Introduction: Bosworth fracture-dislocation is an unusual variant of ankle joint fracture and dislocation, which has a high clinically missed diagnosis rate due to poor visibility on X-ray. At the same time, successful closed manipulations in an ankle joint fracture and dislocation are difficult because of the fibula attachment at the posterolateral ridge of the tibia or at the fractured end of the posterior tibia.Patient concerns: A 56-year-old man visited the hospital for further evaluation of a swollen, deformed right ankle resulting from a tumble 4 hours ago. There were no obvious complications such as skin damage or blood vessel and nerve damage at the time of treatment.Diagnosis: The patient was diagnosed with Bosworth fracture-dislocation according to clinical history and X-ray examination and computed tomography (CT), which indicated posterolateral talus dislocation; the distal end of the proximal fibular fragment was inserted behind the tibia.Interventions and outcomes: The patient initially failed to receive twice manipulative reductions. After the first attempt, roentgenograms and CT scan still showed a dislocated ankle and the proximal end of the fibula fracture was still inserted at the fractured end of the posterior tibia although the sound of the joint mounted into the acetabulum was heard. Then the inferior tibiofibula joint was fully exposed and locked with an inferior tibiofibula screw. Review roentgenograms and CT suggested that the ankle was manipulated successfully, and the broken end achieved perfect alignment, leading to a satisfactory function restoration.Lessons: Early diagnosis and reduction of Bosworth fracture-dislocation is recommended, which can significantly decrease complications. The characteristic clinical manifestations of Bosworth fracture-dislocation are worthy of attention, including extreme external rotation of the ankle and difficulty in reduction. Simultaneously, the axilla sign on mortise roentgenograms, posterior dislocation of the talus on the lateral roentgenogram and fibular position relative to the talus on the external oblique roentgenogram are intrinsically valuable. During the surgery, the floating position can better expose the broken fracture end and allow standard lateral roentgenograms. For the reduction of Bosworth fracture-dislocation, it is important to avoid repeated attempts at closed reduction. Intraoperative reduction of the fibula is the key.