Objective To evaluate the effectiveness of total knee arthroplasty (TKA) using three-dimensional (3D) printing technology for knee osteoarthritis (KOA) accompanied with extra-articular deformity. Methods Between March 2013 and December 2015, 15 patients (18 knees) with extra-articular deformity and KOA underwent TKA. There were 6 males (6 knees) and 9 females (12 knees), aged 55-70 years (mean, 60.2 years). The mean disease duration was 10.8 years (range, 7-15 years). The unilateral knee was involved in 12 cases and bilateral knees in 3 cases. The clinical score was 57.44±1.06 and the functional score was 60.88±1.26 of Knee Society Score (KSS). The range of motion of the knee joint was (72.22±0.18)°. The deviation of mechanical axis of lower limb was (18.89±0.92)° preoperatively. There were 8 cases (10 knees) with extra-articular femoral deformity, 5 cases (5 knees) with extra-articular tibial deformity, and 2 cases (3 knees) with extra-articular femoral and tibial deformities. Bone models and the navigation templates were printed and the operation plans were designed using 3D printing technology. The right knee joint prostheses were chosen. Results The operation time was 65-100 minutes (mean, 75.6 minutes). The bleeding volume was 50-150 mL (mean, 90.2 mL). There was no poor incision healing, infection, or deep venous thrombosis after operation. All patients were followed up 12- 30 months (mean, 22 months). Prostheses were located in the right place, and no sign of loosening or subsidence was observed by X-ray examination. At last follow-up, the deviation of mechanical axis of lower limb was (2.00±0.29)°, showing significant difference when compared with preoperative one (t=13.120, P=0.007). The KSS clinical score was 87.50±0.88 and function score was 81.94±1.41, showing significant differences when compared with preoperative ones (t=27.553, P=0.000; t=35.551, P=0.000). The range of motion of knee was (101.94±1.42)°, showing significant difference when compared with preoperative one (t=31.633, P=0.000). Conclusion For KOA accompanied with extra-articular deformity, TKA using 3D printing technology has advantages such as individualized treatment, reducing the difficulty of operation, and achieving the satisfactory function.