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INTRODUCTION: Premature peripheral artery disease (PAD), defined as occurring before or at the age of 50, is a poorly studied subset of PAD due to its low incidence. It has been associated with a higher risk of progression to chronic limb-threatening ischemia (CLTI), major adverse limb events and mortality. Etiology is multifactorial, with genetics and environmental causes at play, with the most common risk factors being smoking, diabetes, and hypertension. METHODS: A single-center retrospective study was conducted in a Portuguese tertiary center, including all patients up to the age of 50 submitted to a revascularization procedure for CLTI from May 2011 to November 2023. The primary endpoint was a composite outcome of mortality, amputation and/or reintervention. The secondary endpoints were reintervention, amputation and mortality rates, and amputation-free survival (AFS). RESULTS: Ninety-one patients were included (74% male, median age 48). The most common risk factors were smoking (69%), diabetes (45%) and hypertension (44%). Most patients presented as Fontaine grade IV (79%). Thirty-two patients (35%) had aortoiliac lesions, while 38 (42%) had femoropopliteal, and 21 (23%) had infra-popliteal disease. During the median follow-up period of 7.4 years (IQR 4.3-10.3), 57 patients (63%) underwent reintervention, amputation, or died. The reintervention rate was 40%, while amputation was performed on 25% of patients, and 29% of patients died. Median AFS was 4.7 years (IQR 0.8-7.8). Subgroup analysis comparing diabetic and non-diabetic patients and endovascular vs. open surgery were performed. The 30-day amputation rate was significantly higher in diabetics (12% vs. 2%, p=.05), but there were no other significant differences in subgroup analysis. CONCLUSION: Premature CTLI is associated with poor outcomes and high mortality rates, with most patients undergoing reintervention or amputation. Further studies are needed to identify non-traditional risk factors to improve outcomes in this young population.