The authors report a case of trigeminal trophic syndrome (TTS) that occurred as a complication of a neurosurgical procedure. Three years after a second surgical treatment for a meningioma of the cerebellopontine angle, this 32-year-old woman developed TTS with progressive skin ulcers on the left ala nasi and the left side of the forehead and chin. Trigeminal trophic syndrome is an extremely rare cause of facial ulceration. It occurs as a consequence of trigeminal nerve damage or impaired central sensory connections. To the authors' knowledge, this is the first report of lesions in the dermatomes of all three branches of the nerve after a neurosurgical procedure. Early recognition of this disorder is important, as treatment is difficult and often unsatisfactory. Many clinicians are not aware of this disease, thus, it may be more common than previously thought. The importance of recognizing and diagnosing TTS, as well as its treatment, are discussed.