Abstract Purpose Hypertrophic cardiomyopathy (HCM) is a complex cardiac disorder, that influences patients' physical capacity and quality of life, as well as the risk of sudden cardiac death (SCD). Patients with HCM often experience emotional distress due to the uncertainty of their condition and have a threatening illness perception (the way individuals make sense of their illness, which can have a significant impact on adherence to treatment). Current studies suggest that some HCM patients could benefit from hybrid cardiac telerehabilitation (HCTR) in terms of somatic and psychological status. This study is part of a research project investigating the safety and effectiveness of hybrid cardiac telerehabilitation for HCM patients. The specific focus of this part is to evaluate the changes in mental health status and illness perception of HCM patients who have been referred for cardiac telerehabilitation compared to those receiving usual care. Methods An RCT study was conducted, with the inclusion criteria for the study of being above 18 y.o., HCM without LVOT obstruction, NYHA Class II or III, LVEF ≥ 50%, and having CIED. The study involved telerehabilitation (HCTR) and control (CG) groups. Both groups were completing questionnaires assessing illness perception (Brief IPQ) and overall psychosocial distress (GHQ-28) with its 4 domains: somatic symptoms, anxiety, social impairment, and depression. Psychological assessment was performed at the beginning and at the end of telerehabilitation/outpatient care, and at 3, 6 and 12 months after the end of care (T1/T2/T3/T4/T5). Results There was a significant improvement in the overall distress level in the HCTR group (p=0.003) but not in the CG (p=0.559). It remained significant in the HCTR group for the T2 (p<0.001), T3 (p=0.008), and T4 (p<0.001). The positive change was observed in somatic symptoms only in the HCTR group: for T2 (p=0.025) and T3 (p=0.032) compared to the T1. The gradual decrease in anxiety level was observed in the HCTR group, but not in the CG, with a significant difference for T4 (p=0.013) compared to T1. Although there was an improvement in social impairment in both groups (p=0.049 for the CG, and p=0<0.001 for the HCTR group), only in the HCTR group it held a stable pattern after the intervention (p<0.001 for T2; p=0.013 for T3; p=0.032 for T4). There were no changes in the level of depressive symptoms in either of the compared groups. There were also no significant differences in the illness perception (p=0.200 for the HCTR group and p=0.090 for the CG), although the HCTR group showed short-term improvement after three months of HCTR (T2) (p=0.032). Conclusions There are significant benefits of hybrid cardiac telerehabilitation for mental health, level of distress, and illness perception in HCM patients. It is worth noticing that this improvement is short-term, and prolonged effects might require providing additional support during extended care.overall distress in HCTR vs CGsubscales of GHQ_28 in HCTR vs CG