Abdominal Epilepsy: Report of two cases and literature review

Abdominal Epilepsy: Report of two cases and literature review | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Abdominal Epilepsy: Report of two cases and literature review Ying Zhang, Ying An, Jialong Qi, Zhengji Song This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4485374/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Abdominal epilepsy (AE) is a rare form of epilepsy characterized by paroxysmal abdominal pain accompanied by syncope. It often presents diagnostic challenges due to its diverse clinical manifestations. Herein, we present two cases, a 28-year-old male and a 55-year-old female, with a decade-long history of intermittent abdominal pain and syncope. Despite multiple consultations, a definitive diagnosis remained elusive until electroencephalography (EEG) revealed characteristic abnormalities. Two cases underscore the importance of recognizing AE as a potential cause of unexplained abdominal pain and syncope. Therefore, we recommend that for patients with unexplained abdominal pain accompanied by altered consciousness, after ruling out common causes, consideration should be given to AE. In the meantime, we summarize the possible molecular mechanisms for AE development. Abdominal epilepsy Abdominal pain Dysfunction of consciousness Electroencephalography Brain-gut axis Figures Figure 1 Figure 2 Introduction Abdominal pain is a nonspecific symptom that can be caused by a variety of pathological conditions, including porphyria, cyclic vomiting syndrome, irritable bowel syndrome (IBS), copper toxicity, malrotation, and abdominal migraines, often leading to misdiagnosis. Additionally, emotional and psychological factors can complicate accurate diagnosis.Abdominal epilepsy (AE) is a rare type of epilepsy characterized by paroxysmal abdominal pain accompanied by alterations in consciousness. The mechanisms underlying AE remain unclear, though some studies suggest that its occurrence may be related to the brain-gut axis and brain lesions.Early recognition and appropriate management of AE are crucial to prevent unnecessary investigations and improve patient outcomes. Here, we describe two cases that underscore the importance of clinical suspicion and comprehensive assessment in patients presenting with recurrent abdominal pain and altered consciousness. We also review recent advancements in understanding the pathophysiology of AE. Case reports Case 1 This case involves a 55-year-old middle-aged woman who initially presented with intermittent abdominal pain at the age of 8. The abdominal discomfort progressed over time, accompanied by sweating, fatigue, and a sense of bowel movement. Subsequently, she experienced episodes of loss of consciousness. These episodes lasted for approximately 1–2 minutes, and the patient would regain consciousness with relief from abdominal pain. The second similar episode occurred at the age of 15, with subsequent episodes recurring irregularly (every 3–5 years) and lasting for 30 to 60 minutes, resolving spontaneously. Over the past year, the frequency of episodes had increased, occurring four times and often associated with fatigue. The patient's neurological examination, laboratory tests, and imaging studies showed no significant abnormalities, but in EEG evaluation, mildly abnormal findings were observed. Further, a 24-hour ambulatory EEG was conducted. The results revealed that the interictal EEG during both wakefulness and sleep demonstrated bilateral spikes and slow waves (epileptic waves), with a higher frequency on the left side. The patient initiated treatment with levetiracetam (500 mg/q12), and over the past two months of follow-up, she has not experienced further episodes. Case 2 A 28-year-old male presented with a 10-year history of intermittent abdominal pain and syncope. The abdominal pain, occurring following exertion, was described as paroxysmal colic without fixed localization. Concurrently, there was a sensation of defecation, followed by syncope lasting approximately one minute. The episodes recurred once annually, often during the Spring Festival period. Physical examinations of the abdomen and