SUBMITTED 23 JAN 23 1 REVISION REQ. 8 MAR 23; REVISION RECD. 10 APR 23 2 ACCEPTED 9 MAY 23 3 ONLINE-FIRST: MAY 2023 4 DOI: https://doi.org/10.18295/squmj.5.2023.038 5 6 Peri-ictal Water Drinking in an Omani Patient with Bilateral Mesial 7 Temporal Sclerosis 8 Younis Al-Mufargi,1 *Haifa Alabri,2 Arunodaya R. Gujjar3 9 10 Departments of 1Human Clinical Anatomy and 3Medicine, Sultan Qaboos University, Muscat, 11 Oman; 2Department of Medicine, Sultan Qaboos University Hospital, Sultan Qaboos University, 12 Muscat, Oman 13 *Corresponding Author’s e-mail: jood@squ.edu.om 14 15 Abstract 16 Peri-ictal water drinking (PIWD) is a rare vegetative manifestation of temporal lobe epilepsy 17 without a definite lateralization value. We report a case of PIWD in a 22-year-old Omani man 18 with post-concussion syndrome and epilepsy presented to a tertiary care hospital in Muscat, 19 Oman in 2021 for evaluation of paroxysmal events. His behaviour of PIWD was misinterpreted 20 by his family until characterized in the epilepsy-monitoring unit as a manifestation of epilepsy 21 that was treated medically. To our knowledge, this is the second reported case in our region. 22 Keywords: Peri-ictal water drinking, Ictal Spitting, Epilepsy, Autonomic 23 24 Introduction 25 Vegetative manifestations have been well-described in patients with epilepsy during the seizure 26 in both pediatric and adult age groups.1,2 Peri-ictal vegetative symptoms (PIVS) may occur 27 without any particular relationship to the cause of epilepsy.3 PIVS include cardiovascular, 28 respiratory, gastrointestinal, and urinary signs and symptoms. Peri-ictal water drinking (PIWD) 29 is an infrequently described automatism in an epileptic patient, often among those with temporal 30 lobe epilepsy.3,4 Among patients with focal epileptic seizure, 65 cases only are reported to have 31 mailto:jood@squ.edu.om Peri-ictal water drinking behavior.4 PIWD has been defined as the urge to drink water ictally or 32 up to 2-minute in the post-ictal phase of epileptic seizures.1 Peri-ictal water drinking as a 33 manifestation of epileptic seizure signifies a reliable sign of lateralization to a non-dominant 34 temporal lobe.5 35 36 Herein, we report a case of peri-ictal water drinking (PIWD) in a 22-year-old man with post-37 concussion syndrome with bilateral mesial temporal sclerosis. To our knowledge, this is the 38 second reported case in our region. We found that PIWD has localizing but no lateralizing value. 39 40 Case report 41 A 22-year-old right-handed man had a road traffic accident at the age of 11 years and suffered a 42 post-concussion syndrome with behavioural changes of impulsiveness and seizures. His seizures 43 were described as chest discomfort followed by lip and hand automatism and excessive water 44 drinking during the event. His family thought that he would drink water to relive his chest 45 discomfort. After that, he was routinely offered a bottle of water whenever his seizure started, in 46 the context of this recurrent habitual behaviour. 47 48 To better characterize his seizure semiology and localize the ictal onset, he was admitted for 3-49 day in the Epilepsy Monitoring Unit at a tertiary care hospital in Muscat, Oman for long-term 50 EEG evaluation (Figure 1). During evaluation, his anti-seizure medications (ASM) including 51 Lacosamide 200 mg BID, Sodium Valproate SR 750mg AM/1000 mg PM and Clobazam 15mg 52 AM/20 mg PM were tapered off. His interictal recording showed intermittent slowing in the right 53 temporal chain and bitemporal sharp waves: 80% on the right (maximum at F8>T4), 20% on the 54 left (maximum at F7>T3). He had five seizures of the same semiology. Seizures started with him 55 holding his chest, either due to discomfort or as a reaction to a gastric aura. This was followed by 56 swallowing movements and hand automatism. Out of 5 seizures, 3 seizures had clear water 57 drinking during the seizure or after the seizure terminated. He would ask for or grab a nearby 58 water bottle to drink multiple times. The water bottles were 500 ml, and he drank almost 2/3 the 59 amount of the bottle in each seizure. During the other 2 seizures, the patient was alone, and his 60 behaviour seemed restless, looking for water through his bed sheets but not found. He would 61 always ask for water and no other types of fluids. 