Interferon (INF)-associated retinopathy occurs in 15-64% of INF-treated patients, transforming this complication into a significant risk for visual impairment. This retinopathy has been described as an ocular complication with a variable clinical course, usually benign and asymptomatic. The most common findings are hemorrhages and cotton wool spots. Atypical ocular side effects include branch or central retinal artery occlusion, central retinal vein occlusion, anterior ischemic optic neuropathy, optic disc edema, neovascular glaucoma and vitreous hemorrhage. Some case series suggest that in most cases the clinical course of the disease is benign, asymptomatic and without long-term consequences and therefore do not recommend any specific treatment; they only recommend the discontinuation of INF in patients with severe manifestations or risk factors such as hypertension or diabetes mellitus. The case reported here presents an atypical manifestation of INF-associated retinopathy consisting of a mixed retinal vascular occlusion (arterial and venous), associated with severe occlusive inflammatory microangiopathy with extensive retinal damage by ischemia and a torpid clinical course despite suspension of treatment. These varieties of occlusive vascular events have not yet been found simultaneously in the literature and neither with an unfavorable clinical course. Although the clinical course of INF-associated retinopathy in most cases is asymptomatic, there may be complications with risk to vision, which is less common. The magnitude and severity of the consequences associated with INF therapy are to be determined in prospective further studies.