Reverse amblyopia following gene therapy-induced visual improvement in a child with retinal pigment epithelium-65 inherited retinal dystrophy.

Voretigene neparvovec can improve visual function in children with RPE65-associated inherited retinal dystrophy. However, the effect of rapid asymmetric visual improvement on binocular development and amblyopia remains uncertain.A young child underwent sequential treatment 8 months apart. After right-eye treatment, visual acuity improved, fixation switched and a manifest left esotropia developed. Left-eye visual acuity subsequently deteriorated. We describe this as reverse amblyopia in a novel context: reversal of the interocular amblyopic pattern following treatment-induced visual improvement without fellow-eye penalisation. Despite left-eye treatment, refractive correction and attempted occlusion, the marked visual asymmetry persisted. This case highlights a potential binocular effect of rapid unilateral visual improvement and the need for close monitoring between sequential treatments.