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NEUROLOGY 1993;43:1078
© 1993 American Academy of Neurology

Epileptic negative myoclonus

R. Guerrini, MD, C. Dravet, MD, P. Genton, MD, M. Bureau, MD, J. Roger, MD, G. Rubboli, MD and C. A. Tassinari, MD

Institute of Developmental Neuropsychiatry (Dr. Guerrini), University of Pisa and Institute for Scientific Research in Child Neuropsychiatry Stella Maris, Pisa, Italy; the Centre Saint Paul (Drs. Dravet, Genton, Bureau, and Roger), Marseille, France; and the Institute of Neurology (Drs. Rubboli and Tassinari), University of Bologna, Bellaria Hospital, Bologna, Italy.

Five patients with partial epilepsy of diverse etiology insidiously developed action-activated jerks. The disorder was limited to one arm in two patients and to the legs in another, and was multifocal in the remaining two. Each jerk was related to an EMG silent period lasting 100 to 400 msec, causing a lapse followed by resumption of posture. Simultaneous EEG-EMG recording showed each postural lapse to be time-locked with a sharp or spike and slow-wave transient over the contralateral sensorimotor cortex, where almost continuous paroxysmal activity occurred. The three patients who were able to cooperate during neurologic evaluation also exhibited motor neglect in the most affected body segment and decreased awareness of the disorder. In three patients, the phenomenon was medically resistant, and in two of them it was continuous and could be defined as epilepsia partialis continua. In the other two, medical treatment induced remission of EEG, motor, and neuropsychological abnormalities. This disabling movement disorder can be classified as "epileptic negative myoclonus" and may result from focal-discharge-related transient disruption of cortical function in the sensorimotor cortex.

Address correspondence and reprint requests to Dr. Renzo Guerrini, Institute of Child Neuropsychiatry, University of Pisa, Via dei Giacinti 2, 56018 Calambrone, Pisa, Italy.

Presented in part at the annual meeting of the American Epilepsy Society, Philadelphia, PA, December 8-11, 1991.

Received July 29, 1992. Accepted for publication in final form October 12, 1992.




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