The impact of trochlear dysplasia on patellofemoral arthroplasty (PFA) has rarely been reported in the literature. The purpose of this study was to explore the effectiveness of PFA in patients with patellofemoral osteoarthritis (PFOA) associated with trochlear dysplasia. From January 2014 to March 2018, 35 patients with PFOA and trochlear dysplasia (29 females and 6 males), underwent PFA, were included in the present retrospective study. Radiological measurements including the patellar tilt (PT), congruence angle (CA), Blackburne-Peel ratio (BPR), tibial tuberosity-trochlear groove (TT-TG) distance, and lateral trochlear inclination (LTI) were assessed pre- and postoperatively to evaluate the changes in patellofemoral alignment. The patient-reported functional outcome was assessed using the Oxford Knee Score. The mean follow-up time was 24 months (range, 18-42 months). The intraclass correlation coefficients were excellent for all measurements. The patellofemoral alignment and knee function were significantly improved postoperatively. The PT decreased from 23.3 ± 6.3 degrees preoperatively to 6.4 ± 2.3 degrees postoperatively (p < 0.001). The CA decreased from 32.3 ± 8.6 degrees preoperatively to 10.2 ± 3.6 degrees postoperatively (p < 0.001). The lateral trochlear inclination increased from 8.3 ± 4.1 degrees preoperatively to 16.0 ± 1.2 degrees postoperatively (p < 0.001). The TT-TG distance decreased from 18.2 ± 3.8 mm preoperatively to 11.5 ± 3.3 mm postoperatively (p < 0.001). The BPR did not significantly change postoperatively (p = 0.390). The average Oxford Knee Score improved from 19.5 preoperatively to 29.2 at 6 months postoperatively (p < 0.001), 37.9 at 1 year postoperatively (p < 0.001), and 39.1 at final follow-up (p < 0.001). No patient developed patellofemoral malalignment or prosthesis loosening during short-term follow-up. PFA achieved favorable therapeutic results in patients with PFOA associated with trochlear dysplasia.