Purpose: Non-arteritic anterior ischemic optic neuropathy (NA-AION) is typically a disorder of patients 50 years and older predisposed to the vascular and structural optic disc risk factors. We present an interesting case of sequential development of NA-AION in a 45-year-old patient with end-stage renal disease undergoing hemodialysis. Methods: Observational case report. Results: A 45-year-old female on hemodialysis for chronic renal failure complained of sequential acute onset sudden painless gross diminution of vision in right eye followed by the left eye. At presentation, fundus examination revealed secondary optic atrophy and a pallid disc edema with few hemorrhage in the right and left eye, respectively. Ambulatory blood pressure recorded nocturnal dips of diastolic blood pressure. Fundus fluorescein angiography showed hypoperfusion of the left optic disc, confirming the diagnosis of NA-AION. She was treated with oral steroids, but with no improvement. Conclusion: Both treating nephrologist and ophthalmologist should be aware of this uncommon but potentially blinding complication, to permit its early recognition and prevent the occurrence in the fellow eye. Also, care should be taken to prevent and treat any hypotensive episodes during and following dialysis therapy in such high-risk patients.