Popliteal artery entrapment (PAE) is an uncommon syndrome. Although this phenomenon can cause claudication in younger populations, other clinical features may vary. The present report describes a case of a 48-year-old woman who was referred for treatment of a pulsatile mass in the right popliteal fossa. Computed tomography scan and magnetic resonance imaging demonstrated popliteal aneurysm associated with PAE. Resection of abnormal musculotendinous structures, aneurysmectomy and subsequent revascularization were successfully performed. Although PAE may lead to poststenotic dilatation and aneurysm formation, huge aneurysm without clinical signs of ischemia in a patient with PAE is extremely rare.Popliteal artery entrapment (PAE) is an uncommon syndrome. Although this phenomenon can cause claudication in younger populations, other clinical features may vary. The present report describes a case of a 48-year-old woman who was referred for treatment of a pulsatile mass in the right popliteal fossa. Computed tomography scan and magnetic resonance imaging demonstrated popliteal aneurysm associated with PAE. Resection of abnormal musculotendinous structures, aneurysmectomy and subsequent revascularization were successfully performed. Although PAE may lead to poststenotic dilatation and aneurysm formation, huge aneurysm without clinical signs of ischemia in a patient with PAE is extremely rare.