TMJ ankylosis is classified with true and false type. A true ankylosis is defined as any condition that produces fibrous or bony adhesions between the articular surfaces of the temporomandibular joint. The main causes of true ankylosis are trauma or infection. A false ankylosis results from pathologic conditions outside the joint that limit mobility of the mandible such as myogenic disorders, coronoid impingement or rare direct bony fusion between maxilla and mandible. The treatment of choice of TMJ ankylosis is surgical intervention. We experienced the male patient with complete mouth opening limitation since 45 years before. This patient has true TMJ ankylosis and rare bony synostosis between maxilla and mandible in the right posterior region. We performed surgical intervention and had a favorable result.