Congenital brachial artery true aneurysms are exceedingly rare. Most are pseudoaneurysms secondary to trauma or infection. We report a boy aged 2 years who presented with painless, pulsatile swelling on the medial aspect of the right arm, 4 cm above the elbow joint that had been present since birth. Spiral CT angiography showed a fusiform aneurysm of the distal right brachial artery with a peripheral crescent-shaped thrombus. Distal arteries were normally opacified. There was no evidence of abnormal dilation or stenosis in any other artery. The aneurysm was surgically resected, with vascularisation re-established using a reversed great saphenous vein graft. His postoperative course was uneventful. Early surgery should be performed for moderately sized to large aneurysms that recently increased in size, exhibited luminal thrombus formation or caused neurovascular distal limb compromise. Early surgery could prevent complications such as a ruptured aneurysm, thromboembolism or limb ischaemia or loss.
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