<h4>Purpose</h4>To investigate the effects of voretigene neparvovec (VN) treatment in patients with RPE65-related retinal dystrophies, with particular focus on chorioretinal atrophy (CRA) and its impact on visual function.<h4>Methods</h4>Twenty-five patients treated with VN were evaluated over a 12-month follow-up and stratified according to the presence of postoperative CRA, classified into nummular, mixed and touchdown patterns.<h4>Results</h4>Best-corrected visual acuity (BCVA) improved by a median of 11 Early Treatment Diabetic Retinopathy Study (ETDRS) letters (P < 0.001). Here, RPE65 is linked to inherited retinal dystrophy.