revue neurologique 169 (2013) 903–910 Available online at ScienceDirect www.sciencedirect.com Brief communication Peri-ictal water drinking and other ictal vegetative symptoms: Localizing and lateralizing the epileptogenic zone in temporal lobe epilepsy? Two case reports and review of the literature La sensation de soif et autres symptômes neurovégétatifs durant la crise : quelle valeur localisatrice et latéralisatrice dans les épilepsies du lobe temporal ? À propos de deux cas et revue de la littérature L. Errguig a,b,*, F. Lahjouji a, H. Belaidi a, M. Jiddane b,c, A. Elkhamlichi b,d, T. Dakka b, R. Ouazzani a,b a Department of Clinical Neurophysiology, Hôpital des Spécialités Centre Hospitalier Ibn Sina, Rabat Institut BP 6220, Rabat, Maroc b Laboratory of physiology, faculty of medicine and pharmacy of Rabat, university Med V Souissi, Rue Lamfadel Cherkaoui, Rabat Institut BP 6527, Rabat, Maroc c Department of Neuroradiology, Hôpital des Spécialités Centre Hospitalier Ibn Sina, Rabat Institut BP 6220, Rabat, Maroc d Department of Neurosurgery, Hôpital des Spécialités Centre Hospitalier Ibn Sina, Rabat Institut BP 6220, Rabat, Maroc info article abstract Article history: Peri-ictal behavior disorders can be helpful in localizing and lateralizing seizure onset in Received 8 November 2012 partial epilepsies, especially those originating in the temporal lobe. In this paper, we present Received in revised form the case of two right-handed women aged 36 and 42 years who presented with partial 14 May 2013 seizures of mesial temporal type. Both of the patients had drug resistant epilepsy and Accepted 11 June 2013 undergone presurgical evaluation tests including brain magnetic resonance imaging, video- Available online 15 October 2013 EEG monitoring and neuropsychological testing. The two patients had hippocampal scle- Keywords: temporal lobe discharges documented during video-EEG recordings. Anterior temporal rosis in the right temporal lobe and exhibited PIWD behavior concomitant with right Peri-ictal water drinking lobectomy was performed in one case with an excellent outcome after surgery. The patient Temporal lobe epilepsy was free of seizures at 3 years follow-up. We reviewed other publications of peri-ictal Ictal vegetative symptoms autonomic symptoms considered to have a lateralizing significance, such as peri-ictal Localizing and lateralizing ictal vomiting, urinary urge, ictal pilo-erection. Clinicians should search for these symptoms, symptoms even if not spontaneously reported by the patient, because they are often under-estimated, Surgical treatment both by the patients themselves and by physicians. Additionally, patients with lateralizing * Corresponding author. Department of clinical neurophysiology, Hôpital des Spécialités Centre Hospitalier Ibn Sina, Rabat Institut BP 6220, Rabat, Maroc. E-mail address : lerrguig@hotmail.com (L. Errguig). 0035-3787/$ – see front matter # 2013 Published by Elsevier Masson SAS. http://dx.doi.org/10.1016/j.neurol.2013.06.005 904 revue neurologique 169 (2013) 903–910 auras during seizures have a significantly better outcome after epilepsy surgery than those Mots-clés : without lateralizing features. Sensation de soif péri-critique # 2013 Published by Elsevier Masson SAS. Épilepsie du lobe temporal Symptômes neurovégétatifs péricritiques Symptômes critiques localisateurs et latéralisateurs Traitement chirurgical r é s u m é Les symptômes per critiques peuvent aider dans la localisation de la zone épileptogène en matière d’épilepsies, particulièrement les épilepsies du lobe temporal (ELT). À travers ce travail, les auteurs rapportent l’observation de deux patientes ayant une épilepsie mésiotemporale pharmaco-résistante en rapport avec une sclérose hippocampique droite. Ces patientes, droitières, âgées de 36 et 42 ans, bénéficièrent d’une évaluation préchirurgicale comportant une imagerie cérébrale par résonance magnétique, un enregistrement EEGvidéo et un bilan neuropsychologique. La sensation de soif fut exprimée par les 2 patientes et coı̈ncida avec une décharge épileptique au niveau temporal antérieur droit lors de l’enregistrement EEG-vidéo. Une lobectomie temporale antérieure fut réalisée chez une patiente avec une évolution post-chirurgicale favorable après 3 ans (classe Ia d’Engel). Par ailleurs, les auteurs font une revue de la littérature des autres symptômes neurovégétatifs