Trigeminal trophic syndrome is a rare condition resulting from self-manipulation of the skin after a peripheral or central injury to the trigeminal system. The syndrome consists of a classic triad of anesthesia, paresthesias, and secondary persistent or recurrent facial ulcerations. The most common causes include destruction of the trigeminal ganglion, rhizotomy, and stroke. We describe a patient who developed the syndrome as a sequel to brainstem infarction and trigeminal neuropathy. Whereas a-lipoic acid and gabapentin were ineffective, a remarkable benefit was achieved by administering carbamazepine (200 mg 3 times a day), which influences both neuropathic and behavioral factors in this rare syndrome. Our experience with the presented case, together with the scarce information in the literature, indicates that carbamazepine should be the first treatment option for trigeminal trophic syndrome.