| Summary: | Dilation throughout the brachial artery in the setting of an arteriovenous fistula is a common occurrence, but focal aneurysmal dilation is not often visualized. Progressive enlargement of a focal arterial segment warrants intervention before negative sequelae. We present the case of a 38-year-old man with history of left upper extremity brachiocephalic fistula who had an enlarged brachial artery and progressive aneurysmal dilation of the distal aspect after ligation and excision of a dilated venous outflow component. The patient was successfully treated with resection and end-to-end reconstruction of the brachial artery, with resolution of pain and improvement in the functionality of his extremity. This case highlights the possible challenges encountered in such situations, when the anatomy is so distorted that it is difficult to clearly delineate on preoperative imaging. Keywords: Arteriovenous fistula, Aneurysmal dilatation, Arteriomegaly, Venous outflow
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