Dislocation of the mandibular condyle into the middle cranial fossa is extremely rare, It is often misdiagnosed initially because of its rarity and nonspecific symptoms, Accurate diagnosis is the key to effective management which can also keep patient from unnecessary treatment The authors presented a case of a 19-year-old man with dislocated right condyle entering the middle cranial fossa, The mandible was "ankylosed" in an ipsilateral lateral excursion, and the diagnosis was confirmed by plain radiography and computed tomography, Conservative treatment was tried initially but failed, Therefore open reduction with manipulation followed by repair of dura and glenoid fossa was performed successfully without intracranial complications, The occlusion was stable and no limitation of opening mouth during postoperative follow-up, Due to its rarity, the etiology, diagnosis, possible treatment plan from closed reduction to open reduction were reviewed, and hopefully it could be a reference for future management.