Numbness and ulceration of the face, particularly erosion of ala of the nose, sometimes occur after sensory denervation in the territory of the divisions of the trigeminal nerve. The incidence is uncertain and usually follows surgical treatments for trigeminal neuralgia. Such condition is known as trigeminal trophic syndrome (TTS), although some authors believe it to be a special form of dermatitis artefacta. Trigeminal trophic syndrome most commonly affects adults, after iatrogenic, vascular, viral, or neoplastic damage to the trigeminal nerve. We present a rare case of TTS in a 32-year-old woman who was referred to us with progressive numbness in the right upper and lower lip region.