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Atlantoaxial rotatory dislocation (AARD) is an uncommon disorder of childhood in which clinical diagnosis is generally difficult and often made late. It is very rare in adults because of the unique biomechanical features of the atlantoaxial articulation. We report a case of post-traumatic AARD in an adult. Reduction was difficult to obtain by skull traction and gentle manipulation. Therefore, the patient was treated surgically by an open reduction, transpedicular screw fixation, and posterior C1-2 wiring with graft. The normal atlantoaxial relation was restored with disappearance of torticollis. Postoperatively, the patient remains neurologically intact and has radiographic documentation of fusion. Atlantoaxial transpedicular screw fixation can be one of the treatment options for the AARD.