BACKGROUND: Patients with hypertrophic cardiomyopathy (HCM) are generally restricted from exercising. We aimed to investigate the effects of exercise on cardiorespiratory fitness, cardiac structure, cardiac function, and clinical safety for patients with HCM. METHODS: We performed a systematic review and meta-analysis by searching PubMed, Web of Science, Scopus, Embase, and CENTRAL up to April 2025. Dichotomous outcomes were pooled using risk ratios (RRs), while continuous outcomes were pooled using mean differences (MDs) or standardized mean differences (SMDs), all with 95% confidence intervals (CIs), using R version 4.3.1. RESULTS: With the inclusion of seven studies-three randomized controlled trials and four observational studies-our cohort comprised 9702 patients. Compared to the control group, exercise was significantly associated with increased cardiorespiratory fitness (peak oxygen consumption [peak VO2] (mL∙kg-1∙min-1) (MD: 1.78; 95% CI [0.98, 2.59]; P < 0.01) and VO2 at an anaerobic threshold (mL∙kg-1∙min-1) (MD: 1.27; 95% CI [0.34, 2.20]; P < 0.01), reduced body mass index (kg/m2) (MD: -0.67; 95% CI [-1.17, -0.18]; P < 0.01), reduced left ventricular wall thickness (mm) measured by echocardiography (MD: -0.56; 95% CI [-1.01, -0.11]; P = 0.01), increased quality of life physical factor (SMD: 0.66; 95% CI [0.34, 0.97]; P < 0.01), and decreased incidence of all-cause mortality (RR: 0.71; 95% CI [0.60, 0.85]; P < 0.01). CONCLUSION: This study suggests that structured exercise interventions are safe and associated with improvements in cardiorespiratory fitness, left ventricular wall thickness, body mass index, and quality of life physical factor in patients with HCM, without increasing the risk of arrhythmias. Although a lower mortality rate was observed, this finding was mainly driven by observational studies and should therefore be interpreted with caution. Larger randomized controlled trials are needed to confirm these findings.