This study aims to report the ocular manifestations in a patient with atypical hemolytic uremic syndrome (aHUS), treated with ravulizumab, highlighting the reversibility of hypertensive retinal changes. We present a retrospective case report of a 27-year-old man diagnosed with aHUS, who underwent comprehensive systemic evaluation after a respiratory infection and a hypertensive crisis. Despite the absence of ophthalmologic complaints, ocular examination revealed grade IV hypertensive retinopathy accompanied by exudative retinal detachment. The patient received a combination of hemodialysis, intensive antihypertensive therapy, and ravulizumab. Remarkably, within one week of treatment initiation, a significant decrease in papilledema and a complete resolution of the exudative retinal detachment were observed. This case highlights the potential for rapid reversal of severe ocular manifestations associated with systemic microangiopathic disorders, such as aHUS. The striking improvement in retinal pathology suggests that the early and aggressive management, including complement inhibition with ravulizumab, may play a crucial role in restoring ocular integrity. Furthermore, the findings advocate for routine ophthalmologic screening in aHUS patients, even in the absence of visual symptoms, to enable prompt identification and intervention in subclinical retinal involvement. Interdisciplinary collaboration between nephrology and ophthalmology is essential to optimize both systemic and ocular outcomes in such complex cases.