per critiques décrits comme ayant une valeur localisatrice et même latéralisatrice dans les ELT tels que : les vomissements, l’urgenturie, la pilo-érection. Ces symptômes doivent être recherchés lors de l’interrogatoire car, en plus, ils représentent un facteur de bon pronostic postchirurgical lorsqu’ils sont présents. # 2013 Publié par Elsevier Masson SAS. 1. Introduction The clinical features of focal epilepsy often help to localize the seizure onset zone. Autonomic symptoms, such as ictal vomiting or peri-ictal urinary urge are frequent during epileptic seizures and have a lateralizing significance. Periictal water drinking (PIWD) has been reported in a few papers as a localizing and lateralizing symptom in temporal lobe epilepsy (TLE). TLE with hippocampal sclerosis (HS) is often drug resistant and requires surgical treatment and, ideally, complete resection of the epileptic zone. Diagnostic elements that help in the presurgical estimation of the epileptic zone are seizure semiology, electrophysiological recordings, neuroimaging, functional testing and neuropsychological assessment. Ictal semiology improves lateralization and localization of the epileptic zone and adds additional informations to the ictal EEG. Additionally, patients with lateralizing auras during seizures have a better outcome after surgery than those without lateralizing features. In this paper, the authors report the case of two patients who had TLE secondary to hippocampal sclerosis in the right side, with PIWD behaviour during their seizures. They also reviewed the literature using Pubmed for the expressions ‘‘peri-ictal drinking’’, ‘‘peri-ictal water drinking’’, ‘‘epilepsy AND drinking’’, and ‘‘thirst AND seizure’’, and evaluated the cases obtained for localization and lateralization of the epileptic focus. Other vegetative symptoms, reported in the literature in terms of temporal lobe epilepsy, are also discussed with evaluation of their localizing and lateralizing value. 1.1. Case 1 A right-handed woman, 42 years old, manifested her first seizures at age 30. Ictal clinical symptoms included epigastric aura, distress, tachycardia, staring, unresponsiveness with automatisms of hands, oro-alimentary automatisms, dystonic posturing of the left hand, ictal water drinking, and finally post-ictal confusion. Secondary generalisations were rare. Because of drug resistance pharmacotherapy, she underwent presurgical evaluation with a non-invasive protocol including interictal EEG, video-EEG monitoring, brain MRI and neuropsychological testing. Interictal EEG (Fig. 1) revealed epileptic discharges in the right anterior temporal lobe. Ictal EEG (Figs. 2 and 3) localized the onset epileptic zone in the right anterior temporal lobe and the patient asked for water during the seizure. Brain MRI (Fig. 4) revealed right hippocampal sclerosis. Neuropsychological testing concluded to visual memory deficiency with preserved verbal memory. The patient benefited from anterior temporal lobectomy (ATL), as all data (clinical features, ictal EEG findings and MRI) were concordant. Since her ATL, she is seizure-free with a follow-up of 3 years. 1.2. Case 2 A right-handed woman aged 36 years old, started her epilepsy history at the age of 8. Her seizure semiology included epigastric aura, tachycardia, staring, hypersalivation, motor and verbal automatisms with feeling of thirst. Secondary generalisations were also rare in this patient. EEG, even interictal and ictal (Fig. 5), showed right temporal abnormalities in the anterior temporal regions. Brain MRI (Fig. 6) revealed hippocampal sclerosis in the right side. Neuropsychological testing resulted in visuospatial memory disorder with respect to verbal memory. The patient epilepsy was drug resistant, she is awaiting surgical treatment. 2. Discussion Our two patients had focal epilepsy originating in the right temporal region, and both of them presented the PIWD revue neurologique 169 (2013) 903–910 905 Fig. 1 – A to E: Interictal EEGs showing an overload of slow theta waves occupying consistently the right anterior temporal region and these slow waves are more frequent as we approach the crisis (E). Fig. 2 – Ictal EEG showing aura (arrow) and the onset (rapid rythms) in the right anterior temporal lobe coinciding with sensation of thirst in case 1. 