Artery of Percheron (AOP) is an uncommon anatomical variation in the posterior circulation neurovasculature. It is a single artery arising from P1 segment of posterior cerebral artery (PCA) which supplies bilateral paramedian thalami and the rostral midbrain and as a result, occlusion of it leads to bilateral thalamic and mesencephalic infarction. Due to very low incidence (ranges from 0.1% to 2% of all ischemic strokes) and varied presentation, the diagnosis of AOP infarction requires strong clinical and radiological suspicion and hence, AOP infarction is often missed and patients are rarely thrombolysed. Here we discuss a patient with acute altered sensorium who recovered completely in short time following treatment with intra-venous thrombolysis for bilateral medial thalamic ischemic stroke due to AOP occlusion.