Migrainous infarction (MI) is a rare complication of migraines that accounts for 0.5–1.5% of all ischemic strokes. Although the pathogenesis of MI is still debated, cortical spreading depression and the consequent biochemical cascade and hemodynamic changes are presumed to play an important role. Here we describe a case of MI and systematically review the literature on the complex and possibly bidirectional relationship between migraine and stroke. A 44-year-old female with history of migraine with visual aura presented at the Emergency Department due to a sudden onset of left limb paresis and hypoesthesia. Brain magnetic resonance imaging revealed right fronto-parietal ischemic stroke. Two days after hospitalization, the patient experienced a prolonged visual aura and showed ultrasound evidence of intracranial artery vasospasm. To date, there have been 33 published articles on a total 119 patients with MI, although intracranial vasospasm has rarely been reported. Sustained hyperexcitability of cortical neurons, impairment of γ-aminobutyric acid inhibitory circuitry, altered serotonergic transmission, release of vasoconstrictive molecules, and cerebral blood flow changes have been proposed as pathogenic mechanisms of MI. The present case provides insight into the pathophysiological link between stroke and migraine, thus aiding clinicians in therapeutic decision-making although additional studies are needed to clarify the clinical, neuroradiological, and ultrasound findings that link MI and stroke-related migraine.