Aim: To describe a case of ophthalmic artery occlusion associated with anisocoria and revealing a cavernous sinus thrombosis due to sinusitis.

Observation:   A 48-year-old man with a history of diabetes presented acutely with loss of vision and proptosis in the left eye. Ophthalmologic examination concluded in a left ophthalmic artery occlusion with anisocoria and total ophthalmoplgia. Cardiac assessment was normal. Magnetic resonance imaging (MRI) revealed left ophthalmic artery and internal carotid occlusions, left cavernous and transverse sinus thrombosis and sphenoid sinusitis. The patient underwent extensive haematological and medical assessment to search for embolic sources and disease causing thrombophilia. The patient recovered from the thrombosis episode, but sustained permanent blindness.

Conclusion: Ophthalmic manifestations may be the only signs revealing cavernous sinus thrombosis which must be usually suspected. Visual prognostic was very poor.

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