nervous system yielded unremarkable findings. Routine stool tests, abdominal ultrasonography, and gastrointestinal endoscopy revealed normal results. Similarly, an MRI of the head and echocardiography demonstrated no abnormalities. However, EEG revealed characteristic abnormalities during sleep, including single and short bursts of high-amplitude sharp waves and 2-4Hz spike-and-wave complexes, with a predominance on the right side. Topographic EEG waves indicated increased delta wave power in the bilateral frontal regions, consistent with a diagnosis of epilepsy. The patient commenced regular administration of sodium valproate (500mg bid). Subsequently, he remained seizure-free during the 10-year follow-up period, with no recurrence of abdominal pain or syncope. Table 1 Comparison of AE Progression in two cases. Case Age of onset Clinical Presentation Episode Frequency EGG Treatment Follow-up Case1 8-year-old, Intermittent abdominal pain with syncopal episodes lasting 47 years. Resolve spontaneously every 3–5 years, lasting 0.5-1 hour each. wakefulness and sleep demonstrated bilateral spikes and slow waves (epileptic waves), with a higher frequency on the left side. Levetiracetam (500 mg bid) Ten years, No recurrence. Case2 18-year-old Intermittent abdominal pain with syncope lasting 10 years. Once yearly, lasting approximately 1 minute During sleep, intermittent high-amplitude sharp waves and spike-slow wave complexes were observed, predominantly on the right side, occurring 24 times. Valproate (500mg bid) Four months, No recurrence. Discussion Epilepsy, a chronic neurological disorder, affects approximately 70 million people worldwide. AE, also known as AE syndrome, is a type of epilepsy characterized by recurrent abdominal pain. It accounts for approximately 1% of all epilepsy cases. However, its pathogenesis remains poorly understood. The brain-gut-microbiota axis, which governs inflammatory pathways, plays a pivotal role in A [ 1 – 4 ] . Dysregulation in this axis, characterized by imbalances in neural networks, neuroendocrine signaling, and immune responses, may contribute to the development of epilepsy. Researchers found intestinal pathogens probiotics could upregulate the production of epilepsy-promoting metabolites, the secretion of inflammatory factors, and so on, which lead to abnormal GABA/glutamate ratio and then induce epilepsy [ 5 ] . Additionally, cortical dysregulation in the autonomic nervous system, manifested by disruptions in ion channels and synaptic protein expression, has been implicated in AE pathophysiology [ 6 , 7 ] . Lesions affecting the central autonomic nervous system, particularly in subcortical and hypothalamic regions, may disrupt visceral sensory perception and consciousness, further linking to the cortical hub of the autonomic nervous system [ 8 ] . Notably, EEG abnormalities, primarily in the temporal lobes, are common in AE, suggesting a neuroexcitatory pathway involving the amygdala and the dorsal motor nucleus of the vagus nerve [ 9 ] . While temporal lobe epilepsy (TLE) is the typical association, various etiological factors, including prematurity, cortical malformations, and brain tumors, may contribute to the development of A [ 10 , 11 ] . Moreover, insular and opercular regions have emerged as potential sources of visceral sensations [ 12 ] , adding complexity to AE diagnosis. These advancements underscore the multifactorial nature of AE pathogenesis and emphasize the importance of further research to elucidate its mechanisms fully. AE primarily affects the pediatric population, with fewer occurrences reported in adolescents and adults. Our two cases presented in childhood but were likely diagnosed until adolescence or later due to the similarity of abdominal pain symptoms with other non-abdominal epileptic disorders. This similarity often leads to misdiagnosis as conditions such as porphyria, cyclic vomiting syndrome, copper poisoning, enteritis, peptic ulcer, or abdominal migraines, especially when patients have recurrent episodes over the years and various examinations fail to detect significant abnormalities, or even when symptoms resolve on their own without the need for