Inherited eye diseases, particularly degenerative retinal dystrophies, lead to blindness in early childhood and cause lifelong restrictions for patients. Gene therapy approaches with adeno-associated viral vectors have shown promising results to treat certain RPE65-related eye diseases in rodents and other animal models. Luxturna is the first adeno-associated viral gene therapy, which was approved in 2017 to treat patients with the RPE65 mutation. AAV vectors have effective gene expressions with low levels of toxicity. However, lentiviral vectors have a greater gene expressions than AAV vectors with higher transduction capacities, which make them alternative vehicles in gene therapy. In this work, which was part of the Matet et al. 2017 study, the tolerance and efficacy of a lentiviral vector were evaluated in five non-human primates. The study was conducted with subretinal injections of LV-RPE65 at low and high doses with vehicle injections. Multiple OCT scans and fundus images were performed at different time points to evaluate retinal morphological changes after injections. Additionally, retinal layer thickness measurements were performed using a segmentation program (DiOCTA) in two areas: the central fovea and an area of interest within the injected site. Retinal alterations were observed in both vehicle and LV-RPE65 injected eyes. The fundus images revealed discrete pigmentary changes as a result of the retinal detachment and reattachment in vehicle and LV-RPE65 injected animals. In most eyes, the thinning of the retinal layers, especially the outer nuclear and photoreceptor layers, was apparent at the level of the fovea. The results of this work show that the LV vector was well tolerated at the low dose in LV-RPE65 injected eyes, considering the fact that no adjuvant anti-inflammatory treatments were applied. The morphology of the retinal layers was preserved most in the eyes which received the lower dose of LV-RPE65. The irreversible retinal changes that occurred in eyes injected with the high dose of the LV vector are assumed to be due to the liability of the foveal area in the case of retinal injuries. Additional experiments on a larger scale will be required to determine the safe dose of the vector and to improve the subretinal delivery route in order to increase and maintain the tolerance of the vector. The study affirms that LV vectors could be potential alternative vehicles in gene therapies, not only for RPE65-associated retinal disorders, but also for other congenital retinal dystrophies