Posterior reversible encephalopathy syndrome (PRES) is a reversible clinical and neuroradiological syndrome which may appear at any age and characterized by headache, altered consciousness, seizures, and cortical blindness. The exact incidence is still unknown. The most commonly identified causes include hypertensive encephalopathy, eclampsia, and some cytotoxic drugs. Vasogenic edema related subcortical white matter lesions, hyperintense on T2A and FLAIR sequences, in a relatively symmetrical pattern especially in the occipital and parietal lobes can be detected on cranial MR imaging. These findings tend to resolve partially or completely with early diagnosis and appropriate treatment. Here in, we present a rare case of unilateral PRES developed following the treatment with pazopanib, a testicular tumor vascular endothelial growth factor (VEGF) inhibitory agent.