Patients with May‐Thurner syndrome (MTS) are at elevated risk of developing an extensive left iliofemoral deep vein thrombosis (DVT; localized blood clot) due to an anatomical variant where the right common iliac artery compresses the left common iliac vein against the lumbar spine. While MTS was initially presumed to be rare when it was first anatomically defined in 1957, case reports of this syndrome have recently become more frequent, perhaps due to improved imaging techniques allowing for enhanced visualization of the iliac veins. Still, the population burden of this condition is unknown, and there is speculation it may be higher than generally perceived. In the present review, we (a) review history of how MTS became recognized, (b) describe practical challenges of studying MTS in population‐based settings due to the specialized imaging required for diagnosis, (c) discuss why the contribution of MTS to DVT may be underestimated, (d) describe uncertainty regarding the degree of venous compression which leads to DVT, and (e) outline future research needs. Our goal is to raise awareness of MTS and spark additional research into the epidemiology of this condition, which may be an underappreciated causative venous thromboembolism risk factor.