A 56-year-old man with refractory bitemporal epilepsy was monitored in the Epilepsy Monitoring Unit (EMU). In a video-EEG captured seizure, brief oroalimentary automatisms were followed by increased inspiratory effort, accompanied by prominent, visible tracheal movements and audible inspiratory stridor. The patient's oxygen saturation rapidly declined to 62%; persistent severe hypoxemia ended with spontaneous effective respiration commencing at seizure end. Subsequent seizures necessitated intensive care unit admission for respiratory distress, and ventilator support. This case suggests that ictal laryngospasm, a rare seizure manifestation, may represent another potential mechanism of sudden unexpected death in epilepsy (SUDEP). [Published with video sequence on www.epilepticdisorders.com].
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From the NINDS Center for SUDEP Research (L.V., M.R.S.R., R.K.S., D.F., M.N., C.S., B.K.G., B.Z., A.Z., S.S., J.O., R.M.H., B.D., L.B., O.D., G.B.R., P.R., S.D.L.); Epilepsy Center (L.V., N.L., J.P.H., N.J.H., N.S., X.Z., V.R.-M., S.D.L.) and Division of Pulmonary, Critical Care and Sleep Medicine (K.S.), University Hospitals Cleveland Medical Center, OH; University of Iowa School of Medicine (R.K.S., B.K.G., G.B.R.), Iowa City; NYU Langone School of Medicine (D.F., O.D.), New York; Sidney Kimmel Medical College (M.N.), Thomas Jefferson University, Philadelphia, PA; Institute of Neurology (C.S., B.D.), University College London, UK; Feinberg School of Medicine (S.S.), Northwestern University, Chicago, IL; Department of Neurobiology and Brain Research Institute (J.O., R.M.H.), University of California, Los Angeles (UCLA); Department of Neurology (L.B.), Columbia University, New York, NY; and Department of Clinical Neuroscience (P.R.), Centre Hospitalier Universitaire Vaudois, Lausanne, Switzerland.
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