Background: Trigeminal trophic syndrome (TTS) is one of the rare causes of ulceration over the face, which occurs due to self-manipulation or rubbing of the facial skin after an injury to the trigeminal nerve. Case Report: We report a case of trigeminal trophic syndrome in a 60 years old lady, known case of trigeminal neuralgia, who presented with ulcer and swelling of the right side of the face. She was treated, counseled and is being followed up. Conclusion: Trigeminal trophic syndrome, though a rare complication, should be suspected as a cause of unilateral facial ulceration in a patient with trigeminal neuralgia.