To report a patient with non-arteritic anterior ischemic optic neuropathy with submacular fluid and peripapillary swelling.

A 53-year-old patient visited our clinic complaining of acute visual loss in the right eye which began two days previously. The patient had no clinical history of ocular pain on eye movement. A relative afferent pupillary defect was observed in the right eye. Fundoscopic examination showed optic disc edema and an elevated macular lesion (1 disc diameter). Fluorescein angiography (FAG) revealed blocking by serous detachment in the peripapillary area in the early phase and peripapillary leakage in the late phase. Spectral domain optical coherence tomography (SD-OCT) showed submacular fluid and peripapillary swelling. Intravenous steroid injection was administrated. After five days of treatment, visual acuity improved to 0.2; one month later, visual acuity was 0.5. subretinal fluid absorption was observed with remaining lipid deposits.

In non-arteritic anterior ischemic optic neuropathy, submacular fluid accumulation can occur due to destruction of retinal glial cells. Accumulation of submacular fluid can induce sudden visual loss and may predict visual prognosis. The macular area requires careful evaluation in non-arteritic anterior ischemic optic neuropathy patients.