Vascular pythiosis, a vascular infectious disease in hemoglobinopathy patients, caused by Pythium insidiosum, has an endemic area in tropical and subtropical countries. According to literature review, suprainguinal vascular pythiosis leads to 100% of mortality. The authors report a 35-year-old thalassemic patient who presented with a right inflammatory pulsatile groin mass and right limb ischemia. The computerized tomography angiography indicated a false aneurysm at the right external iliac artery and thrombosed entire right leg arteries. The management comprised antifungal agent, immunotherapy, and surgical removal of all infected arteries (high up to the right common iliac artery and above-knee amputation). The patient was found in a good condition at 36 months after the follow-up period.
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