Ictal Epileptic Headache in an Elderly Patient with a Hippocampal Tumor

Dear Editor, The interesting phenomenon of an association between a headache and epileptic seizure is often observed. Seizure-related headache has been subdivided into four types based on the temporal relationship: preictal headache, ictal headache, postictal headache, and interictal headache.1 Ictal headache is a very rare type of seizure-related headache,2 which has resulted in video-EEG recordings of ictal headache being exceptionally rare.3,4 In the literature, the origin of ictal headache has been predominantly reported to be the occipital lobe, and ictal headache most commonly occurs in young individuals.2,4,5 Here we report an elderly patient with a hippocampal tumor who presented with ictal headache as the sole manifestation. This is a rare case of the ictal events being documented with video-EEG recordings. A 74-year-old right-handed man was admitted to hospital with the presentation of the sudden onset of headache. His headache was unresponsive to analgesics such as paracetamol, ibuprofen, and acetaminophen, and had started approximately 1 month earlier with episodes of severe intensity that lasted for up to 1 minute. The headache was characterized by a sudden lightning strike of pain affecting the entire head. The patient was conscious during these episodes and could remember the situation occurring around him. The episodes had become much more frequent (up to 20 times per day) immediately preceding hospitalization, and they occurred daily. During a video-EEG recording session, the patient complained of a sudden, severe headache affecting the entire head. Concurrent with the headache, the EEG showed the onset of rhythmic beta activity beginning in the right temporal area and spreading to the right hemisphere lasting for approximately 30 seconds (Fig. 1A, Supplementary Video 1 in the online-only Data Supplement). The disappearance of ictal activity on the EEG corresponded to the resolution of the headache. No epileptiform discharges were present outside the headache episode. The patient did not report any other symptoms associated with the headache and did not show any other convulsive movements. Brain MRI revealed a solitary enhanced lesion in the right hippocampus, suggesting a brain tumor (Fig. 1B-E). The patient was diagnosed as having a hippocampal tumor and ictal epileptic headache based on the electroclinical and neuroimaging findings. The patient was treated with intravenous phenytoin, which resulted in the attacks subsiding immediately. No further episodes were reported at the 1-month follow-up. The pathophysiological mechanisms underlying ictal headache have not been clearly determined. Headache has previously been associated with activation of the trigeminovascular system.2,3 Cortical spreading depression is an electrophysiological phenomenon representing a neuronal depolarization wave followed by the suppression of bioelectrical activity.2,3 This phenomenon is proposed to induce activation of the trigeminovascular system, which accompanies epileptic discharge, resulting in ictal headache.2,3 Another plausible explanation is the association with synaptic transmission in the trigeminal nucleus. Antiepileptic drugs (AEDs) did not suppress the cortical spreading depression observed in an experimental study, Soo Young Bae Byung In Lee Sung Eun Kim Kyong Jin Shin Sam Yeol Ha JinSe Park Kang Min Park