{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,20]],"date-time":"2026-05-20T17:21:15Z","timestamp":1779297675615,"version":"3.51.4"},"reference-count":15,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2021,11,11]],"date-time":"2021-11-11T00:00:00Z","timestamp":1636588800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Euro J of Neurology"],"published-print":{"date-parts":[[2022,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background and purpose<\/jats:title><jats:p>The core manifestations of leucine\u2010rich glioma\u2010inactivated 1 (LGI1) autoantibody\u2010mediated encephalitis are limbic encephalitis and faciobrachial dystonic seizures. Agrypnia excitata (AE) is a rare syndrome characterized by sleep\u2013wake cycle disruption, autonomic hyperactivation and episodes of oneiric stupor. Only a few diseases are known to present with AE. An autoimmune etiology must be considered when accompanied by neuromyotonia. A case of anti\u2010LGI1 encephalitis presenting with AE is reported.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Detailed clinical, video\u2010polysomnographic, laboratory, radiological and long\u2010term follow\u2010up assessments were performed.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>A previously healthy 58\u2010year\u2010old man was referred for a rapidly progressive change in mental status, characterized by persistent drowsiness and confusion, accompanied by frequent episodes of unconscious gestures ranging from simple stereotyped movements to more complex actions mimicking various daily activities. Other symptoms included tachycardia, hyperhidrosis, mild hyponatremia, rare faciobrachial dystonic seizures, and a single generalized tonic\u2013clonic seizure, but no neuromyotonia. Prolonged video\u2010polysomnography excluded epileptic activity and showed continuous monomorphic slowing of background activity not consistent with a regular wakefulness or sleep state. A brain magnetic resonance imaging scan was unremarkable. Brain fluorodeoxyglucose positron emission tomography revealed hypermetabolism of the hippocampi, amygdala and basal ganglia. Anti\u2010LGI1 antibodies were detected in the cerebrospinal fluid. The sleep disorder resolved progressively after starting immunotherapy.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Agrypnia excitata can be a dominant, treatable manifestation of anti\u2010LGI1 encephalitis. Oneiric stupor episodes are a useful clinical feature for establishing diagnostic suspicion and could provide a window to understanding the mechanisms behind some movement disorders in autoimmune encephalitis.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ene.15152","type":"journal-article","created":{"date-parts":[[2021,10,22]],"date-time":"2021-10-22T23:50:19Z","timestamp":1634946619000},"page":"890-894","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["<i>Agrypnia excitata<\/i> as the main feature in anti\u2010leucine\u2010rich glioma\u2010inactivated 1 encephalitis: a detailed clinical and polysomnographic semiological analysis"],"prefix":"10.1111","volume":"29","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1477-546X","authenticated-orcid":false,"given":"Silvio","family":"Piffer","sequence":"first","affiliation":[{"name":"Department of Emergency Neurology Unit Santa Chiara Hospital Trento 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