Abstract Background Treatment of atlantoaxial dislocation is aimed at reduction and stabilization of the atlantoaxial joint. 3D printing refers to a process where additive manufacturing is achieved under precise computer control. Literature on its utilization in anterior atlantoaxial fixation and fusion is rare. This study is the first report on a 3D-printed locking cage used in the anterior procedure for atlantoaxial dislocation. Methods A middle-aged male in his 40s presented with weakness and numbness of his extremities for 3 years and could only walk slowly with assistance. Imaging studies revealed severe anterior migration of C1, irreducible atlantoaxial dislocation, and severe cervical-medullary compression. A preoperative plan consisting of trans-oral soft tissue release and fixation using tailor-designed 3D-printed cages was devised. Following fluoroscopic confirmation of reduction of the atlantoaxial joints, two customized 3D-printed cages made of titanium alloy were inserted into the bilateral facet joints, which were then locked by six screws into the lateral masses of C1 and C2. The microstructure of the inserted cages was optimized for improved biomechanical stability and enhanced osseo-integration, without the need for bone grafting. In addition, a biomechanical test was performed on seven human cadaveric specimens comparing the novel implant with the conventional C1 lateral mass-C2 pedicle screw construct in three modes of motion (flexion-extension, lateral bending, axial rotation). Results Improvement of neurologic function in the patient was evident immediately after surgery. He was able to walk independently 1 month post-operatively. At the 12-month follow-up, coronal reconstruction of CT demonstrated properly-positioned 3D-printed cages, evidence of osseo-integration at the bone-implant interface, and no subsidence or displacement of the implant. Eighteen months out of surgery, the mJOA score improved to 15, and lateral X-ray confirmed reduction of atlanto-axial dislocation. Additionally, the new construct provided strong fixation comparable to that conferred by conventional constructs as there was no significant difference observed between the two groups in all three directions of motion. Conclusions The novel implant represents a new option in the treatment of irreducible atlantoaxial dislocation. It can provide strong anterior support for solid fixation and fusion with a low profile and a microstructure that obviates the need for bone grafting.