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To report a case of a neonate with acute retinal necrosis, lens vacuoles, and encephalitis associated with herpes simplex virus (HSV) infection. Case report. Retrospective chart review. A male neonate was brought for screening for retinopathy of prematurity at the corrected age of 32 weeks. Slit-lamp examination showed lens vacuoles in both eyes. Fundus examination revealed extensive retinal detachments with prominent retinal whitening, subretinal exudates, and retinal hemorrhage. Computed tomography of the brain showed encephalomalacia. Polymerase chain reaction of cerebrospinal fluid and anterior chamber fluid was both positive for HSV-1. Despite systemic anti-viral therapy, a rhegmatogenous retinal detachment and subsequent proliferative vitreoretinopathy developed in the patient’s right eye. The retinal detachment in the left eye resolved, but significant chorioretinal degeneration occurred. With time lens vacuoles decreased in number. Clinicians should remember this rare, but devastating condition without specific prodromal symptoms.

Multimodal Imaging of a Severe Case of Neonatal Acute Retinal Necrosis and Lens Vacuoles Associated with Herpes Simplex Virus Infection