Introduction: Patients with hypertrophic cardiomyopathy (HCM) are excluded from high intensity activities due to perceived fear of sudden cardiac death. Observational data from athletes with HCM suggest that engaging in high intensity exercise (HIE) may be safe and is associated with higher cardiorespiratory fitness. Whether HIE can safely elicit a superior increase in fitness compared to moderate intensity exercise in patients with HCM is unclear. Methods: Nine HCM patients (49 ± 7 years, 3 female) were assessed for maximal oxygen uptake (VO 2 max, Douglas Bag method), cardiac output (Q c , acetylene rebreathing), and peripheral oxygen extraction (av-O 2 diff, Fick equation) before randomization and after 5 months of MIE or HIE training. Patients completed 3-4 sessions of MIE each week, while the HIE group also incorporated 1-2 supervised high intensity interval training sessions/week from month 3 onwards. Arrhythmias were monitored via pre-existing implantable cardiac defibrillators or implantable loop recorders placed prior to training. Results: Five months of MIE increased absolute VO 2 max by 3% and relative VO 2 max by 4%, while HIE consistently increased absolute VO 2 max by 6% and relative VO 2 max by 5% (Figure). Maximal Q c did not change after MIE but increased in all HIE patients (+1.2L/min, 95% CI -1.4 to 3.9), while maximal av-O 2 diff remained stable in both groups. Training compliance was 84 ± 15% in HIE and 93 ± 11% in MIE. There were no serious exercise-related adverse events in either group though two HIE subjects had arrhythmias at rest: 1) 14-beat run of wide complex tachycardia of uncertain mechanism given underlying conduction disease prior to a training session, and 2) 11 beats of non-sustained ventricular tachycardia prior to post exercise testing. Conclusions: Preliminary findings show that five months of HIE safely and consistently increased cardiorespiratory fitness in patients with HCM, though overall the improvements were comparable to MIE.