Anterior tibial plateau fracture is fairly common. This study was aimed at introducing a type of severe anterior tibial plateau fracture (anterior tibial plateau fracture-dislocation) and evaluating its clinical characteristics and treatment strategies. In this study, 18 patients with severe anterior tibial plateau fracture (study group) were enrolled between November 2006 and August 2014, and their data were compared with those of 21 patients treated for normal Schatzker type VI tibial plateau fracture (control group) between January 2010 and August 2014. At the last follow-up, bony union had been achieved in both groups. The incidence of ligament injury was higher in the case of anterior tibial plateau fracture than control group. The average range of motion in the study group was 0.56 to 109 degrees, while that in the control group was 1.81 to 117 degrees. The average Hospital for Special Surgery scores and Lysholm scores in the study group were significantly lower than those in the control group. Further, the incidence of postoperative complications and reduction loss were higher for anterior tibial plateau fracture cases than for normal Schatzker type VI fracture. Our findings also showed a significantly higher rate (22.2%) of popliteal artery injury in the study group than in the control group. Anterior tibial plateau fracture-dislocation is a special type of Schatzker type VI fracture with very low incidence and most commonly characterized by the anterior subsidence of the tibial component, irreducible dislocation of the knee joint, and varying degrees of neurovascular and knee-joint peripheral ligaments injuries, as well as high incidence of complications during fixation surgery. The treatment of anterior tibial plateau fracture-dislocation is challenging even for experienced surgeons.