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Aneurysmal degeneration of the brachial artery is a rare complication of distal native fistulas. Chronic mechanical stresses due to high flow, as well as immunosuppressant drugs following renal transplantation, are the main factors implicated in the pathophysiological mechanism. We report a case of a kidney transplant patient with a true, symptomatic, brachial artery aneurysm, 8 years after transplantation, and 5 years after radiocephalic fistula ligation at the wrist. The patient underwent open surgical repair, with resection of the aneurysm and end-to-end anastomosis. We present a literature review of the different strategies for treating this unusual entity.