62 63 Table 1 summarize the details. (Any gross variations in the pattern of seizure may be 64 summarized here in 1-3 sentences. Rest of the paragraph may be superfluous. The table gives fair 65 details). In seizure 1, he used his hands to perform body language to his attendant indicating the 66 need to drink. He grabbed the bottle again from his attendant to drink the second and third time 67 after giving it back after first request to drink. In the third time, he pointed to his chest as of 68 explaining the reason for drinking. All 3 events of water drinking in seizure one happened 69 ictally. In seizure 2, he drank twice ictally and 24 seconds post ictally. In both events, he grabbed 70 the water bottle off his attendant’s hand to drink. In seizure 3, the seizure woke him up from 71 sleep while the attendant was not aware of the event. There were no water bottles close to him, 72 so he seemed restless and looking around the room frequently. Finally, he got off bed and walked 73 around the bed but that was not visualized by the video. In the 4th seizure, again he was alone and 74 seemed restless when the seizure started as attendant was not around. There was a bottle close to 75 his pillow, but he did not visualize it. He seemed in discomfort and wiping his face and left nose 76 multiple times with his hands. In seizure 5, it started again by chest sensation as he held his 77 hands against his chest then started swallowing movements. His attendant walked into the room 78 and patient pointed by hand gestures that he wants to drink and again pointed to his chest as of 79 explaining his reasoning. He started spitting and he drank all the bottles given to him (~ 400 ml). 80 81 His MRI brain showed evidence of bilateral hippocampal atrophy and bilateral mesial temporal 82 sclerosis (Figure 2). His PET/CT of brain was unremarkable with no focal hyper or 83 hypometabolism. The patient’s consent was obtained for publication purposes. 84 85 Discussion 86 Peri-ictal water drinking was first reported upon a statistical review of aura in epilepsy among 87 1359 cases by Lennox,6 defined as the urge of drinking water ictally or up to 2-minutes in the 88 post-ictal phase of epileptic seizures.1 Seeking water in epileptic patients has been noticed 89 mostly in ictal phase, yet post-ictal water drinking has been reported as well.1 Reported cases of 90 PIWD in literature remain infrequent all over the world, with around 65 cases have been reported 91 to date, including one case in our region.4,7 92 93 Water-seeking behavior has been correlated electro-clinically and neuropathologically with 94 pathologies in the mesial temporal lobe, emphasizing further on the value of localization 95 hypothesis.8 However, a recently published retrospective study in surgically confirmed patients 96 with focal epilepsy in which PIWD occurs more often in frontal lobe epilepsy (FLE) than 97 temporal lobe epilepsy (TLE) indicating that PIWD might not be specific symptom of TLE.9 The 98 epileptic discharges in our patient’s seizure confirmed the localization hypothesis of temporal 99 lobe origin. The epileptic discharges from the temporal lobe structures propagating to the 100 hypothalamus is proposed to be a stimulus for water-seeking behavior and sense of thirst in 101 epilepsy.8,10 102 103 Several cases of peri-ictal water drinking have been published in the literature highlighting the 104 value of lateralization of peri-ictal water drinking in patients with temporal lobe epilepsy. Water 105 seeking behavior has been concluded as a sign representing epileptic focus