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Introduction A singular procedure involving both a distal tibiofibular synostosis resection with syndesmosis repair by peroneus longus ligamentoplasty has not been reported in the English literature. We report a case of simultaneous distal tibiofibular synostosis resection and syndesmosis stabilization by peroneus longus ligamentoplasty for the treatment of symptomatic distal tibiofibular synostosis formation, following neglected syndesmosis injury. Case presentation A 42-year-old Caucasian man presented with ankle pain and painful range of motion 20 months following ankle trauma. Distal tibiofibular synostosis was identified, and our patient was successfully treated by simultaneous synostosis takedown and peroneus longus ligamentoplasty for distal tibiofibular syndesmosis repair. Conclusions Our experience illustrates that in cases of painful posttraumatic distal tibiofibular synostosis, simultaneous synostosis resection with peroneus longus ligamentoplasty may show good clinical results. Level of evidence 5