medication intervention, this can easily be misdiagnosed as psychogenic disorders. Consequently, the diagnosis may be delayed until adolescence or even later, as was the case in our patients. Upon further history-taking, both patients exhibited significant post-seizure neurological symptoms, including loss of consciousness following abdominal pain. Consequently, we promptly initiated comprehensive 24-hour ambulatory EEG monitoring upon admission, leading to a swift diagnosis of AE upon symptom resolution with antiepileptic drug use. Hence, consideration of AE is warranted in several scenarios [ 13 ] : (1) When patients exhibit paroxysmal gastrointestinal and autonomic symptoms without an apparent cause, such as abdominal pain, vomiting, nausea, flushing, palpitation, or stuttering; (2) When symptoms of central nervous system (CNS) disturbance, such as alterations in mental status, headache, dizziness, or convulsions, are present; (3) When abnormal EEG findings suggestive of epileptic activity are observed; and (4) When symptom improvement is noted with antiseizure medications. Conclusion When confronted with patients presenting recurrent abdominal pain alongside neurological symptoms, it is imperative to entertain a heightened suspicion of AE. A thorough examination, particularly a comprehensive electroencephalogram (EEG), is crucial for confirming the diagnosis. Once the diagnosis is confirmed, systematic and standardized antiepileptic drug therapy should be promptly initiated. Declarations Authors’ contribution Ying Zhang and Ying An wrote the manuscript, Jialong Qi reviewed and edited the manuscript ,Ying An and Zhengji Song admited and treated the patients. Declaration of competing interest We have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper. Ethical Statement The publication of this case report and any accompanying images has been done with the consent of the patients involved. Acknowledgment This work was supported by the National Natural Science Foundation of China (No.82360120) the Yunnan Province Famous Doctor program (KH-SWR-MY-2019-009), Yunnan Provincial Leading Talent Program (L-2017009). References Ding X, Zhou J, Zhao L, et al. Intestinal Flora Composition Determines Microglia Activation and Improves Epileptic Episode Progress. Front Cell Infect Microbiol. 2022. 12: 835217. Gernone F, Uva A, Silvestrino M, Cavalera MA, Zatelli A. Role of Gut Microbiota through Gut-Brain Axis in Epileptogenesis: A Systematic Review of Human and Veterinary Medicine. Biology (Basel). 2022. 11(9): 1290. Gong X, Liu L, Li X, et al. Neuroprotection of cannabidiol in epileptic rats: Gut microbiome and metabolome sequencing. Front Nutr. 2022. 9: 1028459. Mu X, Zhang X, Gao H, Gao L, Li Q, Zhao C. Crosstalk between peripheral and the brain-resident immune components in epilepsy. J Integr Neurosci. 2022. 21(1): 9. Ding M, Lang Y, Shu H, Shao J, Cui L. Microbiota-Gut-Brain Axis and Epilepsy: A Review on Mechanisms and Potential Therapeutics. Front Immunol. 2021. 12: 742449. Mantegazza M, Curia G, Biagini G, Ragsdale DS, Avoli M. Voltage-gated sodium channels as therapeutic targets in epilepsy and other neurological disorders. Lancet Neurol. 2010. 9(4): 413-24. Valtorta F, Benfenati F, Zara F, Meldolesi J. PRRT2: from Paroxysmal Disorders to Regulation of Synaptic Function. Trends Neurosci. 2016. 39(10): 668-679. Ostwal P. Abdominal epilepsy: Is it so common. J Pediatr Neurosci. 2013. 8(3): 253. Mpondo BC, Mwasada G, Nyundo AA. Abdominal epilepsy, an uncommon cause of chronic and recurrent abdominal pain: a case report. Clin Case Rep. 2016. 4(12): 1117-1119. Franzon RC, Lopes CF, Schmutzler KM, Morais MI, Guerreiro MM. Recurrent abdominal pain: when should an epileptic seizure be suspected. Arq Neuropsiquiatr. 2002. 60(3-A): 628-30. Topno N, Gopasetty MS, Kudva A, B L. Abdominal epilepsy and foreign body in the abdomen--dilemma in diagnosis of abdominal pain. Yonsei Med J. 2005. 46(6): 870-3. Jobst BC, Gonzalez-Martinez J, Isnard J, et al. The Insula and Its Epilepsies. Epilepsy Curr. 2019. 19(1): 11-21. Al-Beltagi M, Saeed NK. Epilepsy and the gut: Perpetrator or victim. World J Gastrointest Pathophysiol. 2022. 13(5): 143-156. