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Table of Contents    
CASE REPORT
Year : 2021  |  Volume : 69  |  Issue : 3  |  Page : 737-739

Cardiac Arrest in Frontal Lobe Epilepsy: A Potential Cause of Sudden Unexpected Death in Epilepsy


Department of Neurology and Neuroscience Center, First Hospital of Jilin University, Changchun, China

Date of Submission29-May-2017
Date of Decision08-Aug-2019
Date of Acceptance08-Aug-2019
Date of Web Publication24-Jun-2021

Correspondence Address:
Dr. Weihong Lin
Department of Neurology and Neuroscience Center, First Hospital of Jilin University, No. 71, Xinmin Street, Changchun - 130 000
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.319218

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 » Abstract 


Background: Mechanisms involved in the causation of sudden unexpected death in epilepsy (SUDEP) are not well understood. Ictal asystole has been identified as a cause of SUDEP in patients with temporal lobe epilepsy. Herein, we report a patient who developed cardiac arrest during the ictal period of frontal lobe epilepsy.
Case Report: A 35-year-old man presented with a history of progressive nocturnal stiffness in the left lower extremity since 6 years and that of paroxysmal episodes of altered consciousness. In the last 2 years, he sustained epileptic seizures which presented as closed eyes, wheezy phlegm in the throat, facial pallor, moist cold skin, clenched fists, and limb stiffness; the episodes lasted 3–4 min. The seizures did not respond to antiepileptic therapy. Twenty four-hour electrocardiography monitoring showed transient atrial tachycardia, supraventricular premature beats, and cardiac arrest.
Conclusion: Frontal lobe epilepsy may be a potential cause of SUDEP. Clinician should be aware of this condition.


Keywords: Asystole, cardiac arrest, case report, frontal lobe epilepsy, sudden unexpected death in epilepsy
Key Message: The occurrence of ictal asystole and SUDEP in Frontsl lobe epilepsy is extremely rare. The treating neurologist should be aware of this condition.


How to cite this article:
Zhang X, Cui L, Lin W. Cardiac Arrest in Frontal Lobe Epilepsy: A Potential Cause of Sudden Unexpected Death in Epilepsy. Neurol India 2021;69:737-9

How to cite this URL:
Zhang X, Cui L, Lin W. Cardiac Arrest in Frontal Lobe Epilepsy: A Potential Cause of Sudden Unexpected Death in Epilepsy. Neurol India [serial online] 2021 [cited 2021 Jul 28];69:737-9. Available from: https://www.neurologyindia.com/text.asp?2021/69/3/737/319218




Sudden unexpected death in epilepsy (SUDEP) refers to the sudden unexplained death of a person with epilepsy, in the absence of any obvious traumatic, toxicological, or anatomical cause.[1] The exact cause of SUDEP is still unknown. The most common reported causes of SUDEP include seizure-induced hypoventilation and ictal cardiac arrhythmias such as bradycardia and asystole.[2] We report a patient who sustained cardiac arrest during the ictal period of frontal lobe epilepsy.


 » Case History Top


A 35-year-old, previously healthy man presented to us in May 2016 with a history of progressive nocturnal stiffness in the left lower extremity since 6 years and paroxysmal episodes of altered consciousness. The left-leg stiffness occurred in clusters of 2- to 3-week duration at intervals of ~3 months. During the episodes, he experienced brief intermittent nocturnal attacks which lasted for 2–3 s. During the initial 4 years, there was no disturbance of consciousness. In November 2014, the patient suffered a seizure during which manifested as eyes closed, wheezy phlegm in the throat, facial pallor, moist cold skin, clenched fists, and limb stiffness; the seizure lasted 3–4 min. His relatives volunteered that he opened his eyes and responded to verbal commands during the seizure; however, the patient had no memory of the seizure. In April 2016, two such seizures occurred early in the morning. During the second attack, ambulatory electrocardiography (ECG) indicated cardiac arrest. A diagnosis of epilepsy was made, and oxcarbazepine was prescribed at a dosage of 150 mg twice daily. The patient did not respond to antiepileptic treatment. There was no history of trauma or infection; the family history was unremarkable. Twenty-four-hour ECG monitoring showed transient atrial tachycardia, supraventricular premature beats (34 times with two repetitive beats), and cardiac arrest (3 times) [Figure 1]. Electroencephalogram (EEG) monitoring, brain magnetic resonance imaging, and cardiac electrophysiology showed no significant abnormality [Figure 2]. Ultrasonic cardiogram revealed atrial enlargement, mitral regurgitation, tricuspid regurgitation, and abnormal systolic function of the left ventricle.
Figure 1: Dynamic electrocardiogram showing cardiac asystole

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Figure 2: Electroencephalogram showing no abnormality

