Purpose: To report a case of optic neuropathy and retinopathy from a dose of radiation traditionally thought to be safe to the visual system and discuss strategies for preventing vision loss when using radiation to treat intraocular tumors. Observations: A 44-year-old woman presented with new, painless vision loss in the left eye eighteen months after receiving proton beam radiotherapy (20 Gy dose delivered in two 10 Gy fractions) for a uveal metastasis of lung cancer. The dilated funduscopic examination revealed optic disc swelling and retinal hemorrhages and an MRI of the brain and orbits demonstrated enhancement of the left optic nerve head, findings consistent with radiation optic neuropathy (RON) and retinopathy. Risk factors for developing RON included coincident use of oral chemotherapy and relatively large fractionated doses of radiation. Conclusions and importance: Though cumulative radiation doses to the anterior visual pathway of less than 50 Gy are traditionally felt to be safe, it is important to consider not just the total exposure but also the size of individual fractions. The single-dose threshold for RON in proton beam treatment has yet to be defined. Our case suggests that fractions of less than 10 Gy should be delivered to minimize the risk of optic nerve injury.