Carpal tunnel syndrome (CTS) is the most common type of entrapment neuropathy. The majority of CTS cases are idiopathic and affect females between 40 and 60 years old. Conversely, this report describes two female patients in their mid-30's diagnosed with CTS caused by a median artery in the carpal tunnel using ultrasonography. We visualized the median artery which emerged from the radial artery and common interosseous artery in the proximal forearm of each patient by magnetic resonance angiography (MRA) before surgery. After the vertical incision of the transverse carpal ligament, the anomalous vessel was encountered, which ran over the median nerve at the radial aspect, and a simple mini-open procedure was performed for carpal tunnel release. Postoperatively, the CTS symptoms were relieved in both patients. The purpose of this report is to describe the persistent median artery using MRA in two patients and to report on their postoperative mini-open carpal tunnel release outcomes.