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To report an unusual case of dizygotic twins, in whom one neonate developed acute retinal necrosis from herpes simplex virus (HSV) type 2.Retrospective chart review.A 29-week-old premature infant from in vitro fertilization donor egg with negative maternal and fetal history for HSV-1 and HSV-2 developed vesiculopapular rash upon birth. Fundus examination revealed vitritis with retinal hemorrhages, suggestive of retinitis; the twin, however, had a normal funduscopic examination. A presumptive diagnosis of HSV infection was made, and the patient was started on intravenous acyclovir. Polymerase chain reaction of the anterior chamber fluid was positive for HSV-2; serology of both mother and child was HSV-2 IgG(+), IgM(−). Acute retinal necrosis was managed with 360° of bilateral peripheral laser cerclage. A subsequent rhegmatogenous retinal detachment in one eye was successfully treated with a scleral buckle. Retinal reattachment, macular pigmentary disturbances, and mild optic atrophy were noted at 6 months of follow-up.The authors report a case of isolated congenital acute retinal necrosis in an otherwise healthy neonate, and product of a dichorionic, diamniotic in vitro fertilization pregnancy. Clinicians should consider possible HSV transmission from in vitro fertilization donor egg in the differential diagnosis, despite the negative fetal and maternal histories for HSV infection.