on the non-dominant 106 temporal lobe.11,12 This lateralization may be explained by asymmetrically represented network 107 of central autonomic system implemented in water-seeking behavior, control of fluid and thirst.11 108 However, other studies have shown no consistent lateralization of epileptiform activity in similar 109 cases.3,5,13 In our patient, we could not conclude definite lateralization as he had seizures with 110 water drinking behavior evident in either of the temporal lobes. Even though, seizures number 1 111 and 2 that originated from the right temporal lobe started earlier at 12 and 15 seconds compared 112 to seizure number 5 that may argue that it lateralizes to the non-dominant hemisphere. However, 113 we have incomplete data of seizure number 3 and 4 that may change the equation, therefore, the 114 significance of lateralization in patient presenting with Peri-ictal water drinking is still uncertain 115 as demonstrated in our case. 116 117 The autonomic manifestations of epilepsy have a risk to progress into status epilepticus as 118 reported in the literature.14 Therefore, the importance of detecting other semiologies of epileptic 119 events as autonomic manifestations such as cardiac and respiratory, is very crucial as it may lead 120 to sudden unexpected death in epilepsy (SUDEP). 121 122 As noticed in our case, the patient had also other epileptic semiologies of temporal origin like 123 post-ictal spitting. Spitting as an ictal phenomenon which has been evoked by direct electrical 124 stimulation to temporal lobe in many trials, confirming the value of localization again with no 125 lateralization value.15 126 127 Focal epileptic syndromes are estimated to affect 60% of patients with epilepsy, in which 15% of 128 those patients’ condition cannot be controlled by anti-seizure medications adequately and as 129 observed assumption, half of them may be considered potential candidates for a surgical 130 intervention of epilepsy. A published case of middle-aged man diagnosed with epilepsy since 131 childhood with features of peri-ictal water drinking on anti-seizure medications of right temporal 132 lobe origin, remained seizure-free in 1-year follow up after right anterior temporal resection.7 133 Moreover, a recent retrospective study discussing occurrence of PIWD in focal epilepsy patients 134 with favorable outcome postoperatively, including frontal lobe epilepsy (FLP) and temporal lobe 135 epilepsy (TLE) signifying the value of preoperative evaluation in patients with focal epileptic 136 syndromes.9 In the present case, the patient had bi-temporal lobe epilepsy secondary to bilateral 137 mesial temporal sclerosis that was better controlled with anti-seizure medications but has 138 significant behavioral issues requiring follow up with psychiatry. A multidisciplinary discussion 139 to consider if he would benefit from surgical management aimed at improvement in both his 140 seizures and behavioral issues is planned. 141 142 Conclusion 143 Peri-ictal water drinking, and ictal spitting are infrequent vegetative symptoms seen in the 144 temporal lobe epilepsy without a definite lateralization value. 145 146 Authors’ Contribution 147 YA did the literature review and wrote entire discussion and analysed of the case. HA wrote the 148 case, analysed the EEG findings in detail with tables and supervised the entire paper. AG 149 reviewed the paper. All authors approved the final version of the manuscript. 150 151 Reference: 152 1. Baumgartner C, Koren J, Britto-Arias M, Schmidt S, Pirker S. Epidemiology and 153 pathophysiology of autonomic seizures: a systematic review. Clin Auton Res. 2019 154 Apr;29(2):137–50. https://doi.org/10.1007/s10286-019-00596-x 155 2. Fogarasi A, Janszky J, Tuxhorn I. Autonomic Symptoms during Childhood Partial Epileptic 156 Seizures. Epilepsia. 2006 Mar;47(3):584–8. https://doi.org/10.1111/j.1528-157 1167.2006.00472.x 158 3. Janszky J, Fogarasi A, Toth V, Magalova V, Gyimesi C, Kovacs N, et al. Peri-ictal 159 vegetative symptoms in temporal lobe epilepsy. Epilepsy Behav. 