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-4485374","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":310243112,"identity":"e02965c8-6e7c-40fe-a169-b212b03f76d0","order_by":0,"name":"Ying Zhang","email":"","orcid":"","institution":"Medical school, Kunming University of Science and Technology","correspondingAuthor":false,"prefix":"","firstName":"Ying","middleName":"","lastName":"Zhang","suffix":""},{"id":310243113,"identity":"ee5d3822-f06d-40eb-b391-d6403f87318f","order_by":1,"name":"Ying An","email":"","orcid":"","institution":"Yunnan Digestive Endoscopy Clinical Medical Center, Department of Gastroenterology. The First People's Hospital of Yunnan Province.","correspondingAuthor":false,"prefix":"","firstName":"Ying","middleName":"","lastName":"An","suffix":""},{"id":310243116,"identity":"a7968d74-7850-4b58-a00d-4bdb6fb5bd13","order_by":2,"name":"Jialong Qi","email":"","orcid":"","institution":"Yunnan Digestive Endoscopy Clinical Medical Center, Department of Gastroenterology. The First People's Hospital of Yunnan Province.","correspondingAuthor":false,"prefix":"","firstName":"Jialong","middleName":"","lastName":"Qi","suffix":""},{"id":310243117,"identity":"b4d9eb52-8d6b-4108-bff2-349281d01a61","order_by":3,"name":"Zhengji Song","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA9ElEQVRIie2QsUoDQRCGNwzcNaubcg/JO4wsrI34LLccaJtK0rkhoM2StPEtDu4FBgLarNhuGZvUK5YK8YKN1V5Kwf26GeZj/hnGMpk/iGAjizXuQfCfxsgOKZUFO41TKCvXV3SMglTa93UEgf5oJZh5x7E4U96rj8guJy3BbpsMFsxCceRKvzgtiV2rlooLTCmnwdz3imz0K9d9sI1piRcymSyYh0+OeNetuIrE9sPKOBh7vsYa8MRhH4yGlcq9WYxIIP3TrfTYqMdNoZOKKG+2WH8RCNd0cTa7miyfF7v0LeP6d3V4FSTnD2toaCKTyWT+Pd9MX05+fATKFAAAAABJRU5ErkJggg==","orcid":"","institution":"Yunnan Digestive Endoscopy Clinical Medical Center, Department of Gastroenterology. The First People's Hospital of Yunnan Province.","correspondingAuthor":true,"prefix":"","firstName":"Zhengji","middleName":"","lastName":"Song","suffix":""}],"badges":[],"createdAt":"2024-05-27 13:29:05","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4485374/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4485374/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":58610636,"identity":"2976b298-6c90-4270-9c22-76ef9408569b","added_by":"auto","created_at":"2024-06-18 22:06:44","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":1451265,"visible":true,"origin":"","legend":"\u003cp\u003eLaboratory and imaging findings. A) Normal gastrointestinal endoscopy results. B) Thoracic and abdominal contrast-enhanced computed tomography (CECT): No evidence of neuroendocrine tumors. C) Echocardiography (UCG): No abnormal cardiac structure noted. D)Head magnetic resonance imaging (MRI): No significant abnormalities were detected. E)EEG: Left-sided sharp-slow waves captured during sleep. (case 1)\u003c/p\u003e","description":"","filename":"Figure1.png","url":"https://assets-eu.researchsquare.com/files/rs-4485374/v1/520430eae11c70806ba48f13.png"},{"id":58610637,"identity":"49a4e883-3b7f-4203-80a9-0ae63f3a3df8","added_by":"auto","created_at":"2024-06-18 22:06:44","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":2653243,"visible":true,"origin":"","legend":"\u003cp\u003eLaboratory and imaging findings. A) Sleep EEG: Brief high-amplitude sharp waves and 2-4 Hz slow waves observed predominantly on the right side. Total of 24 sharp-slow wave complexes, each lasting 0.5 seconds. B) Head magnetic resonance imaging (MRI):Bilateral caudate nucleus head small lacunar infarcts. (case 2)\u003c/p\u003e","description":"","filename":"Figure2.png","url":"https://assets-eu.researchsquare.com/files/rs-4485374/v1/63d5bacf48a2769987bf836c.png"},{"id":69978216,"identity":"1064cc87-7af2-474f-af11-fe0dbcfc1ec8","added_by":"auto","created_at":"2024-11-27 07:32:46","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":6983970,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4485374/v1/950440d7-6683-4adc-801f-258bfad7d6b1.