OBJECTIVE We aimed to report a novel case of hyperacute computed tomography (CT) perfusion imaging abnormalities in artery of Percheron infarction. METHODS We described a case of a 74-year-old man who presented acutely to the emergency room with witnessed sudden onset altered level of consciousness. RESULTS Although initial hyperacute CT brain imaging was reported as normal, subsequent magnetic resonance imaging revealed bilateral paramedian thalamic infarction. A retrospective review of CT perfusion imaging revealed perfusion mismatch in the bilateral thalami, which was not recognized in the hyperacute setting. CONCLUSIONS Artery of Percheron occlusion is a well-described cause of bilateral paramedian thalamic infarction, but to date, we have not identified any reports of perfusion imaging abnormalities in the hyperacute setting. This case emphasizes the important role of perfusion imaging when clinical presentation is not typical for ischemic stroke, but is suspected, given an acute onset; its recognition may have led to the consideration of thrombolysis acutely in this case (stroke chameleon).