Tentorial dural arteriovenous fistula (DAVF) as a cause of trigeminal neuralgia is extremely rare. Although less than 10 cases have been reported in the literature, all cases presented with ipsilateral trigeminal neuralgia. Here we report a unique case of tentorial DAVF manifesting as contralateral trigeminal neuralgia. A 58-year-old man presented with right-sided trigeminal neuralgia. Cerebral angiography revealed a left tentorial DAVF and the MR imaging demonstrated a variceal venous dilatation occupying the left lateral pontine cistern and multiple venous flow voids adjacent to the right trigeminal nerve root entry zone. Transarterial Onyx embolization resulted in near complete obliteration of the fistula with immediate resolution of facial pain. The patient remains symptom free and without evidence of recurrence during 9 months of follow-up. Transarterial Onyx embolization may be an effective treatment modality for such an usual case.
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