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Background and Aims: Ocular Ischemic Syndrome (OIS) is a rare form of ischemia of both segments of the eye. Itu2019s caused by ocular hypoperfusion due to stenosis or occlusion of the carotid arteries mainly due to atherosclerosis. OIS as the first manifestation of bilateral carotid occlusion is rare.Method: Case-reportResults: A 63-year-old man with history of smoking, excessive alcohol intake, dyslipidemia and polyglobulia, had progressive loss of vision for one month and bilateral periorbital pain in decubitus associated with conjunctival hyperemia for ten days. On the admission day, there was a short-lasting episode of altered state of consciousness associated with involuntary movements of the upper limbs, which had also occurred two days earlier. Neurological examination revealed a bilateral hypovision. Ophthalmologic examination showed a greatly reduced visual acuity in both eyes, medium mydriasis, rubeosis iridis, retinal microaneurysms and cotton-wool spots. CT showed two watershed infarcts in the right frontal and left parieto-occipital location. CTA revealed occlusion of both common and internal carotid arteries. Doppler ultrassound confirmed severe atheromatous disease and showed normal flow in external carotid arteries and inverted flow in ophthalmic arteries. At discharge, the patient maintained hypovision with no other focal deficits. Patient was treated with dual antiplatelet therapy, high-dose statin and general recommendations for vascular risk factor control and hypotension prevention were made.Conclusion: Our patient presented an OIS as the first manifestation of bilateral occlusion of the carotid arteries. Itu2019s important to alert that ocular manifestations may precede those of the cerebrovascular system, and thus, potentially life-threatening events can be prevented.