2007 Aug;11(1):125–9. 160 https://doi.org/10.1016/j.yebeh.2007.04.015 161 4. Pietrafusa N, Trivisano M, de Palma L, Serino D, Moavero R, Benvenga A, et al. Peri-ictal 162 water drinking: a rare automatic behaviour in temporal lobe epilepsy. Epileptic Disord. 163 2015 Dec;17(4):384–96. https://doi.org/10.1684/epd.2015.0776 164 5. Szűcs A, Fogarasi A, Rásonyi G, Kelemen A, Narula L, Tóth V, et al. Peri-ictal water 165 drinking in temporal lobe epilepsy: Is it a reliable lateralizing sign? Epilepsy Behav. 2007 166 Dec;11(4):578–81. https://doi.org/10.1016/j.yebeh.2007.07.012 167 6. Lennox WG. EPILEPSY: XIII. AURA IN EPILEPSY; A STATISTICAL REVIEW OF 168 1,359 CASES. Arch Neurol Psychiatry. 1933 Aug 1;30(2):374. 169 https://doi.org/10.1001/archneurpsyc.1933.02240140138007 170 7. Alanazi GM. A Young man with Peri-ictal water drinking. Neurosciences. 2021 171 Jan;26(1):85–8. https://doi.org/10.17712/nsj.2021.1.20200074 172 8. Cascino GD, Sutula TP. Thirst and compulsive water drinking in medial basal limbic 173 epilepsy: an electroclinical and neuropathological correlation. J Neurol Neurosurg 174 Psychiatry. 1989 May 1;52(5):680–1. https://doi.org/10.1136/jnnp.52.5.680 175 9. Tanno Y, Matsudaira T, Usui N, Ogawa H, Tokumoto K, Kawaguchi N, et al. Periictal water 176 drinking revisited: Occurrence and lateralizing value in surgically confirmed patients with 177 focal epilepsy. Epilepsia Open. 2023 Mar;8(1):173–82. https://doi.org/10.1002/epi4.12690 178 10. Denton DA, McKinley MJ, Weisinger RS. Hypothalamic integration of body fluid 179 regulation. Proc Natl Acad Sci. 1996 Jul 9;93(14):7397–404. 180 https://doi.org/10.1073/pnas.93.14.7397 181 11. Trinka E, Walser G, Unterberger I, Luef G, Benke T, Bartha L, et al. Peri-ictal water 182 drinking lateralizes seizure onset to the nondominant temporal lobe. Neurology. 2003 Mar 183 11;60(5):873–6. https://doi.org/10.1212/01.WNL.0000049459.83589.6C 184 12. Musilová K, Kuba R, Brázdil M, Tyrlíková I, Rektor I. Occurrence and lateralizing value of 185 “rare” peri-ictal vegetative symptoms in temporal lobe epilepsy. Epilepsy Behav. 2010 186 Nov;19(3):372–5. https://doi.org/10.1016/j.yebeh.2010.07.010 187 13. Remillard GM, Andermann F, Gloor P, Olivier A, Martin JB. Water-drinking as ictal 188 behavior in complex partial seizures. Neurology. 1981 Feb 1;31(2):117–117. 189 https://doi.org/10.1212/WNL.31.2.117 190 14. Huang S, Al-Abri H, Sachdeva A, Alkhachroum AM, Shatzman S, Lüders H. Recurrent 191 focal seizures as a feature of status epilepticus presenting as a peri-ictal water drinking. 192 Epilepsy Behav Case Rep. 2018;10:129–32. https://doi.org/10.1016/j.ebcr.2018.09.004 193 15. Quevedo-Diaz M, Campo AT, Vila-Vidal M, Principe A, Ley M, Rocamora R. Ictal spitting 194 in non-dominant temporal lobe epilepsy: an anatomo-electrophysiological correlation. 195 Epileptic Disord. 2018 Apr;20(2):139–45. https://doi.org/10.1684/epd.2018.0963 196 197 Table 1: Summary of seizures, EEG onset, duration and details of water drinking phenomenon. 198 Sz (seizure), sec (seconds), NA (not available). 199 EEG Onset Sz Duration Onset of first drinking during a sz Number of times drinking during the sz Further remarks Sz # 1 Right Temporal 44 sec 12 sec 3 times Sz # 2 Right Temporal 66 sec 15 sec 2 times during a sz and 1 after sz ends (1.5 min after sz ended) Sz # 3 Left Temporal 47 sec NA NA Nocturnal seizure Sz # 4 Left Temporal 54 sec NA NA Left nose wiping Sz # 5 Left Temporal 63 sec 55 sec 1 time Post Ictal spitting 200 201 202 203 204 Figure 1: (A) Ictal EEG onset with rhythmic discharges in the right temporal chain, maximum at 205 F8/T4. (B) Ictal EEG onset with rhythmic discharges in the left temporal chain, maximum at T3 206 207 208 Figure 2: MRI brain (A) Flair, (B) T2 that both showed evidence of bilateral hippocampal 209 atrophy and mesial temporal sclerosis. 210