Vertical one-and-a-half syndrome (VOHS) is a rare ophthalmoplegia syndrome caused by unilateral thalamo-mesencephalic infarct. It is manifested as a conjugate upgaze palsy and a monocular paresis of downward gaze. Recognition of VOHS helps clinical localization. No cases of VOHS with photos or videos of ocular motility have been reported in China. Herein a case of VOHS caused by unilateral thalamo-mesencephalic infarct is reported, in order to improve awareness about the ocular sign.