The artery of Percheron (AOP) is an anatomical variation in the paramedian thalamic artery. With the development of imaging technologies, the AOP is attracting more and more attention. In this article, a case of AOP occlusion-induced bilateral thalamic infarction was reported; the case was characterized by progressive bilateral thalamic infarction which showed no diffusion-limited signals on early diffusion-weighted imaging with progression of the stroke and later showed radiological features consistent with the symptoms only after multiple reexaminations. Through analysis of this case and review of relevant literature, the pathogenesis, clinical manifestations, and radiological features of AOP infarction were investigated, with an expectation of improving the diagnosis and treatment of such diseases.