Background: Ictal epileptic headache (IEH) is caused by a focal epileptic seizure.The diagnosis can be challenging when the headache is isolated without any other symptoms.Case Report: A 16-year-old girl presented with a 5-year history of bilateral frontotemporal headaches with severe intensity lasting for 1-3 min. Past medical, physical, and developmental histories were unremarkable. Head magnetic resonance imaging showed right hippocampal sclerosis. The diagnosis of pure IEH was confirmed by video-electroencephalographic monitoring. The onset and cessation of frontal headache correlated with a right temporal discharge. The patient was diagnosed with right mesial temporal lobe epilepsy. Two years later, her seizures increased despite antiseizure medications. A right anterior temporal lobectomy was performed. The patient remained seizure-free and headache-free for 10 years. Conclusion:IEH should be considered in the differential diagnosis of brief and isolated headache, even if the headache is diffuse or contralateral to the epileptogenic focus.