Treatment Prognosis F/U activity including memorizing and calculating. Besides that, all patients
finding MRI strategy (year) reported that they routinely started playing the game in the evening,
1 NL NL AZG SF 3.5 and fatigue and sleep deprivation are present when playing Zipai,
2 NL NL AZG SF 4.3 which have been regarded as nonspecific facilitating factors of seizures
3 NL NL AZG SF 5.6 [11,12]. Furthermore, all patients demonstrated that they were exposed
4 NL NL AZG SF 6.6
to mental stress and secondhand smoke during the Zipai gambling. Pre-
5 NL NL AZG SF 7
vious studies had reported that emotional stress and acute secondhand
NL: normal; AZG: avoiding the Zipai game; SF: seizure-free; F/U: follow-up period. smoke are risk factors for seizure occurrence [13,14]. Those factors are
associated with a large probability of high seizure susceptibility and
therefore cause potential recurrence risk.
All patients had normal MRI and prolonged EEG findings (Table 2). Clinical characteristics of cognitive task-induced epilepsy are incon-
Patient 5 had received AED (valproic acid) for a few years before coming sistent. Goossens et al. summarized the clinical features of epilepsy in-
to the epilepsy outpatient department of Xiangya Hospital; however, duced by cognitive stimuli as the preponderance of males; onset at
the AED was ineffective. All patients were advised to avoid the Zipai adolescence; a high proportion of patients with generalized tonic–
game and became seizure-free without medication during the follow- clonic, myoclonic, or absence seizures; coexistence with spontaneous
up period (mean: 5.4 years, range: 3.5–7 years). seizures; and abnormality in the EEG findings. They considered that re-
flex epilepsy induced by complex cognitive tasks belongs to a clinical
spectrum of idiopathic generalized epilepsy (IGE) [15]. Today, this
4. Discussion
type of reflex epilepsy is usually considered as a manifestation of praxis
induction which in turn is closely related to juvenile myoclonic epilepsy
Zipai is an ancient and popular gambling game in Southern China,
(JME) [16]. Cognitive effort can cause seizures in patients with JME and
the popularity of which is analogous to poker in Western countries. Re-
a high proportion of these patients have praxis-induced reflex myocloni
flex seizure induced by playing Zipai has not yet been reported.
[17–20]. However, in consideration of noticeable clinical characteristics,
Traditional Zipai cards consist of 80 small rectangular cards with
we believe that a seizure induced by Zipai is a subset of reflex epilepsy
faces showing various numbers from one to ten in Chinese (Fig. 1). In-
with praxis induction, and it is improper to place Zipai-induced epilepsy
depth calculation, judgment, and memorization are key elements in
in the spectrum of JME. First of all, the initial seizure of our patients hap-
winning the gamble. It is usually played by three players. The basic
pened in adulthood. Secondly, no patients presented myocloni in the
rule of Zipai is analogous to Mah-Jong. Gamblers change the cards and
course of the seizures, and there was no abnormality in their prolonged
sort their sequence after getting a card from the draw deck in turn.
EEG and brain MRI findings. Family history of epilepsy and spontaneous
The first player who sorts his/her own cards into specific sequences
events were not reported for any patient. These clinical features were
wins the game.
similar to Mah-Jong-induced epilepsy [6,21]. Furthermore, avoiding
the triggering stimuli is not always effective for patients with JME
with reflex epileptic traits, and a large number of these patients have
seizures usually controlled by an AED [22]. On the contrary, all patients
with Zipai-induced epilepsy became seizure-free without any medica-
tion during the follow-up study.
Because of misdiagnosis and low seizure frequency, most patients
underwent inappropriate treatment or ignored their seizures alto-
gether. On the one hand, no patient had a spontaneous, unprovoked sei-
zure. On the other hand, the utility of AEDs for game-induced seizures is
controversial [6,23,24]; in addition, the drug treatment might also cause
side effects and impose economic burden on patients. Although NPC
treatment was often used as an addition to pharmacological treatment
in most cases, significant effectiveness was observed in patients with re-
flex epilepsy who were treated with NPC treatment alone. For example,
in photosensitive patients, seizures are often precipitated by watching
television. These can be avoided by viewing from a distance and wear-
ing sunglasses [25]. In addition, in a case report about Sudoku puzzle-in-
duced seizures, the patient stopped solving Sudoku puzzles and has
Fig. 1. The appearance of a Zipai card. been seizure-free for more than 5 years [26]. This indicates that control

Please cite this article as: Tan Z-R, et al, Successful treatment of reflex epilepsy with praxis induction by stimulus avoidance only, Epilepsy Behav
(2018), https://doi.org/10.1016/j.yebeh.2018.04.030
 Z.-R. Tan et al. / Epilepsy & Behavior xxx (2018) xxx–xxx
of a specific provocative factor plays a significant role in the NPC treat-
ment of reflex epilepsy. Based on the above reasons, we advised patients
to only avoid Zipai. According to the result of the follow-up study, all pa-
tients became seizure-free for several years after accepting our sugges-
tion, which further confirmed the effectiveness and significance of NPC
treatment for this form of reflex epilepsy.
5. Conclusions
In summary, the Zipai game is an ancient and popular gambling
game in Southern China. We summarized the clinical characteristics of
patients with epilepsy evoked by the Zipai game. Patients' general clin-
ical features are as follows: older age at onset, high proportions of pa-
tients with GTCS, nonexistence of spontaneous events, and normality
of prolonged EEG and brain MRI findings. Simply avoiding the Zipai
game avoided seizures, therefore AED treatment was not needed. The
above several characteristics demonstrate that Zipai-induced epilepsy
may be an unreported subtype form of reflex epilepsy with praxis in-
duction. Nonpharmacological conservative treatment plays a significant
role in the treatment of this form of reflex epilepsy.
Acknowledgments
Zhe-Ren Tan was involved in the conception and writing of the man-
uscript. Yan-Yan Feng, Chen Zhang, and Si-Yuan Zhang contributed to
the collection of patients' clinical data and longitudinal follow-up. Fa-
Fa Tian and Xiao-Yan Long provided critical questions and suggestions
to the manuscript. Guo-Liang Li supervised all aspects of the study, crit-
ically reviewed and revised the manuscript, and approved the final
manuscript as submitted. All authors read and approved the final man-
uscript. All authors thank Jiayin Lu for manuscript polishing. This study
was supported by National Natural Science Foundation of China
(81571276).
Conflicts of interest
The authors have no conflicting interests to declare.
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