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The aim of this article is to provide neuroimaging data on cases with familial hemiplegic migraine (FHM). A 14-year-old male presented normal diffusion-weighted magnetic resonance imaging (DWI) and fluid-attenuated inversion recovery (FLAIR) findings during his first hemiplegic migraine attack. However, magnetic resonance angiography (MRA) showed diffuse narrowing of the right middle cerebral artery. Cerebral blood flow–single-photon emission computed tomography showed right hypoperfusion. A follow-up study showed no abnormal findings. His mother had normal DWI, FLAIR, and MRA findings during her migraine attack. Both patients were diagnosed with FHM2 (p.R763H mutation in ATP1A2 ). This study highlights the importance of neurovascular examinations during the FHM2 headache phase. Further cases are required to clarify the pathophysiology of migraine.