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Abdominal Epilepsy, a Rare Cause of Abdominal Pain: the Need to Investigate Thoroughly As Opposed to Making Rapid Attributions of Psychogenic Causality

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Abdominal Epilepsy, a Rare Cause of Abdominal Pain: the Need to Investigate Thoroughly As Opposed to Making Rapid Attributions of Psychogenic Causality Journal of Pain Research Dovepress open access to scientific and medical research Open Access Full Text Article EDITORIAL Abdominal Epilepsy, a Rare Cause of Abdominal Pain: The Need to Investigate Thoroughly as Opposed to Making Rapid Attributions of Psychogenic Causality This article was published in the following Dove Press journal: Journal of Pain Research – Giuliano Lo Bianco 1 3 Introduction 3 Simon Thomson Abdominal pain is a nonspecific symptom which can be caused by myriad pathol- 4 Simone Vigneri ogies, resulting in frequent misdiagnosis.1 Some pathological conditions can cause 5 Hannah Shapiro paroxysmal gastrointestinal symptoms, such as porphyria, cyclical vomiting, intest- 6,7 Michael E Schatman inal malrotation, peritoneal bands, and abdominal migraine.2 Furthermore, emo- 1Università di Catania, Dipartimento di tional and psychological factors may also play an important role in the presentation Scienze Biomediche e Biotecnologiche of certain patients with gastrointestinal disorders, and accurate diagnosis can be (BIOMETEC), Catania, Italy; 2I.R.C.C.S. CROB Centro di Riferimento confounded by these. An accurate diagnosis may be delayed or even abandoned due Oncologico Basilicata, Rionero in Vulture, to the attribution of “functional” or “psychogenic” causality.3 Physicians in numer- Potenza, Italy; 3Basildon and Thurrock University Hospitals NHSFT, Orsett ous fields of practice too often respond in such a fashion when the more common Hospital, Pain Management and causes of pain conditions are ruled out,4 which potentially puts patients with rare Neuromodulation, London, Essex, UK; 4Pain Medicine Department, Santa Maria pain disorders that are challenging to diagnose at considerable risk for needless, Maddalena Hospital, Occhiobello, Rovigo, prolonged suffering. Further, the stigma associated with being diagnosed with Italy; 5Department of Biopsychology, Tufts University, Medford, MA, USA; a Somatoform Disorder or a Medically Unexplained Symptom (MUS) should not 6 Department of Diagnostic Sciences, Tufts be understated.5 University School of Dental Medicine, Boston, MA, USA; 7Department of Public One extremely rare cause of abdominal pain is abdominal epilepsy, also known as Health and Community Medicine, Tufts autonomic epilepsy.6 The typical symptoms are idiopathic, paroxysmal-episodic University School of Medicine, Boston, 7,8 MA, USA abdominal and periumbilical pain caused by a central nervous system disturbance. In addition, as with common epileptic disorders, abnormal electroencephalograms (EEGs) are usually observed (assuming they are performed), as well as loss or altera- tion of consciousness, somnolence following episodes, and a favorable response to antiepileptic drugs.6,9,10 In some patients, the irregular bouts of abdominal pain, combined with the above symptoms, can point the physician towards a diagnosis of abdominal epilepsy.2 Case Study To illustrate, we present the case of a fifty-two-year-old woman with a 15-year history of recurrent, paroxysmal abdominal pain, absence episodes and two genra- lized seizures diagnosed as abdominal epilepsy by a gastroenterologist and neurol- ogist following a lengthy period of clinical assessment. The abdominal epilepsy was experienced as abdominal pain occurring suddenly, Correspondence: Michael E Schatman resolving spontaneously and lasting for 10–15 mins with palpitation and stuttering. Tel +1425647-4880 Email [email protected] These attacks occcured at random intervals, sometimes every few days and submit your manuscript | www.dovepress.com Journal of Pain Research 2020:13 457–460 457 DovePress © 2020 Lo Bianco et al. This work is published and licensed by Dove Medical Press Limited. The full terms of this license are available at https://www.dovepress.com/ – http://doi.org/10.2147/JPR.S247767 terms.php and incorporate the Creative Commons Attribution Non Commercial (unported, v3.0) License (http://creativecommons.org/licenses/by-nc/3.0/). