Purpose of reviewThe “Extreme Exercise Hypothesis” is characterized by a U-shaped or reverse J-shaped, dose-response curve between physical activity volumes and cardiovascular health outcomes. In this review, we summarize recent findings that may support or refute the “Extreme Exercise Hypothesis.” Furthermore, we discuss potential cardiovascular health implications of the cardiac anatomical, structural, contractility, and biomarker abnormalities that have been reported in some veteran endurance athletes.Recent findingsEmerging evidence from epidemiological studies and observations in cohorts of endurance athletes suggest that potentially adverse cardiovascular manifestations may occur following high-volume and/or high-intensity long-term exercise training, which may attenuate the health benefits of a physically active lifestyle. Accelerated coronary artery calcification, exercise-induced cardiac biomarker release, myocardial fibrosis, atrial fibrillation, and even higher risk of sudden cardiac death have been reported in athletes.SummaryThere is primarily circumstantial evidence that supports the “Extreme Exercise Hypothesis.” Subclinical and atherosclerotic coronary artery disease (CAD) as well as structural cardiovascular abnormalities and arrhythmias are present in some of the most active veteran endurance athletes and need appropriate clinical follow-up to reduce the risk for adverse cardiovascular outcomes. Future studies are warranted to establish the long-term cardiovascular health effects of these findings in veteran endurance athletes.