The artery of Percheron (AOP) is a single dominant thalamo-perforating artery that supplies bilaterally the medial thalami with variable contribution to the rostral midbrain. Occlusion of the AOP causes indeed variable and unspecific clinical symptoms due to this complex anatomy, and very often this diagnosis is delayed with the impossibility of recurring to intravenous thrombolysis (rTPA). Here, we report a case of AOP stroke that received a prompt diagnosis and therapy, owing to the availability of MR brain scan, showing a DWI/FLAIR mismatch typical of hyperacute infarctions. This case points out the importance of a high level of suspicion of AOP stroke, together with the correct implementation of imaging studies.