Study Objectives: In nocturnal frontal lobe epilepsy (NFLE), seizures occur almost exclusively during NREM sleep. Why precisely these seizures are sleep-bound remains unknown. Studies of patients with nonlesional familial forms of NFLE have suggested the arousal system may play a major role in their pathogenesis. We report the case of a patient with pharmaco-resistant, probably cryptogenic form of non-familial NFLE and strictly sleep-bound seizures that could be elicited by alerting stimuli and were associated with ictal bilateral thalamic and right orbital-insular hyperperfusion on SPECT imaging. Design: Case report. Setting: University Hospital Zürich. Patients or Participants: One patient with pharmaco-resistant epilepsy. Conclusion: This case shows that the arousal system plays a fundamental role also in cryptogenic non-familial forms of NFLE.
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