906 revue neurologique 169 (2013) 903–910 Fig. 3 – Ictal EEG showing a flattening of background activity (arrow) in the right anterior temporal region. behaviour during their seizure in the video-EEG recordings concomitant with discharges in the right anterior temporal region. A total of 35 PIWD cases with TLE were found in the literature (Table 1). The epileptogenic zone was located in the right or non-dominant temporal lobe in 22 cases (63%). In a series of 65 patients with focal epilepsy, Trinka et al. (2003) identified 10 patients with PIWD (15%). A previous report by Rémillard et al., 1981 discussed 20 patients with complex partial seizures associated with ictal desire for water or actual water consumption. All of their patients had EEG evidence of revue neurologique 169 (2013) 903–910 907 Fig. 4 – Brain MRI, with FLAIR sequence in case 1, showing a hypersignal in the right hippocampus. non-dominant temporal involvement; six of them were documented with ictal EEG. In another series of 55 TLE patients (Szucs et al., 2007), PIWD was observed in one or more recorded seizures of eight patients (14.5%). Five patients had left temporal and three had right temporal lobe resections. At 2–10 years of follow-up, all operated patients were reported as Fig. 6 – Brain MRI, with FLAIR sequence in case 2, showing a hypersignal in the right hippocampus. Fig. 5 – A: Interictal EEG showing, few minutes before ictal recording, slow waves in the right anterior temporal lobe. B: Ictal EEG in case 2, with sensation of thirst (dotted arrow) and the aura (arrow) coinciding with rapid rhythms in the right anterior temporal lobe. C: Ictal EEG with spike and waves exhibited in the anterior temporal lobes with clear predominance in the right side. D: Post ictal EEG with delta slow waves in the right anterior temporal lobe. 908 Table 1 – PIWD in various publications in the literature suggesting a lateralization of the epileptogenic zone in the non-dominant temporal lobe. Suspected side of epileptogenic region Cerebral lesion Imaging method Interictal EEG Seizure pattern Video-EEG monitoring Crapanzano et al. (1993) Cascino and Sutula, 1989 Rémillard et al. (1981) Right Right hippocampal atrophy MRI Right temporal Not recorded Right Right hippocampal sclerosis MRI Right temporal sharp waves and slowing in drowsiness Right anterior temporal Right temporal Yes Left amygdala, Hippocampus in 4/10 cases Right temporal in 6/10 cases Widening of the anterior horn of left temporal lateral ventricle (2) PEG Bitemporal (2), right temporal (6), left temporal (2), Yes (2), not recorded (8) Right Postmeningitis (1) Right hemispheric atrophy (2) others (5) Right frontotemporal tumor Left temporal (deep electrodes) (2), right hemispheric slow and sharp activity trains (2), not recorded (6) Not recorded Not recorded 7 Right Right temporal epileptic abnormality Yes Right temporal Right temporal resection 3 Right Yes Right temporal Right temporal resection Bitemporal independent Spikes (3), right temporal sharp waves (2), left anterior temporal Spikes (3) Right temporal rhythmic theta or rhythmic activity (3), left temporal rhythmic crescendo pattern (5) Despland et al. (1985) Trinka et al. (2003) Janszky et al. (2007) Szucs et al. (2007) 3 right and 5 left Left hippocampal sclerosis (3), right hippocampal sclerosis (1), bilateral, R > L hippocampal sclerosis (1), normal MRI (1), right temporal oligoastrocytoma (1), left temporal cavernoma (1) MRI Yes Operation Right temporal anterior lobectomy None (10) Operation of the tumor Left temporal resection (5), right temporal resection (3) revue neurologique 169 (2013) 903–910 Author revue neurologique 169 (2013) 903–910 Table 2 – Other localizing and lateralizing peri-ictal vegetative symptoms. Lateralizing ictal features (Symptoms and authors references) Presumed brain region Ictal spitting Caboclo et al. (2006) Renier, 2004 Kellinghaus et al. (2003) Ictal vomiting Sekimoto et al. (2007) Schindler and Wieser, 2006 Schauble et al., 2002 Devinsky et al. (1995) Postictal nose wiping Hirsch et al., 1998 Ictal pallor Fogarasi et al., 2005a Ictal smile Fogarasi et al., 2005b Ictal urinary urge Gurgenashvili et al. (2011) Loddenkemper et al. (2003) Baumgartner et al. (2000) Unilateral ictal piloerection Haykal and Abou-Khalil, 2012 Dominant TL/ Non-dominant TL Right or left TL Ipsilateral TL Left TL Right TL Non-dominant TL Ipsilateral TL having an Engel I or 2 outcomes. In a series