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"\u003cp\u003eAbdominal Epilepsy: Report of two cases and literature review\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eAbdominal pain is a nonspecific symptom that can be caused by a variety of pathological conditions, including porphyria, cyclic vomiting syndrome, irritable bowel syndrome (IBS), copper toxicity, malrotation, and abdominal migraines, often leading to misdiagnosis. Additionally, emotional and psychological factors can complicate accurate diagnosis.Abdominal epilepsy (AE) is a rare type of epilepsy characterized by paroxysmal abdominal pain accompanied by alterations in consciousness. The mechanisms underlying AE remain unclear, though some studies suggest that its occurrence may be related to the brain-gut axis and brain lesions.Early recognition and appropriate management of AE are crucial to prevent unnecessary investigations and improve patient outcomes. Here, we describe two cases that underscore the importance of clinical suspicion and comprehensive assessment in patients presenting with recurrent abdominal pain and altered consciousness. We also review recent advancements in understanding the pathophysiology of AE.\u003c/p\u003e"},{"header":"Case reports","content":"\u003cp\u003e \u003cstrong\u003eCase 1\u003c/strong\u003e \u003cp\u003eThis case involves a 55-year-old middle-aged woman who initially presented with intermittent abdominal pain at the age of 8. The abdominal discomfort progressed over time, accompanied by sweating, fatigue, and a sense of bowel movement. Subsequently, she experienced episodes of loss of consciousness. These episodes lasted for approximately 1\u0026ndash;2 minutes, and the patient would regain consciousness with relief from abdominal pain. The second similar episode occurred at the age of 15, with subsequent episodes recurring irregularly (every 3\u0026ndash;5 years) and lasting for 30 to 60 minutes, resolving spontaneously. Over the past year, the frequency of episodes had increased, occurring four times and often associated with fatigue.\u003c/p\u003e \u003c/p\u003e \u003cp\u003eThe patient's neurological examination, laboratory tests, and imaging studies showed no significant abnormalities, but in EEG evaluation, mildly abnormal findings were observed. Further, a 24-hour ambulatory EEG was conducted. The results revealed that the interictal EEG during both wakefulness and sleep demonstrated bilateral spikes and slow waves (epileptic waves), with a higher frequency on the left side. The patient initiated treatment with levetiracetam (500 mg/q12), and over the past two months of follow-up, she has not experienced further episodes.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003cstrong\u003eCase 2\u003c/strong\u003e \u003cp\u003eA 28-year-old male presented with a 10-year history of intermittent abdominal pain and syncope. The abdominal pain, occurring following exertion, was described as paroxysmal colic without fixed localization. Concurrently, there was a sensation of defecation, followed by syncope lasting approximately one minute. The episodes recurred once annually, often during the Spring Festival period.\u003c/p\u003e \u003c/p\u003e \u003cp\u003ePhysical examinations of the abdomen and nervous system yielded unremarkable findings. Routine stool tests, abdominal ultrasonography, and gastrointestinal endoscopy revealed normal results. Similarly, an MRI of the head and echocardiography demonstrated no abnormalities. However, EEG revealed characteristic abnormalities during sleep, including single and short bursts of high-amplitude sharp waves and 2-4Hz spike-and-wave complexes, with a predominance on the right side. Topographic EEG waves indicated increased delta wave power in the bilateral frontal regions, consistent with a diagnosis of epilepsy.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eThe patient commenced regular administration of sodium valproate (500mg bid). Subsequently, he remained seizure-free during the 10-year follow-up period, with no recurrence of abdominal pain or syncope.\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003eComparison of AE Progression in two cases.