Purpose: It is proposed to present a clinical case of a patient with a paracentral acute middle maculopathy (PAMM) as the first sign of presentation of severe internal carotid artery (ICA) dissection.Methods: An initial basic ophthalmologic examination of the patient was performed, followed by an optical coherence tomography (OCT) and an OCT‐angiography (OCT‐A) of each eye. Finally, the patient was evaluated by a neurologist, performing a cerebral and supra‐aortic trunks (SAT) computed tomography (CT) angiography.Results: We present a 54‐year‐old patient who came to the emergency room complaining of central blurred vision in his left eye (OS) during 24 h. On ophthalmological examination, it is remarkable a visual acuity (VA) of counting fingers at 1 m in OS. Eye fundus (EF) of OS showed small oval whitish macular lesions. Macular OCT of OS revealed hyperreflective bands in the internal nuclear layer (INL) and OCT‐A showed areas with low flow at deep capillary plexus (DCP) and macular vascular density (MVD) parameters decreased compared to the right eye (OD). During the examination, the patient began to feel a slight loss of strength in his right hemibody with dizziness, so he was explored by a neurologist and a cerebral and SAT CT angiography was performed, which reported severe left ICA dissection with an area of hypoperfusion in the territory of the left middle cerebral artery, but without intracranial vascular occlusion. Since there was no image of occlusion and given the mild neurological symptoms, endovascular treatment was rejected and anticoagulation with enoxaparin was established. Two months later, VA of OS improved to 1, with no significant lesions in the EF. The OCT of OS showed a slight thinning of the INL and the OCT‐A revealed a greater flow in the DCP with better MVD values.Conclusions: This entity should be considered in patients with visual disturbances. PAMM may be the first sign of ICA pathology.
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