ArticleAnnals of neurology2021
Postictal Death Is Associated with Tonic Phase Apnea in a Mouse Model of Sudden Unexpected Death in Epilepsy.
Article in Annals of neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 44 papers.
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Who cites it
44 citing papers in PubMed, 55 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
- 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
- Severe, Non-apneic Respiratory Dysfunction and Hypoxia following Generalized Convulsive Seizures.Annals of neurology · 2026Article
- Forgotten muscle: diaphragm and sudden infant death syndrome.World journal of pediatrics : WJP · 2026Article
- Article
- Translational Research Symposium: Current State of SUDEP Research.Epilepsy currents · 2026Article
- Base editing rescues seizures and sudden death in a SCN8A mutation-associated developmental epileptic encephalopathy model.The Journal of clinical investigation · 2026Article
- Respiratory dysfunction and fatal apnea during pilocarpine-induced status epilepticus.Epilepsia · 2026Article
- Behavior Decoding Delineates Seizure Microfeatures and Associated Sudden Death Risks in Mouse Models of Epilepsy.Annals of neurology · 2026Article
- Caught on Tape: How Video Clarified Focal to Bilateral Tonic-Clonic Seizures?Epilepsy currents · 2025Article
- Enhancing sudden unexpected death in epilepsy (SUDEP) research through development of common data elements.Epilepsia open · 2025Article
- Spontaneous Seizure Outcomes in Mice Using an Improved Version of the Pilocarpine Model of Temporal Lobe Epilepsy.International journal of molecular sciences · 2025Article
- Artificial intelligence in preclinical epilepsy research: Current state, potential, and challenges.Epilepsia open · 2025Review
- Persistent Na+ current couples spreading depolarization to seizures in Scn8a gain-of-function mice.Brain : a journal of neurology · 2025Article
- Article
- Sudden Unexpected Death in Epilepsy: Central Respiratory Chemoreception.International journal of molecular sciences · 2025Review
- Seizures and premature death in mice with targeted Kv1.1 deficiency in corticolimbic circuits.Brain communications · 2025Article
- Parvalbumin interneuron impairment causes synaptic transmission deficits and seizures in SCN8A developmental and epileptic encephalopathy.JCI insight · 2024Article
- Uncovering Diaphragm Cramp in SIDS and Other Sudden Unexpected Deaths.Diagnostics (Basel, Switzerland) · 2024Article
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Authors and funding
12 authors at 3 institutions in 1 country.
Funding
Abstract
objectiveSudden unexpected death in epilepsy (SUDEP) is an unpredictable and devastating comorbidity of epilepsy that is believed to be due to cardiorespiratory failure immediately after generalized convulsive seizures.
methodsWe performed cardiorespiratory monitoring of seizure-induced death in mice carrying either a p.Arg1872Trp or p.Asn1768Asp mutation in a single Scn8a allele-mutations identified from patients who died from SUDEP-and of seizure-induced death in pentylenetetrazole-treated wild-type mice.
resultsThe primary cause of seizure-induced death for all mice was apnea, as (1) apnea began during a seizure and continued for tens of minutes until terminal asystole, and (2) death was prevented by mechanical ventilation. Fatal seizures always included a tonic phase that was coincident with apnea. This tonic phase apnea was not sufficient to produce death, as it also occurred during many nonfatal seizures; however, all seizures that were fatal had tonic phase apnea. We also made the novel observation that continuous tonic diaphragm contraction occurred during tonic phase apnea, which likely contributes to apnea by preventing exhalation, and this was only fatal when breathing did not resume after the tonic phase ended. Finally, recorded seizures from a patient with developmental epileptic encephalopathy with a previously undocumented SCN8A likely pathogenic variant (p.Leu257Val) revealed similarities to those of the mice, namely, an extended tonic phase that was accompanied by apnea.
interpretationWe conclude that apnea coincident with the tonic phase of a seizure, and subsequent failure to resume breathing, are the determining events that cause seizure-induced death in Scn8a mutant mice. ANN NEUROL 2021;89:1023-1035.
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