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed. For permission for commercial use of this work, please see paragraphs 4.2 and 5 of our Terms (https://www.dovepress.com/terms.php). Lo Bianco et al Dovepress sometimes once a month. Her symptoms during these At that point, the patient underwent exhaustive inves- episodes included cramps, episodic vomiting, headache tigations including routine blood tests, electrocardiogra- and pain experienced as a pressure sensation in her abdo- phy, abdominal ultrasound and upper gastrointestinal men that radiated to her lumbar area. Although the patient endoscopy showing only a hiatal hernia. Gastric biopsies experienced two episodes of generalized seizures, her were normal and no other abnormalities were found in any abdominal pain was typically not associated with convul- of the other general medical and neurological investiga- sions or loss of consciousness, although was typically tions. An EEG record was within normal limits. followed by somnolence, lethargy, and increased sleep. Adding pregabalin reduced the frequency and severity The patient also reported absence episodes, independent of abdominal pain and associated lethargic episodes. The of the abdominal pain. She described the absence episodes patient’s current medication regimen is fluoxetine 20 mg as “like watching the world from the TV—I know what is bid, carbamazepine 200 mg qam and 400 mg qhs, prega- happening but I cannot interact.” The patient’s pain was balin 75 mg bid, and codeine/paracetamol 30 mg/500 mg reportedly not helped by any medication. up to eight tablets qd prn. The patient denies any subse- At the age of 10 years, the patient reportedly experienced quent seizure episodes, and reports abdominal pain control an episode in which she felt dizzy and lost the ability to with which she is satisfied. According to the patient, the concentrate, experienced frequent odd sensations in her left addition of the pregabalin was the factor that she saw as ear persisting until the present and, occasionally, a pulling most salient to her recovery. sensation on her teeth. In spite of the unremarkable results Written consent to publish this report was provided yielded by an EEG investigation, the episodes of compro- from the patient prior to authors’ initiation of the writing mised consciousness continued. Her emotional response to of this article. these symptoms was a depressed mood throughout her ado- lescence and she was consequently referred for counseling. Discussion During her mid-20s, the patient continued to experience The pathophysiology and etiology of abdominal epilepsy sensations in her left ear, with these symptoms progressing remain unknown. Research suggests that the insula and to the point that she believed that she was suffering from Sylvian fissure might play an important role, contributing some type of auditory condition. At the age of thirty seven, to the etiology of the pathology.10 In some cases, brain the patient suffered her first episode of the abdominal symp- tumors have been assessed as the cause of abdominal toms combined with a generalized seizure. Specifically, she epilepsy.6,11 In other investigations, the possible role of awakened with abdominal pain during the night and went to somatosensory area I of the brain in pain perception has the toilet, where she collapsed and had a generalized seizure. been suggested, with the development of partial seizures This event included tongue biting and incontinence, with the representing a potential link between abdominal pain and entire episode lasting ten minutes. She was confused post- a parietal lobe hemorrhage.12 Other studies on abdominal ictally, and the patient required several days to fully recover. epilepsy have indicated that linked causes could be right She was prescribed carbamazepine and reportedly did not parietal and occipital encephalomalacia, biparietal atrophy suffer any further generalized seizures for well over a decade. and bilateral perisylvian polymicrogyria.13 In addition to At the age of fifty, the patient suffered another general- abdominal pain, the main symptoms associated with focal ized seizure. As was the case in her initial seizure, she epilepsy with ictal abdominal pain or abdominal epilepsy reportedly experienced abdominal pain immediately prior include nausea, vomiting, lethargy, and hunger.10,14 to the generalized seizure. Symptoms included loss of A 2001 study demonstrated that over 4% of patients with consciousness and involuntarily shaking of her limbs, focal epilepsy experienced painful epileptic auras, with 5% although no incontinence or tongue biting. Following experiencing abdominal pain in cases of temporal lobe this second seizure in 2018, the patient underwent an epilepsy and 50% experiencing abdominal pain among MRI of her brain. Results indicated the presence of those with frontal lobe epilepsy.15 Numerous studies a pituitary lesion which was further evaluated with dedi- have suggested that in some cases, abdominal symptoms cated pituitary imaging. This imaging confirmed a 10 mm- might be the only element of epileptic activity.2,10,16,17 sized Rathke’s cyst. Although an endocrinological referral The key features required in specifying a diagnosis of was made, all endocrinologic assessments were within abdominal epilepsy are often idiopathic and irregular normal limits. abdominal symptoms, loss of consciousness and focal 458 submit your manuscript | www.dovepress.com
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