A case with unilateral acute retinal necrosis (ARN) following herpes simplex virus (HSV) encephalitis was reported. A 40-year-old man presented with unilateral visual loss. He had a history of HSV encephalitis 2 months previously, and had been successfully treated with intravenously acyclovir. The ophthalmologic examination in his left eye suggested ARN syndrome. The best-corrected visual acuity was 20/222. Magnetic resonance imaging (MRI) showed high density signals from the optic chiasm to optic disc. The patient underwent encircling scleral buckling, lensectomy, and vitrectomy with endolaser photocoagulation and silicone oil tamponade. Postoperatively, the high density signals on MRI decreased, the retina maintained attached, and the visual acuity at the final visit improved to 20/20. Unilateral ARN can develop after HSV encephalitis. Acyclovir should be administered over the long term to control the virus.
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