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Idiopathic recurring stupor

Institute of Neurology, University of Bologna, Bologna, Italy (Drs. Tinuper, Montagna, Plazzi, Avoni, Cerullo, Cortelli, Sforza, and Lugaresi)
Pharma Division, Preclinical Research, F. Hoffmann-La Roche Ltd, Basel, Switzerland (Drs. Bonetti and Schoch)
Department of Neurology, Johns Hopkins University, Baltimore, MD (Dr. Rothstein)
Fidia-Georgetown Institute for the Neurosciences, Georgetown University, Washington, DC (Dr. Guidotti).

Idiopathic recurring stupor (IRS) is a disease of unknown pathogenesis presenting with recurrent stuporous states. We describe three IRS patients in whom there were no metabolic, toxic, or structural brain dysfunctions. Ictal EEGs were characterized by fast (14- to 16-Hz), unreactive background activity. Flumazenil, a benzodiazepine receptor antagonist, promptly resolved the clinical and EEG picture. In all patients, ictal plasma determination showed a marked increase in benzodiazepine-like activity identified as endozepine-4. IRS may be due to an unexplained excess of endozepine-4.

Address correspondence and reprint requests to Dr. Paolo Tinuper, Clinica Neurologica, via Ugo Foscolo 7,40123 Bologna, Italy.

Presented in part at the 117th annual meeting of the American Neurological Association, Toronto, ON, Canada, October 1992.

Received June 22, 1993. Accepted for publication in final form September 16, 1993.

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