Purpose: To alert ophthalmologists to the recognition of cortical visual loss as the presenting feature in patients with reversible posterior leukoencephalopathy syndrome (RPLES). Unique radiologic findings are paramount to the diagnosis. Design: Interventional case report. Methods: A patient was seen with perioperative bilateral cerebral visual loss that was misinterpreted initially as an irreversible ischemic event. Further detailed analysis of the radiologic findings and clinical history led to the correct diagnosis. Main Outcome Measures: Visual acuity and magnetic resonance imaging (MRI) of the brain. Results: Recognition of the correct diagnosis of RPLES led to the institution of antihypertensive therapy and recovery of normal vision. Conclusions: The diagnosis of RPLES should be considered in all patients with acute cerebral visual loss, especially in the setting of recent surgery, blood transfusion, chemotherapy, immunosuppressant use, hypertension, eclampsia, or seizures. Prompt diagnosis requires close collaboration with a radiologist and an emergent MRI study, which ideally should include diffusion- weighted imaging with calculation of an apparent diffusion coefficient map. Differentiation from acute cerebral ischemia is important in order to avoid permanent visual loss by prompt and vigorous treatment of exacerbating factors such as intermittent hypertension. Prompt diagnosis will also help to avoid potentially dangerous invasive procedures such as thrombolytic therapy. Purpose: To alert ophthalmologists to the recognition of cortical visual loss as the presenting feature in patients with reversible posterior leukoencephalopathy syndrome (RPLES). Unique radiologic findings are paramount to the diagnosis. Design: Interventional case report. Methods: A patient was seen with perioperative bilateral cerebral visual loss that was misinterpreted initially as an irreversible ischemic event. Further detailed analysis of the radiologic findings and clinical history led to the correct diagnosis. Main Outcome Measures: Visual acuity and magnetic resonance imaging (MRI) of the brain. Results: Recognition of the correct diagnosis of RPLES led to the institution of antihypertensive therapy and recovery of normal vision. Conclusions: The diagnosis of RPLES should be considered in all patients with acute cerebral visual loss, especially in the setting of recent surgery, blood transfusion, chemotherapy, immunosuppressant use, hypertension, eclampsia, or seizures. Prom pt diagnosis requires close collaboration with a radiologist and an emergent MRI study, which ideally should include diffusion-weighted imaging with calculation of an apparent diffusion coefficient map. Differentiation from acute cerebral ischemia is important in order to avoid permanent visual loss by prompt and vigorous treatment of exacerbating factors such as intermittent hypertension. Prompt diagnosis will also help to avoid potentially dangerous invasive procedures such as thrombolytic therapy.