Pterygoid hamular bursitis is an infrequent cause of orofacial pain due to a hypertrophic pterygoid hamulus. The pain is often referred to the pharynx, temporomandibular region and facial zone, and requires a differential diagnosis with other craniofacial entities. This paper describes a patient with pterygoid hamular bursitis that presented pain of neuropathic characteristics in the left retromolar region, associated with odynophagia and temporomandibular joint disorders. Based on the clinical and radiological findings, a surgical resection of the pterygoid hamulus was decided. After the surgical procedure the patient still reported symptoms so additional specific treatments such as peripheral nerve block and infiltration were performed. Four months later, the patient developed a squamous cell carcinoma on the left margin of the tongue, which was surgically treated. At present (thirty months follow-up), the patient has no pain or signs of tumor relapse.