A case of cheiro-oral syndrome presented with an extremely unusual distribution of sensory impairment involving the left intra-and peri-oral regions and tips of the lateral three fingers. A computed tomogram scan of the brain revealed a small infarction in the contralateral thalamus consistent with occlusion of the thalamogeniculate artery involving the nucleus ventralis posterior. This case gives evidence of the concept of the somatotopic organization of the human sensory thalamus.