BackgroundSuperficial temporal artery aneurysm is a rare vascular abnormality without specific clinical symptoms. In this case report, we present the case of a patient with superficial temporal artery aneurysm who was diagnosed with migraine headache at first.Case presentationA 60-year-old Iranian man with a previous history of headaches, who did not respond properly to the treatments following the initial diagnosis of migraine, presented with a painless lump in the left temporal region, and he was diagnosed with superficial temporal artery aneurysm via Doppler ultrasound. Finally, surgical removal of the left superficial temporal artery aneurysm was performed.ConclusionsThis case shows the importance of vascular causes in the approach to headache etiologies, especially when the headache is prolonged without proper responses to treatment. Computed tomography angiography and magnetic resonance angiography are appropriate diagnostic methods for aneurysm detection that should be considered in future studies.