Introduction: Temporomandibular joint (TMJ) ankylosis is a serious condition that affects the masticatory system and is referred as permanent movement disfunction of the mandible caused by bilateral or unilateral fibrous or bony adhesions leading to numerous consequences. The causes may be congenital or acquired and include trauma, arthritis, infection, neoplasms, previous TMJ surgery etc. Surgical treatment consists of extensive resection of the ankylotic mass and reconstruction of the ramus-condyle unit with autogenous or alloplastic grafts. A combination of surgical treatment and physiotherapy is usually needed in order to achieve maximum rehabilitation and functionality of the mandible. The purpose of this poster presentation is to present the surgical management of an unusual case of unilateral TMJ ankylosis and review the literature. Case report: A thirty-seven-year old male patient presented complaining of the inability to open his mouth since his birth. Clinical examination revealed congenital unilateral TMJ ankylosis. The lateral movement of the mandible was impossible. The patient was treated with resection of the ankylotic bone mass, replacement of the condyle with costo-chondral graft and replacement of articular disc with the temporal fascia. Since the surgery was completed the patient shows no signs of relapse. Conclusion: TMJ ankylosis can be congenital or acquired. Its treatment requires detailed medical history, clinical and radiographic examination. Surgical resection of the ankylotic bone and coronoid process is necessary and a graft that resembles to the ramus-condyle unit can be placed. Immediate postoperative kinesiotherapy and regular recalls are of great importance.