Unobstructed binocular vision is required during the critical period of vision development to obtain optimal visual acuity in each eye and binocular stereopsis. In this article, we report 18-year follow-up of a full-term, otherwise healthy infant noted to have dense premacular hemorrhage occluding the visual axis in the left eye on retinal imaging performed 48 hours after birth. Serial examinations by the retina service were performed weekly for 10 weeks as the hemorrhage resolved spontaneously. Shortly thereafter, visual acuity revealed fixation was present, but the mother noted intermittent left eye esodeviation. At 90 days of life, the infant was seen by pediatric ophthalmology and started on 1 to 2 hours patching of the right eye daily for the esotropia, which was maintained through 24 months. At 18 years of age, the patient had orthophoria alignment, no spectacle correction, vision of 20/20 in the right eye and 20/25 in the left eye, and normal binocularity and stereopsis. Intermittent esodeviation on the left eye was observed when the patient was fatigued. Early identification of a visual axis occlusion led to prompt referral for the esotropia and initiation of patching therapy. This patient ultimately achieved a very favorable visual functional outcome that may not have been possible without early detection and management. This case report describes the longest-term published follow-up of a neonatal macular hemorrhage. [Ophthalmic Surg Lasers Imaging Retina 2024;55:XX-XX.].