\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"7\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c4\" colnum=\"4\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c5\" colnum=\"5\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c6\" colnum=\"6\"\u003e\u003c/div\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c7\" colnum=\"7\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCase\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003eAge of onset\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003eClinical Presentation\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c4\"\u003e \u003cp\u003eEpisode Frequency\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c5\"\u003e \u003cp\u003eEGG\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c6\"\u003e \u003cp\u003eTreatment\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c7\"\u003e \u003cp\u003eFollow-up\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCase1\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e8-year-old,\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eIntermittent abdominal pain with syncopal episodes lasting 47 years.\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003eResolve spontaneously every 3\u0026ndash;5 years, lasting 0.5-1 hour each.\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003ewakefulness and sleep demonstrated bilateral spikes and slow waves (epileptic waves), with a higher frequency on the left side.\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003eLevetiracetam\u003c/p\u003e \u003cp\u003e(500 mg bid)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003eTen years, No recurrence.\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eCase2\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e \u003cp\u003e18-year-old\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e \u003cp\u003eIntermittent abdominal pain with syncope\u003c/p\u003e \u003cp\u003elasting 10 years.\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c4\"\u003e \u003cp\u003eOnce yearly, lasting approximately 1 minute\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c5\"\u003e \u003cp\u003eDuring sleep, intermittent high-amplitude sharp waves and spike-slow wave complexes were observed, predominantly on the right side, occurring 24 times.\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c6\"\u003e \u003cp\u003eValproate\u003c/p\u003e \u003cp\u003e(500mg bid)\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c7\"\u003e \u003cp\u003eFour months, No recurrence.\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eEpilepsy, a chronic neurological disorder, affects approximately 70\u0026nbsp;million people worldwide. AE, also known as AE syndrome, is a type of epilepsy characterized by recurrent abdominal pain. It accounts for approximately 1% of all epilepsy cases. However, its pathogenesis remains poorly understood. The brain-gut-microbiota axis, which governs inflammatory pathways, plays a pivotal role in A\u003csup\u003e[\u003cspan additionalcitationids=\"CR2 CR3\" citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e]\u003c/sup\u003e. Dysregulation in this axis, characterized by imbalances in neural networks, neuroendocrine signaling, and immune responses, may contribute to the development of epilepsy. Researchers found intestinal pathogens probiotics could upregulate the production of epilepsy-promoting metabolites, the secretion of inflammatory factors, and so on, which lead to abnormal GABA/glutamate ratio and then induce epilepsy\u003csup\u003e[\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e]\u003c/sup\u003e. Additionally, cortical dysregulation in the autonomic nervous system, manifested by disruptions in ion channels and synaptic protein expression, has been implicated in AE pathophysiology\u003csup\u003e[\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e, \u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e]\u003c/sup\u003e. Lesions affecting the central autonomic nervous system, particularly in subcortical and hypothalamic regions, may disrupt visceral sensory perception and consciousness, further linking to the cortical hub of the autonomic nervous system\u003csup\u003e[\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e]\u003c/sup\u003e. Notably, EEG abnormalities, primarily in the temporal lobes, are common in AE, suggesting a neuroexcitatory pathway involving the amygdala and the dorsal motor nucleus of the vagus