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Persistent median artery (PMA) thrombosis is a rare cause of carpal tunnel syndrome (CTS) with only a few cases reported in the literature. The bifid median nerve (BMN) is often associated with PMA and may be a factor in the development of CTS. There is a paucity of information in the literature on the management options for CTS secondary to PMA thrombosis. This paper presents two cases of CTS with associated PMA thrombosis and BMN and offers a discussion on diagnostic and management options.