Abstract

BackgroundThe 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 presentationA 70-year-old woman was undergoing mechanical ventilation and video-electroencephalography (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.ConclusionGiven 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.

Highlights

  • The mechanism of sudden unexpected death in epilepsy remains poorly understood

  • Overnight continuous video-EEG captured another generalized tonic clonic seizure that lasted for 3 min while receiving nursing care, which was followed by postictal generalized EEG suppression (PGES) that lasted for approximately 2 min (Fig. 1)

  • A majority of Sudden unexpected death in epilepsy (SUDEP) occurs during sleep, in bed and in the prone position, with most cases being unwitnessed [7]

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Summary

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.

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