nerve\u003csup\u003e[\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e]\u003c/sup\u003e. While temporal lobe epilepsy (TLE) is the typical association, various etiological factors, including prematurity, cortical malformations, and brain tumors, may contribute to the development of A\u003csup\u003e[\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]\u003c/sup\u003e. Moreover, insular and opercular regions have emerged as potential sources of visceral sensations\u003csup\u003e[\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e]\u003c/sup\u003e, adding complexity to AE diagnosis. These advancements underscore the multifactorial nature of AE pathogenesis and emphasize the importance of further research to elucidate its mechanisms fully.\u003c/p\u003e \u003cp\u003eAE primarily affects the pediatric population, with fewer occurrences reported in adolescents and adults. Our two cases presented in childhood but were likely diagnosed until adolescence or later due to the similarity of abdominal pain symptoms with other non-abdominal epileptic disorders. This similarity often leads to misdiagnosis as conditions such as porphyria, cyclic vomiting syndrome, copper poisoning, enteritis, peptic ulcer, or abdominal migraines, especially when patients have recurrent episodes over the years and various examinations fail to detect significant abnormalities, or even when symptoms resolve on their own without the need for medication intervention, this can easily be misdiagnosed as psychogenic disorders. Consequently, the diagnosis may be delayed until adolescence or even later, as was the case in our patients. Upon further history-taking, both patients exhibited significant post-seizure neurological symptoms, including loss of consciousness following abdominal pain. Consequently, we promptly initiated comprehensive 24-hour ambulatory EEG monitoring upon admission, leading to a swift diagnosis of AE upon symptom resolution with antiepileptic drug use.\u003c/p\u003e \u003cp\u003eHence, consideration of AE is warranted in several scenarios\u003csup\u003e[\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e]\u003c/sup\u003e: (1) When patients exhibit paroxysmal gastrointestinal and autonomic symptoms without an apparent cause, such as abdominal pain, vomiting, nausea, flushing, palpitation, or stuttering; (2) When symptoms of central nervous system (CNS) disturbance, such as alterations in mental status, headache, dizziness, or convulsions, are present; (3) When abnormal EEG findings suggestive of epileptic activity are observed; and (4) When symptom improvement is noted with antiseizure medications.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eWhen confronted with patients presenting recurrent abdominal pain alongside neurological symptoms, it is imperative to entertain a heightened suspicion of AE. A thorough examination, particularly a comprehensive electroencephalogram (EEG), is crucial for confirming the diagnosis. Once the diagnosis is confirmed, systematic and standardized antiepileptic drug therapy should be promptly initiated.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eAuthors\u0026rsquo; contribution\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eYing Zhang and Ying An wrote the manuscript, Jialong Qi reviewed and edited the manuscript ,Ying An and Zhengji Song admited and treated the patients.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eDeclaration of competing interest\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWe have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eEthical Statement\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe publication of this case report and any accompanying images has been done with the consent of the patients involved.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgment\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis work was supported by the National Natural Science Foundation of China (No.82360120) the Yunnan Province Famous Doctor program (KH-SWR-MY-2019-009), Yunnan Provincial Leading Talent Program (L-2017009).\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eDing X, Zhou J, Zhao L, et al. Intestinal Flora Composition Determines Microglia Activation and Improves Epileptic Episode Progress. Front Cell Infect Microbiol. 