Background: Pathological fracture of the distal femur giant cell tumor (GCT) occasionally leads to articular surface defect. The conventional endoprosthesis replacement needs to remove the both condyles, and then the patients probably face lower prosthesis survival rate and multiple joint replacement, especially in young patients. The aims of this study were to evaluate the feasibility of 3D-printed custom-made prosthesis in repairing the articular surface defect at a short-term follow-up.Methods: We retrospectively reviewed fourteen patients admitted and treated from January 2012 to January 2018 with mean age of 43.9 years (range 29 to 69 years). Diagnoses were primary pathological fracture of distal femur giant cell tumor (GCT) which articular surface of one condyle is defect. 10 lesions were located in medial femoral condyle and 16 in Lateral femoral condyle. Twelve patients were treated with three-dimension-printed custom-made prosthesis to repaired of articular surface defect (3D-printed group), and the other Fourteen patients were treated with endoprosthesis replacement (EPR group). Operation time, blood loss was compared between the groups. Functional results were assessed with MSTS score. The range of motion (ROM), local recurrences and complications were statistically compared.Results: Mean Follow-up is 33.5 months (range 26 to 43 months). In the 3D-printed group, operation time and blood loss were significantly less (p < 0.05). Differences in functional outcomes of the MSTS were observed between the two groups from three-month follow-up (p < 0.05) to twenty-four-month follow-up (p < 0.05), and the score were higher in the 3D-printed group than the EPR group. The ROM of the 3D-printed group was statistically better than the EPR group at 6 and 9 months after operation (p < 0.05). Local recurrence and complication rates were similar in the two groups (p = 1).Conclusion: 3D-printed custom-made prosthesis represent a promising reconstructive technique in articular surface defect caused by pathological fracture of GCT of distal femur. It could be able to achieve satisfactory oncological and better short-term functional results compared with endoprosthesis replacement. Further studies are needed to evaluate the stability of the implants and the wear of the articular surface with larger samples at long-term.