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Ocular signs are unusual in the presentation of cranial dural arteriovenous fistulas in locations other than the cavernous sinus. Between 2000–2015, 13 patients met the inclusion criteria for this retrospective analysis. The most common signs were chemosis (61.5%), loss of visual acuity (38.5%), exophthalmia (38.5%), and ocular hypertension (7.7%). Dural arteriovenous fistulas presenting with ocular signs were classified into 4 types due to their pathologic mechanism (local venous reflux into the superior ophthalmic vein, massive venous engorgement of the cerebrum responsible for intracranial hypertension, compression of an oculomotor nerve by a venous dilation, or intraorbital fistula with drainage into the superior ophthalmic vein).