Trochlear dysplasia is a developmental condition characterised by an abnormally flat or dome shaped trochlea and is an important contributing factor to patellofemoral instability and recurrent patellar dislocation. We prospectively studied a cohort of 54 consecutive patients (59 knees) with patellofemoral instability secondary to trochlear dysplasia, treated with a trochleoplasty by a single surgeon over a 5 year period. Patients were recruited from the senior author's specialist knee clinic and pre-operatively, multiple patient-centred scores were recorded. After the trochleoplasty, these were repeated along with a patient satisfaction questionnaire. Of the 54 patients and 59 knees operated, 39 of the patients (44 knees) were female and 15 (15 knees) male and 40 patients (42 knees) had follow-up of 12 months or more. Their mean age at surgery was 21 years and 6 months and mean length of follow up 2 years. One patient was unable to attend for follow up due to the development of a pelvic Ewings sarcoma. A statistically significant improvement in outcomes was demonstrated when the pre and post-operative scores were compared (mean scores of Oxford 26 to 19, WOMAC 23 to 17, IKDC 54 to 72, Kujala 62 to 76, Lysholm 57 to 78, p values <0.001 for all scores). 93% (50 patients) were satisfied with the outcome of their procedure. We conclude that in the short and medium term, the results of trochleoplasty are encouraging in this challenging group of patients.