Percheron syndrome or synchronous bilateral thalamic infarction is a rare entity with a difficult clinical diagnosis. The complex structure of the thalamus in nuclei, as well as its peculiar irrigation and individual anatomical variability means that ischemic lesions can occur bilaterally in both thalamus due to the obstruction of a single artery, characterizing this picture. We present a case with acute ischemic lesions in both thalamus and anterior midbrain, compatible with the obstruction of the Percheron artery.