Introduction: Temporomandibular Joint Ankylosis (TMJA) is a pathological condition characterized by limitation of the opening of the mouth caused primary by trauma but also by infection, inflammatory and congenital diseases. TMJA can be treated through the utilization of autogenous grafts or alloplastic TMJ prostheses. There are two types of alloplastic TMJ prostheses: stock or custom. In the last years custom alloplastic TMJ prostheses are the emerging treatment modality for TMJ ankylosis. Presentation of the case: A 16-year-old male patient presented with a 1 cm interincisal opening for about 2 years and inability to chew and speak. He referred ankylosis appeared after a trauma. The ankylotic masses were of 17,6 mm on the right side and 19,6 mm on the left side. A bilateral removal of the ankylosis and a custom-made prostheses reconstruction was programmed. Clinical discussion: Treatment options for TMJ ankylosis are various. Stock alloplastic TMJ prostheses may not suit all patients due to anatomical variations. Thus, custom alloplastic TMJ prostheses have emerged as the preferred treatment modality for TMJ ankylosis. We also conducted a review of the literature regarding patients who underwent bilateral custom-made prostheses. This treatment Conclusion: Custom alloplastic TMJ prostheses are considered an optimal treatment modality for reconstructing the TMJ in adult and young adult patients with TMJ ankylosis. The implantation of custom-made prostheses is not associated with any peri- and postoperative complications.