2022. 12: 835217.\u003c/li\u003e\n\u003cli\u003eGernone F, Uva A, Silvestrino M, Cavalera MA, Zatelli A. Role of Gut Microbiota through Gut-Brain Axis in Epileptogenesis: A Systematic Review of Human and Veterinary Medicine. Biology (Basel). 2022. 11(9): 1290.\u003c/li\u003e\n\u003cli\u003eGong X, Liu L, Li X, et al. Neuroprotection of cannabidiol in epileptic rats: Gut microbiome and metabolome sequencing. Front Nutr. 2022. 9: 1028459.\u003c/li\u003e\n\u003cli\u003eMu X, Zhang X, Gao H, Gao L, Li Q, Zhao C. Crosstalk between peripheral and the brain-resident immune components in epilepsy. J Integr Neurosci. 2022. 21(1): 9.\u003c/li\u003e\n\u003cli\u003eDing M, Lang Y, Shu H, Shao J, Cui L. Microbiota-Gut-Brain Axis and Epilepsy: A Review on Mechanisms and Potential Therapeutics. Front Immunol. 2021. 12: 742449.\u003c/li\u003e\n\u003cli\u003eMantegazza M, Curia G, Biagini G, Ragsdale DS, Avoli M. Voltage-gated sodium channels as therapeutic targets in epilepsy and other neurological disorders. Lancet Neurol. 2010. 9(4): 413-24.\u003c/li\u003e\n\u003cli\u003eValtorta F, Benfenati F, Zara F, Meldolesi J. PRRT2: from Paroxysmal Disorders to Regulation of Synaptic Function. Trends Neurosci. 2016. 39(10): 668-679.\u003c/li\u003e\n\u003cli\u003eOstwal P. Abdominal epilepsy: Is it so common. J Pediatr Neurosci. 2013. 8(3): 253.\u003c/li\u003e\n\u003cli\u003eMpondo BC, Mwasada G, Nyundo AA. Abdominal epilepsy, an uncommon cause of chronic and recurrent abdominal pain: a case report. Clin Case Rep. 2016. 4(12): 1117-1119.\u003c/li\u003e\n\u003cli\u003eFranzon RC, Lopes CF, Schmutzler KM, Morais MI, Guerreiro MM. Recurrent abdominal pain: when should an epileptic seizure be suspected. Arq Neuropsiquiatr. 2002. 60(3-A): 628-30.\u003c/li\u003e\n\u003cli\u003eTopno N, Gopasetty MS, Kudva A, B L. Abdominal epilepsy and foreign body in the abdomen--dilemma in diagnosis of abdominal pain. Yonsei Med J. 2005. 46(6): 870-3.\u003c/li\u003e\n\u003cli\u003eJobst BC, Gonzalez-Martinez J, Isnard J, et al. The Insula and Its Epilepsies. Epilepsy Curr. 2019. 19(1): 11-21.\u003c/li\u003e\n\u003cli\u003eAl-Beltagi M, Saeed NK. Epilepsy and the gut: Perpetrator or victim. World J Gastrointest Pathophysiol. 2022. 13(5): 143-156.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Abdominal epilepsy, Abdominal pain, Dysfunction of consciousness, Electroencephalography, Brain-gut axis","lastPublishedDoi":"10.21203/rs.3.rs-4485374/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4485374/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eAbdominal epilepsy (AE) is a rare form of epilepsy characterized by paroxysmal abdominal pain accompanied by syncope. It often presents diagnostic challenges due to its diverse clinical manifestations. Herein, we present two cases, a 28-year-old male and a 55-year-old female, with a decade-long history of intermittent abdominal pain and syncope. Despite multiple consultations, a definitive diagnosis remained elusive until electroencephalography (EEG) revealed characteristic abnormalities. Two cases underscore the importance of recognizing AE as a potential cause of unexplained abdominal pain and syncope. Therefore, we recommend that for patients with unexplained abdominal pain accompanied by altered consciousness, after ruling out common causes, consideration should be given to AE. In the meantime, we summarize the possible molecular mechanisms for AE development.\u003c/p\u003e","manuscriptTitle":"Abdominal Epilepsy: Report of two cases and literature review","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-06-18 22:06:40","doi":"10.21203/rs.3.rs-4485374/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"7c6989ea-bcf2-4fa6-989d-8a85731512d6","owner":[],"postedDate":"June 18th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2024-11-27T07:24:25+00:00","versionOfRecord":[],"versionCreatedAt":"2024-06-18 22:06:40","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4485374","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4485374","identity":"rs-4485374","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}