Carpal tunnel release is a common surgical procedure performed by hand surgeons. The procedure is typically straightforward; however, uncommon causes of median nerve compression encountered intraoperatively may add complexity. We describe a 67-year-old man with carpal tunnel syndrome and an intraoperative finding of a compressive vascular malformation during a mini-open carpal tunnel release. A space-occupying malformation of a persistent median artery was bisecting the nerve and thought to be responsible for the patient’s symptoms. The compression was relieved through extended carpal tunnel release without requiring removal of the vascular malformation. The patient’s symptoms improved postoperatively. Hand surgeons undertaking this procedure should be aware of a potential vascular malformation and be prepared to address the condition intraoperatively.

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