Case Report The sign of the cross as a learned ictal automatism? Richard Wennberg a, * , Mary-Pat McAndrews a , Dominik Zumsteg a,b , Jose Luis Perez Velazquez c a Krembil Neuroscience Centre, Toronto Western Hospital, University of Toronto, Toronto, Ont., Canada b Department of Neurology, University Hospital Zurich, Zurich, Switzerland c Brain and Behaviour Centre, Neuroscience and Mental Health Programme, Hospital for Sick Children, University of Toronto, Toronto, ON, Canada article info Article history: Received 8 April 2009 Accepted 11 April 2009 Available online 17 May 2009 Keywords: Temporal lobe epilepsy Seizures Automatism Sign of the cross Conditioning Learning Memory abstract Described here is a case of a patient who made the sign of the cross during right mesial temporal seizures, documented by intracranial depth electrode and simultaneous scalp video-EEG. The patient was ulti- mately found to have predominantly left temporal lobe epilepsy, and she was rendered seizure free for many years following a left anterior temporal lobe resection. Most interestingly, however, was a sugges- tion that in her case, making the sign of the cross may have represented a learned ictal behavioral phe- nomenon: the patient had been forced, over a period of many years, to make this gesture as an atonement in the postictal period. The movement ultimately came to be performed unconsciously, during the ictus, associated with a lateralized seizure discharge in the right temporal lobe. In contrast to seizure-induced experiential phenomena and typical motor automatisms, where the behavioral manifestations have no recognized association with learning, we wondered whether the pathophysiological mechanisms of chronic focal epilepsy had subserved in this case a psychological learning process, whereby right tempo- ral seizures were ultimately able to recruit and activate an adjacent neural memory circuit. Ó 2009 Elsevier Inc. All rights reserved. 1. Introduction The recent report in this Journal of ictal signum crucis in four patients with right temporal lobe epilepsy [1] brought to our minds the case of a patient we saw a number of years ago who also made the sign of the cross during some right temporal seizures. Our patient was ultimately found to have predominantly left tem- poral lobe epilepsy, and she was rendered seizure free for many years following a left anterior temporal lobe resection. Most inter- estingly, however, was a suggestion that in her case, making the sign of the cross may have represented a type of conditioning (de- fined loosely) or a learned ictal phenomenon. 2. Case The patient, 46 years old at the time of her presurgical investi- gations, was a right-handed woman with complex partial seizures since early childhood. She had a history of febrile convulsion at the age of 2 years preceding the onset of her chronic, medically refrac- tory epilepsy. Her typical complex partial seizures were preceded by a brief epigastric aura and occasionally followed by a postictal aphasia. They were occurring approximately once every 2 weeks at the time of her investigations, in addition to less frequent sec- ondarily generalized seizures. There was no family history of epilepsy. The patient had one sis- ter and two brothers, with whom she was not in contact. She left home at age 21 and had been estranged from her parents long be- fore their deaths more than 15 years later. Routine EEGs showed bilateral independent anterior temporal interictal epileptiform and nonepileptiform abnormalities, right greater than left. Brain MRI revealed bilateral mesial temporal atrophy and sclerosis, much worse on the left side. In-hospital scalp video/EEG recording documented five complex partial sei- zures, two with secondary generalization, each associated initially with staring, unresponsiveness, lip smacking, chewing and swal- lowing automatisms, and fumbling automatisms of the hands and upper extremities. The clinical manifestations of each seizure preceded the first ictal epileptiform changes on the scalp EEG by 25–30 seconds. Once apparent, the ictal EEG changes invariably showed bitemporal involvement with a questionable right tempo- ral lead. Formal neuropsychological testing revealed average intelli- gence and evidence of bilateral temporal dysfunction, with greater impairment of nonverbal than verbal learning and memory reten- tion. An intracarotid sodium amytal test showed the patient’s left hemisphere to be dominant for language. Memory was poorer fol- lowing the right injection, but the results indicated no significant risk for global amnesia should either temporal lobe be resected. Given the bilateral electrographic, imaging, and neuropsycho- logical findings, and the lateralization mismatch between the scalp 1525-5050/$ - see front matter Ó 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.yebeh.2009.04.017 * Corresponding author. Address: Toronto Western Hospital, 5W444, 399 Bath- urst Street, Toronto, Ont., Canada M5T 2S8. Fax: +1 416 603 5768. E-mail addresses: r.wennberg@utoronto.ca, Richard.Wennberg@uhn.on.ca (R. Wennberg). Epilepsy & Behavior 15 (2009) 394–398 Contents lists available at ScienceDirect Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh