A 48-year-old woman with neurofibromatosis type 1 (NF1) experienced progressive forearm swelling coupled with impending compartment syndrome. Computed tomography angiography revealed a ruptured aneurysm of the proximal radial artery, multiple fusiform radial artery aneurysms, and a high independent ulnar artery origin. Compartment syndrome required prompt hematoma evacuation. Radial artery reconstruction, technically demanding due to vessel wall fragility, was deemed unnecessary because of satisfactory blood supply to the hand. Histologic findings indicated NF1-related vascular abnormalities also in the apparently normal radial artery as well as in a forearm vein, suggesting diffused vasculopathy. This case report is the first on ruptured radial artery aneurysm in NF1-related polianeurysmatic degeneration.
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