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Abdominal pain associated with musical hallucinations: a case report

Sir, Abdominal pain is a very uncommon clinical manifestation of epilepsy, occurring in 2–4% of cases [1]. Amongst these patients, painful abdominal crisis are described in approximately 30% of cases. The pain is, generally, abrupt in onset, localized to the mid-abdomen or upper abdomen, and is brief in duration (few minutes). In some cases, pain may last longer. Sometimes, pain can be the only manifestation of epilepsy and, in such cases, patients are often misdiagnosed as having psychogenic pain [2]. We report the case of a patient who presented abdominal pain associated with musical hallucinations. A 45-year-old Caucasian female was admitted in June 2003 in our Neurological Department for a 10-year history of abdominal pain attacks. The pain was described as being of sudden onset, generally lasting from 2 min to 2 h, with increasing severity, then disappearing abruptly as it started. The patient localized the pain deeply in the periomphalic region and described it as an intestinal colic. Seldom the patient observed diarrhea immediately after the crisis. At presentation, the patient suffered from one to three attacks per month. The previous diagnosis was of psychogenic pain. Anamnestic investigation disclosed that, in the previous 5-year period, the patient suffered of complex musical hallucinations. The hallucinations usually consisted of both the melodies and the words of a specific song. The patient was able to identify the singer, a popular Italian artist. The musical hallucinations, generally, occurred immediately after the abdominal pain attacks. Seldom, there was no clear relationship with pain. They started abruptly and were identified, sometimes after a period of doubt, as hallucinations. On admission in our hospital, neurological examination and her optic fundus were normal. There was no family history for epilepsy or migraine. No history of birth injury or head trauma was reported. Pure-tone audiometry was normal. Routine blood, urine analysis, ECG, chest X-rays brain CT scan, MRI of the brain and of the spine were normal. During EEG registration the patient developed a crisis, with abdominal pain followed by musical hallucinations. The exam disclosed an evolutionary pattern with continuous spikes and sharp-waves complexes beginning in the left mid-temporal region (T5), then spreading to anterior temporal region (T3), parietal (P3) and frontal (F1, F3 and F7) regions (Fig. 1). Treatment was started with carbamazepine gradually titrated up to 800 mg/day. After a 6-month period, both abdominal pain attacks and musical hallucinations disappeared. After a 1-year period without attacks, the patient spontaneously reduced carbamazepine to 400 mg/day and the episodes returned. As returning to the prescribed dosage during a 12month period, she has had no further recurrence of abdominal pain or musical hallucinations. To the best of our knowledge, this is the first report of musical hallucinations associated with abdominal pain of epileptic origin. This case report raises several points of discussion. First of all, the presumed site of origin of the crisis is unclear. Studies in animal models showed that electrical stimulation of the splanchnic nerve cause-evoked responses both in the contralateral primary sensory area (SI) and in bilateral second sensory area (SSII) [3]. In the monkey, the anterior part of SSII responds to low-threshold stimuli whilst the posterior portion responds to noxious stimuli. In man, the primary sensation of pain has been thought to be located primarily at the level of the thalamus, with little or no cortical representation of pain. However, several reports in patients with vascular and neoplastic diseases showed that several areas of the cerebral cortex may process nociceptive information. Recent studies, using both positron emission tomography and functional magnetic resonance imaging, showed that painful stimuli causes significant activation of the contralateral SI, SSII and anterior cingulated cortices [4]. Then, in our patient no brain abnormality was disclosed and the aetiology of painful seizures remained obscure. Epileptic abdominal pain was described in association with birth injury, febrile seizures, head trauma, vascular abnormalities and tumours. Finally, the pathophysiological mechanisms underlying abdominal pain in epilepsy are unclear. The most probably explanation for the epileptic pain is that seizures cause spreading depression that interferes with inhibitory afferent input to SII from the thalamus [5]. In conclusion, our case report shows that patients with abdominal pain of unexplained origin must be evaluated also for epilepsy. EEG studies and a therapeutic trail with anticonvulFigure 1 The EEG registration of the seizure: increasing spikes and sharp-waves in the left anterior-mid temporal lobe.