of 97 TLE patients, Musilová et al. (2010) reviewed 380 seizures. All their patients were classified as Engel I at least 2 years after surgery. PIWD was noted in 25% of patients and occurred significantly more often in patients with TLE of the non-language-dominant hemisphere. Physiologically, normal drinking behavior is controlled by tissue osmolality and vascular volume. Information from peripheral receptors is processed in the hypothalamus and then is conveyed to various brain structures that initiate a search for water and drinking. The hippocampus and the amygdala are linked with the hypothalamus by a variety of multisynaptic reciprocal pathways. Thus, epileptic activity generated in the mesial temporal lobe may propagate into the hypothalamus and inappropriately activate water seeking. The lateralizing value of PIWD can be explained by a possible hemispheric specific representation of fluid control, thirst and water seeking behaviour. These assumptions need to be confirmed by invasive EEG recordings or ictal SPECT studies. The lateralizing value of PIWD in our cases, like in the other series, relies on postoperative seizure freedom. According to Szucs et al., 2007, the difference of these results could be explained by the fast contralateral propagation in some cases, because in cases of quick contralateral spread, the laterality of any semiological element may be unclear. In these cases, it is uncertain which of the two involved temporal lobes generated the PIWD. Another explanation for the contradictory data might be that the numbers of patients in these studies were too small. A well-powered study involving more patients can resolve this problem. The authors reviewed other peri-ictal (ictal or postictal) vegetative symptoms (PIVS) described in both adult and childhood epilepsy (Table 2), and are of clinical and scientific importance. In adults, these symptoms are commonly observed in temporal lobe epilepsy (TLE) and can include cardiovascular 909 (blood pressure changes, heart arrhythmia— tachycardia/ bradycardia), respiratory (hyperventilation/bradypnea or apnea), gastrointestinal (hypersalivation, nausea, retching, and vomiting), cutaneous (pilo-erection, or ‘‘goose bumps,’’ and sweating), pupillary (mydriasis, miosis), genital, sexual, and urinary or bowel (urinary urge and flatulence) manifestations. Some of them are rare, such as coughing, water drinking, vomiting, spitting, urinary urge, flatulence, yawning, and piloerection. The literature data concerning the lateralization value of these PIVS are mostly controversial. Most articles analyzing their lateralizing value have demonstrated that PIVS are mostly linked to the temporal lobe of the non-speech-dominant hemisphere. Other studies and case reports have excluded a lateralizing value for these symptoms (Fauser et al., 2004; Caboclo et al., 2006; Scücs et al., 2007; Janszky et al., 2007). Looking for these symptoms is of particular interest in our department since we started epilepsy surgery in patients with drug resistant mesial temporal lobe epilepsy in 2005. We rely on a basic non-invasive presurgical evaluation protocol including brain magnetic resonance imaging, video-EEG monitoring, long-term interictal EEG and neuropsychological assessment (Lahjouji et al., 2007). Surgery was indicated if a good electro-clinico-radiological correlation was obtained. About 70 TLE patients benefited from temporal lobectomy with an Engel class I outcome in 90% at 3 years of follow-up. 3. Conclusion Lateralizing signs in seizure semiology are an important face in the complete presurgical evaluation of epilepsy patients. Knowledge and recognition of semiological lateralizing signs during seizures are important in the presurgical evaluation of epilepsy surgery candidates and add further information to video/EEG monitoring, neuro-imaging, functional mapping, and neuropsychological evaluation. In our country, presurgical evaluation relies exclusively on clinical data and non-invasive tests. Thus, symptoms with localizing value are very important to search. Peri-ictal water drinking behaviour is rarely reported in the literature. It has a good localizing and lateralizing significance in the non-dominant temporal lobe. Nevertheless, this sign, as the others, cannot lateralize the epilepsy focus with 100% certainty. 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