Fig. 1: Patients with a MYBPC3 VUS identified as deleterious by STRUM (STRUM+) are associated with an increased risk for adverse hypertrophic cardiomyopathy (HCM)-related outcomes.

Selection within Sarcomeric Human Cardiomyopathy Registry (SHaRe) of patients with HCM carrying a single MYBPC3 missense variant of uncertain significance (VUS) is shown on the left. One hundred five patients carry a single MYBPC3 missense VUS, covering 77 distinct MYBPC3 VUS. Kaplan–Meier curves, median event free survival (years), and hazard ratio with corresponding 95% confidence interval (CI) reveal that patients carrying a STRUM+ MYBPC3 VUS (red) exhibited a higher rate of adverse HCM-related outcomes (overall composite) compared with patients carrying a STRUM- variant (black).