Purpose: To report a case of an infant born at 30 weeks gestational age (GA) who, at 37 weeks GA, presented with bilateral acute retinal necrosis (ARN) syndrome and herpes simplex virus (HSV) encephalomalacia.
Methods: Observational case report.
Results: A premature infant was found to have ARN based on dilated funduscopic examination and positive HSV serologies. Herpes simplex virus encephalomalacia was diagnosed base on magnetic resonance imaging (MRI).
Conclusion: To our knowledge, this is the youngest reported patient with ARN. This case demonstrates that neonatal ARN may present with posterior chorioretinal lesions and highlights the importance of considering HSV infection of the central nervous system with MRI findings of cystic encephalomalacia.