High-altitude retinopathy (HAR) is a retinal disorder caused by prolonged exposure to hypoxic environments at high altitudes, posing a significant threat to visual health and potentially leading to severe vision impairment. There have been limited references of HAR, with observations of its clinical manifestations and prognosis remaining insufficient. Some reports suggest that HAR may spontaneously resolve without eliciting any noticeable symptoms. This case focuses on the disease progression and follow-up results of a typical HAR patient, and a comprehensive ophthalmic examination was conducted, encompassing not only fundus examination and optic coherence tomography (OCT), but also fundus fluorescein angiography, Humphrey visual field examination, and electroretinogram.A 41-year-old healthy Chinese man ascended from an altitude of 143 ft (43.5 m) to 13,780 ft (4200 m) rapidly. After living locally for 4 months, he developed decreased vision accompanied by visual field defects in his left eye.Ophthalmic examination revealed bilateral papilledema, peripapillary hemorrhage in his left eye, and retinal hemorrhage in the macular area of optic papilla. He was diagnosed with HAR.Peribulbar injection of triamcinolone acetonide in the left eye, along with oral ginkgo biloba tablets to improve microcirculation, as well as mecobalamin tablets and vitamin B1 tablets for neural nutrition.After 4 months, the patient's visual field defect decreased, and retinal hemorrhage was absorbed.This case comprehensively reveals the clinical features of HAR, and highlights that HAR can lead to permanent visual function impairment if the individual fails to promptly leave the pathogenic environment.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.