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A 69-year-old Chinese woman with underlying perinuclear anti-neutrophil cytoplasmic antibody associated with vasculitis (p-ANCA vasculitis) with no previous history of ocular involvement experienced one week of blurred vision in both eyes associated with pain. Ophthalmological evaluation demonstrated severe visual loss in both eyes, with pale optic discs but without other signs of ocular vasculitis. Magnetic resonance imaging (MRI) of the brain and orbit revealed bilateral enhancement of the optic nerve sheath with classical tram-track and doughnut signs. Intravenous methylprednisolone was given for five days and marked improvement of vision was seen. In patients with p-ANCA vasculitis, bilateral optic perineuritis (OPN) is uncommon but can be one of the treatable causes to be considered, with good response to prompt steroid treatment.