Background: Habitual patellar dislocation in extension (HPD-E) is a distinctive subtype of recurrent patellar dislocation (RPD); HPD-E represents the most severe type of patellar maltracking in RPD. It has been reported that the presence of preoperative patellar maltracking is associated with a worse clinical outcome after medial patellofemoral ligament (MPFL) reconstruction (MPFL-R). Purpose: To describe the radiological characteristics of HPD-E and to compare clinical outcomes after MPFL-R among patients with and without preoperative HPD-E. Study Design: Cohort study; Level of evidence, 3. Methods: From January 2012 to December 2015, a total of 230 consecutive patients (246 knees) with RPD were treated with MPFL-R alone or combined with tibial tubercle osteotomy. Among them, 28 patients diagnosed with HPD-E by preoperative 3-dimensional computed tomography (CT; HPD-E group) were matched in a 1:1 fashion to 28 control participants who did not show HPD-E (control group). Routine radiography and CT were performed to evaluate patellar height, trochlear dysplasia, tibial tubercle–trochlear groove distance, and torsional deformities. The mean patellar laxity index and lateral patellar translation assessed with stress radiography were measured preoperatively and postoperatively to quantify MPFL laxity. At minimum 2-year follow-up, patient-reported outcomes (Kujala, Lysholm, and Tegner scores), patellar maltracking, and redislocation rates were compared between the HPD-E and control groups. Results: The radiological characteristics of the HPD-E group were as follows: 89% (25/28) of patients had severe trochlear dysplasia (Dejour type B or D), and the mean femoral anteversion angle was 35.5° ± 4.7°. At the final follow-up, the HPD-E group had a significantly lower Kujala score (76.2 vs 84.5, respectively; P = .001), Lysholm score (75.4 vs 86.6, respectively; P < .001), and Tegner score (4.1 vs 5.8, respectively; P = .021) compared with the control group. The postoperative patellar laxity index (43% vs 19%, respectively; P < .001) and redislocation rate (25% vs 0%, respectively; P = .01) were significantly higher in the HPD-E group than in the control group. Conclusion: Preoperative 3-dimensional CT is a reliable method of identfying patients with HPD-E. Treatment of HPD-E by MPFL-R alone or combined with tibial tubercle osteotomy resulted in a higher redislocation rate, more severe MPFL residual laxity, and lower patient-reported outcome scores compared with patients without HPD-E who underwent MPFL-R.