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A woman in her 30s was administered linezolid treatment for pneumonia. Without continuous medical prescriptions, the patient self-administered linezolid for a total of 9 months, which subsequently led to symptoms of bilateral visual impairment and central scotomas. Fundus examination revealed blurred disc margins, accompanied with mild edema, flame-shaped hemorrhages around the optic disc, and mild tortuosity and dilation of retinal veins in both eyes. Visual field testing indicated the presence of central scotomas in both eyes. Visual evoked potentials testing showed reduced amplitudes in both eyes. After excluding other causes of optic neuropathy, the patient was tentatively diagnosed with bilateral toxic optic neuropathy (linezolid-associated optic neuropathy) based on these findings. After discontinuing linezolid, the patient's visual function gradually improved. This case suggests that close monitoring of visual function and neurological symptoms in patients receiving linezolid treatment is crucial, and early identification of relevant abnormalities and timely discontinuation of the drug can effectively improve patient prognosis.