Sudden unexpected death after acute symptomatic seizures in a patient on mechanical ventilation

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Martinez et al. Acta Epileptologica (2020) 2:23 https://doi.org/10.1186/s42494-020-00032-y

CASE REPORT

Open Access

Sudden unexpected death after acute symptomatic seizures in a patient on mechanical ventilation Raisa C. Martinez1, Naoum P. Issa1, Shasha Wu1, Xi Liu2, Sandra Rose1 and James X. Tao1*

Abstract Background: The mechanism of sudden unexpected death in epilepsy remains poorly understood. Seizure induced cardiac arrhythmia, central and obstructive apneas have been proposed as possible causes of death. Here we report a unique case of seizure related sudden unexpected death in a patient whose airway was fully protected by intubation and mechanic ventilation in the absence of fatal cardiac arrhythmia. Case presentation: A 70-year-old woman was undergoing mechanical ventilation and videoelectroencephalography (EEG) monitoring following two convulsive seizures with ictal hypoventilation and hypoxemia. Several hours after intubation, she suffered another generalized tonic clonic seizure lasted for 3 min and developed postictal generalized EEG suppression in the presence of stable vital signs with SpO2 > 90%. EEG suppression persisted throughout the postictal phase. There was a significant fluctuation of systolic blood pressure between 50 and 180 mmHg with several bouts of hypotension < 60 mmHg. She remained unresponsive after the convulsive seizure and died of diffuse cerebral edema 12 h later. Autopsy revealed no clear cause of death, except for possible hypoxic and ischemic injury leading to the diffuse cerebral edema. Conclusion: Given the reliable periictal airway protection, neither seizure induced central apnea nor obstructive apnea appeared to be the direct cause of death in this unique case. In the absence of fatal cardiac arrhythmia, diffuse cerebral edema secondary to seizure-induced autonomic dysfunction, hypotension and hypoxemia might be the cause of death, highlighting the etiological heterogeneity of sudden unexpected death in epilepsy. Keywords: Convulsive seizure, SUDEP, Mechanic ventilation, Apnea, Prone position

Background Sudden unexpected death in epilepsy (SUDEP) is the leading cause of premature death in patients with chronic refractory epilepsy [1]. Over the last several decades, several risk factors for SUDEP have been proposed including chronic uncontrolled epilepsy, the duration of epilepsy, young age, male sex and intellectual disability [2–4]. Pooled data indicated that the frequency of generalized tonic clonic seizures (GTCS) is the most important risk factor for SUDEP [5]. Lack of night-time * Correspondence: [email protected] 1 Adult Epilepsy Center, Department of Neurology, The University of Chicago, 5841 South Maryland Ave. MC2030, Chicago, IL 60637, USA Full list of author information is available at the end of the article

supervision and absence of nocturnal listening device are also important risk factors [6]. Nevertheless, the mechanisms of SUDEP have remained poorly understood. SUDEP commonly occurs during sleep and in bed with most cases being unwitness