Case Report Ictal yawning in a patient with drug-resistant focal epilepsy: Video/EEG documentation and review of literature reports Nicola Specchio a, ⁎, Antonio Carotenuto a, b , Marina Trivisano c , Simona Cappelletti d , Federico Vigevano a , Lucia Fusco a a Division of Neurology, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy b Medical School, University of Naples, Naples, Italy c Clinic for Nervous System Diseases, University of Foggia, Foggia, Italy d Unit of Clinical Psychology, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy abstract article info Article history: Received 17 June 2011 Revised 16 July 2011 Accepted 15 August 2011 Available online 14 September 2011 Keywords: Epilepsy Ictal yawning Malformation of cortical development Eyelid myoclonia Video-electroencephalography Drug-resistant epilepsy Yawning is an involuntary sequence of mouth opening, deep inspiration, brief apnea, and slow expiration. Few cases of yawning as a clinical sign of epileptic seizures, ictally or postictally, have been reported. We report the video/EEG documentation of yawning as an ictal sign in a 31-year-old patient affected by drug-resistant focal epilepsy symptomatic of bilateral perisylvian polymicrogyria. Since the age of 10 she has had seizures charac- terized by yawning, staring, and eye blinking. Bilateral rhythmic frontotemporal spikes and waves character- ized her EEG. We reviewed all reported cases and compared clinical and EEG features. We believe that yawning as part of an epileptic seizure might be considered a rare automatic behavior, like other automatisms frequently reported in epileptic seizures. Automatisms are more frequently described in patients with tempo- ral lobe epilepsy, and involvement of the temporal lobe in most of the published cases may have led to this ex- planation. It is possible that yawning within epileptic seizures could be considered activation of distinct symptomatogenic cortex rather than a release phenomenon. This rare ictal manifestation should be recog- nized as epileptic to avoid misdiagnosis and treatment failure. © 2011 Elsevier Inc. All rights reserved. 1. Introduction Yawning is a physiological event that occurs in both animals and humans. In mammals it is an involuntary sequence of mouth opening, deep inspiration, brief apnea, and slow expiration [1]. The physiolog- ical mechanism underlying yawning remains unknown, but various brain structures are related to this event. Several physiological events can lead to yawning such as drowsi- ness, sleepiness, and emotional states [2]; also, yawning may be in- duced by pathological conditions such as stroke, encephalitis, Parkinson's disease, migraine, frontal lobe and brainstem tumors, and amyotrophic lateral sclerosis [3–6]. Numerous neurotransmitters, neuropeptides, and hormones are implicated in the control of yawn- ing. Neuroendocrine substances such as dopamine, acetylcholine, se- rotonin, nitric oxide, noradrenaline, oxytocin, and steroid hormones have a facilitatory effect; opioid peptides and GABA have an inhibito- ry effect [7]. There is evidence of contagious yawning, which occurs in 40–60% of adult humans and is probably related to mirror-neuron system activity [8]. Despite this evidence, the role and physiology of yawning are not clear. Few cases of yawning as a clinical sign of epileptic seizures have been reported; the majority of patients described had symptomatic temporal lobe epilepsy, and yawning was reported as an ictal or post- ictal sign [9–15]. Our aim was to contribute to the understanding of the clinical se- miology and neurophysiological features of patients affected by symptomatic focal epilepsy with seizures characterized by yawning in association with staring and eye blinking. Moreover, we reviewed all reported cases and compared their clinical and EEG features. 2. Case report This 31-year-old woman was born at term after an uneventful pregnancy. She was the first child of healthy nonconsanguineous par- ents. Perinatal moderate hypoxia and bilateral hip dislocation were reported. No family history of epilepsy or other neurological disorders was reported. Moderate psychomotor retardation was evident since the first year of life. At the age of 16 months she started to have focal seizures charac- terized by psychomotor arrest and left- or right-sided head and eye deviation, sometimes followed by tonic posturing of the upper limbs. Several EEGs showed bilateral frontal and frontal and temporal epilep- tiform abnormalities. Seizure frequency was almost daily soon after onset. The patient was treated with carbamazepine (30 mg/kg/day), Epilepsy & Behavior 22 (2011) 602–605 ⁎ Corresponding author at: Division of Neurology, Bambino Gesù Children's Hospital, IRCCS, Piazza Sant’ Onofrio 4, 00165, Rome, Italy. Fax: + 39 0668592463. E-mail address: nicola.specchio@opbg.net (N. Specchio). 1525-5050/$ – see front matter © 2011 Elsevier Inc. All rights reserved. doi:10.1016/j.yebeh.2011.08.013 Contents lists available at SciVerse ScienceDirect Epilepsy & Behavior journal homepage: www.elsevier.com/locate/yebeh