Purpose Topresenta case of hypertensive choroidopathy with an atypical presentation on a healthy young man, and the end-stage renal diseaseeventually developed. Case report A 25-year-old man without a significant previous medical history presented to our clinic with progressively worsening blurry vision in the left eye for 4 days.Dilated fundus examination revealed diffuse arteriolar narrowing, arteriovenous nicking, only a few sites of flame-shaped hemorrhage, and disc edema in both eyes. Moreover, multiple deep, round, and yellow patches at the level of retinal pigment epithelium and extensiveexudativeretinal detachment were also found. Rapid progression to renal failure developed within 2 weeks after hospitalized for systemic evaluation ofmalignant hypertension. Even though blood pressure was well controlled thereafter, it progressed to an end-stage renal disease, which required lifelong hemodialysis. Conclusions and Importance In young adults with an acute episode of hypertension, choroidopathy can be the predominant ophthalmic feature. Certain fundoscopic signs may correlate with theend-organ damage to thekidney. Timely diagnosis and immediate referral for treatment of hypertension are critical to prevent irreversible ocular and other end-organdamage.