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Background Exploding Head Syndrome (EHS) is a benign parasomnia characterized by sudden, loud noises or explosive sensations occurring during sleep-wake transitions. Although it is distinct, EHS can be misinterpreted as seizures, complicating diagnosis and management.Case Presentation A 41-year-old Caucasian male with a history of well-controlled combined generalized and focal epilepsy, diagnosed 28 years ago, presented with episodes of sudden perception of loud noises and a pressure-like sensation, associated with an inability to move from the chest up during sleep. These episodes have worsened with an increase of nocturnal carbamazepine dose. The episodes occurred independently of seizures, and he remained fully oriented through them. Magnetic resonance imaging showed focal encephalomalacia in the left frontal operculum and in the superior insular region compatible with chronic ischemic changes. The electroencephalogram was unremarkable. This presentation was compatible with EHS.The patient was reassured, referred for cognitive behavioural therapy, and started on Melatonin.Conclusion This case highlights the importance of differentiating EHS from seizures, as it can mimic a worsening of epilepsy despite being completely benign. Accurate diagnosis prevents unnecessary changes in antiepileptic treatment. Further studies are required to explore the relationship of the individual antiepileptics in epilepsy patients having EHS.sathyajith.ambawatte@gmail.com