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 » Discussion Top


Epilepsy is a common clinical syndrome with recurrent neuronal discharges in the cerebral cortex. Patients with epilepsy are known to be at a higher risk of premature death as compared with that in the general population, and SUDEP is the most common cause. SUDEP accounts for ~7.5% to 17% of all epilepsy-related deaths and for 50% of all deaths in patients with refractory epilepsy.[3] In recent years, an association between epileptic seizures and cardiac dysfunction has been reported.[4] Ictal cardiac arrhythmias, especially bradycardia and asystole, have been shown to be associated with epileptic seizures. Additionally, ictal cardiovascular abnormalities are also common autonomic manifestations of epilepsy. Epileptic activity originating from amygdala, gyrus cinguli, insular cortex, frontopolar, or frontal-orbital regions was proposed to be a cause of cardiac dysfunction including supraventricular tachycardia, sinus tachycardia, sinus bradycardia, sinus arrest, atrioventricular block, and asystole.[5] Epilepsy-related tachycardia is more common in patients with temporal lobe epilepsy, especially those that originate from the medial temporal lobe.[6] We speculate that this may be due to proximity of the medial temporal lobe to the cortical and subcortical centers that regulate the autonomic nervous function (such as insular lobe). Therefore, epileptic discharge could involve the autonomic efferent pathways and cause tachycardia. Compared with tachycardia, post-ictal bradycardia and asystole are relatively rare but severe complications.[7] Epilepsy-related ictal asystole refers to the transient cessation of cardiac electrical activity during an epileptic episode; the condition is defined by the absence of ventricular complexes for >4 s during an ongoing seizure episode confirmed on electroencephalography.[8] In a previous systematic review, the prevalence of ictal asystole in patients with refractory epilepsy was reported to be 0.318%; further, in the majority of patients, the seizure onset (91%) was in the temporal lobe. Moreover, nearly all episodes of ictal asystoles were reported to be self-limiting.[9] In a study by Schuele et al., 80% of the patients with ictal asystole had temporal lobe seizures while 20% of these had extratemporal lobe epilepsy.[10] Ictal asystole caused by frontal lobe epilepsy is extremely rare.

In this case, the patient presented with nocturnal, cluster-onset stiffness in the left lower extremity. Although EEG monitoring showed no peri-ictal seizure activity, the clinical manifestations suggest that the seizure originated from the supplementary motor area in the frontal lobe, which is consistent with a diagnosis of frontal lobe epilepsy. The negative EEG findings might be related to the deep location of the neuronal discharge pathway, which is liable to be missed by scalp electrodes. In the recent 2 years, we observed three grand mal seizures, and a diagnosis of Adams-stokes syndrome caused by asystole was suspected. The patient had no history of cardiovascular disease, and thus we believe that the asystole might be related to frontal lobe epilepsy. Frontal lobe epilepsy is the second most common type of epilepsy after temporal lobe epilepsy, and both forms are characterized by occurrence of partial seizures. The symptoms and clinical manifestations of frontal lobe epilepsy are variable depending on which specific area of the frontal lobe is affected. However, frontal lobe epilepsy manifesting as ictal asystole is rare. As ictal asystole has been identified as a potential cause of SUDEP, we should be aware of the possible link between SUDEP and frontal lobe epilepsy.

However, the optimal preventive and therapeutic strategies for epilepsy-related ictal asystole needs further research in a larger cohort. Early identification of epileptic seizures and potential cardiac dysfunction as well as the risk of SUDEP should be highlighted.


 » Conclusion Top


We presented a patient with frontal lobe epilepsy who developed ictal asystole; our findings suggest a possible linkage between frontal lobe epilepsy and cardiac arrest. Clinicians should be aware of this condition as epilepsy-related cardiac arrest may be associated with SUDEP.

Acknowledgements

The authors thank to Medjaden Bioscience Limited in proofreading the manuscript.

Financial support and sponsorship

National Nature Science Foundation of China (No. 81571264).

Conflicts of interest

There are no conflicts of interest.



 
 » References Top

1.
Nashef L. Sudden unexpected death in epilepsy: Terminology and definitions. Epilepsia 1997;38:S6-8.  Back to cited text no. 1
    
2.
Shorvon S, Tomson T. Sudden unexpected death in epilepsy. Curr Neurolo Neurosci Rep 2010;10:319-26.  Back to cited text no. 2
    
3.
Terra VC, Cysneiros R, Cavalheiro EA, Scorza FA. Sudden unexpected death in epilepsy: From the lab to the clinic setting. Epilepsy Behav 2013;26:415-20.  Back to cited text no. 3
    
4.
Ravindran K, Powell KL, Todaro M, O'Brien TJ. The pathophysiology of cardiac dysfunction in epilepsy. Epilepsy Res 2016;127:19-29.  Back to cited text no. 4
    
5.
Rocamora R, Kurthen M, Lickfett L, Von OJ, Elger CE. Cardiac asystole in epilepsy: Clinical and neurophysiologic features. Epilepsia 2003;44:179-85.  Back to cited text no. 5
    
6.
Garcia M, D'Giano C, Estellés S, Leiguarda R, Rabinowicz A. Ictal tachycardia: Its discriminating potential between temporal and extratemporal seizure foci. Seizure 2001;10:415-9.  Back to cited text no. 6
    
7.
Lanz M, Oehl B, Brandt A, Schulze-Bonhage A. Seizure induced cardiac asystole in epilepsy patients undergoing long term video-EEG monitoring. Seizure 2011;20:167-72.  Back to cited text no. 7
    
8.
Moseley BD, Ghearing GR, Munger TM, Britton JW. The treatment of ictal asystole with cardiac pacing. Epilepsia 2011;52:16-9.  Back to cited text no. 8
    
9.
van der Lende M, Surges R, Sander JW, Thijs RD. Cardiac arrhythmias during or after epileptic seizures. J Neurol Neurosurg Psychiatry. 2016;87(1):69-74.  Back to cited text no. 9
    
10.
Schuele SU, Bermeo AC, Alexopoulos AV, Locatelli ER, Burgess RC, Dinner DS, et al. Video-electrographic and clinical features in patients with ictal asystole. Neurology 2007;69:434-41.  Back to cited text no. 10
    


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