Trigeminal trophic syndrome (TTS) is a rare facial/cranial affection that arises in ulcerations, itch and paresthesia. Etiology is debated, however trigeminal nerve damage seems to be frequent in pathogenetic patterns. The disease may affect any region innervated by the trigeminal nerve, especially the maxillary branch. A case of TTS, trigged by allergic reaction to osteosynthetic materials and involving infraorbital nerve, was presented. The feature that makes this case one-off in the literature is the association with osteolytic lesion surrounding infraorbital nerve. Diagnosis and treatment were difficult and multidisciplinary approach was required. Treatments administered were satisfying and signs and symptoms remitted, however patient quitted follow-up. TTS is a rare disease, diagnosis is difficult to be performed and it is often a diagnosis of exclusion. Treatment is challenging and it requires a multidisciplinary approach and a great compliance of patients.