A rare case of spontaneous carotid-cavernous fistula with contralateral clinical signs (ocular discomfort, pulsating exophthalmos, chemosis, ptosis) is reported. It was associated with partial thrombosis of the ipsilateral cavernous sinus. It was cured with conservative treatment after six months. Anatomical knowledge of the cavernous sinus remains essential for better understanding of this original clinical presentation.
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