Trigeminal neuralgia has been rarely reported in association with dural arteriovenous malformations (DAVMs). We describe a case of trigeminal neuralgia resulting from compression of the trigeminal nerve at its root entry zone by a large tentorial DAVM. Surgical interruption of the draining vein at its exit from the tentorium resulted in complete angiographic obliteration of the fistula with immediate resolution of the facial pain. We review the literature relevant to this topic and discuss the aetiology and pathophysiology of trigeminal neuralgia as well as the management of tentorial DAVMs.