ArticleThe Journal of clinical investigation2018
Severe peri-ictal respiratory dysfunction is common in Dravet syndrome.
Article in The Journal of clinical investigation, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 104 papers.
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Who cites it
104 citing papers in PubMed, 145 citations in OpenAlex.
- Rapid hippocampal kindling of mice as a model of sudden unexpected death in temporal lobe epilepsy.Experimental neurology · 2026Article
- Seizure-induced apnea causes sudden death independent of cortical spike wave discharge in mouse models of Tuberous Sclerosis Complex.Neurobiology of disease · 2026Article
- Prolonged postictal hypercapnia is associated with delayed recovery of consciousness after generalized convulsive seizure.Seizure · 2026Article
- Sex-specific differences in mortality and neurocardiac interactions in the Kv1.1 knockout mouse model of sudden unexpected death in epilepsy (SUDEP).The Journal of physiology · 2026Article
- Effect of epileptogenesis on hypercapnic cardioventilatory response in kainic acid rats.Epilepsia · 2026Article
- Brainstem Disinhibition Destabilizes Breathing in Dravet Syndrome, Leading to Sudden Unexpected Death in Epilepsy (SUDEP).Epilepsy currents · 2026Article
- Severe, Non-apneic Respiratory Dysfunction and Hypoxia following Generalized Convulsive Seizures.Annals of neurology · 2026Article
- Association of Interictal Respiratory Variability and Severity of Postictal Hypoxemia After Generalized Convulsive Seizures.Neurology · 2026Article
- Translational Research Symposium: Current State of SUDEP Research.Epilepsy currents · 2026Article
- AES-NINDS Epilepsy Benchmarks Area IV: Comorbidities of Epilepsy - Where Are We and Where Will We Go?Epilepsy currents · 2026Article
- Multiomic Analyses Reveal Brainstem Metabolic Changes in a Mouse Model of Dravet Syndrome.Cells · 2025Article
- Altered PI3K/mTOR Signaling within the Forebrain Leads to Respiratory Deficits in a Mouse Model of Epilepsy.eNeuro · 2025Article
- Development and characterization of an autoresuscitation test for preclinical SUDEP models.Epilepsia · 2025Article
- Developmental progression of respiratory dysfunction in a mouse model of Dravet syndrome.JCI insight · 2025Article
- Respiratory modulation of heart rate variability after generalized convulsive seizures.Seizure · 2025Article
- The Crucial Interplay Between the Lungs, Brain, and Heart to Understand Epilepsy-Linked SUDEP: A Literature Review.Brain sciences · 2025Review
- Article
- MK-212 precipitates seizure-induced death in amygdala-kindled mice via a non-5-HTEpilepsy & behavior : E&B · 2025Article
- Seizure-related death exhibits a circadian rhythm independent of seizure timing or sleep in a mouse model of Dravet syndrome.The Journal of physiology · 2025Article
- Sudden unexpected death in epilepsy: respiratory vs. cardiac contributions.Cardiovascular research · 2025Review
44 more citing papers are in PubMed but not listed here.
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Authors and funding
10 authors at 2 institutions in 2 countries.
Funding
Abstract
Dravet syndrome (DS) is a severe childhood-onset epilepsy commonly due to mutations of the sodium channel gene SCN1A. Patients with DS have a high risk of sudden unexplained death in epilepsy (SUDEP), widely believed to be due to cardiac mechanisms. Here we show that patients with DS commonly have peri-ictal respiratory dysfunction. One patient had severe and prolonged postictal hypoventilation during video EEG monitoring and died later of SUDEP. Mice with an Scn1aR1407X/+ loss-of-function mutation were monitored and died after spontaneous and heat-induced seizures due to central apnea followed by progressive bradycardia. Death could be prevented with mechanical ventilation after seizures were induced by hyperthermia or maximal electroshock. Muscarinic receptor antagonists did not prevent bradycardia or death when given at doses selective for peripheral parasympathetic blockade, whereas apnea, bradycardia, and death were prevented by the same drugs given at doses high enough to cross the blood-brain barrier. When given via intracerebroventricular infusion at a very low dose, a muscarinic receptor antagonist prevented apnea, bradycardia, and death. We conclude that SUDEP in patients with DS can result from primary central apnea, which can cause bradycardia, presumably via a direct effect of hypoxemia on cardiac muscle.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.