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Voretigene neparvovec (VN), developed for the treatment of inherited retinal dystrophy associated with confirmed biallelic RPE65 mutations (RPE65-IRD), is the first approved retinal gene therapy. Recently, reports have emerged of chorioretinal atrophy (CRA) developing in a subset of patients treated with VN. Although researchers have started to investigate the etiology, detection, classification and clinical course of CRA following treatment with VN, information gaps remain. Here we review current data on CRA in patients treated with VN, propose standardized terminology for describing CRA, and provide guidance on the management of CRA in patients treated with VN.