Development of a Novel Risk Prediction Model for Sudden Cardiac Death in Childhood Hypertrophic Cardiomyopathy (HCM Risk-Kids).
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Norrish Gabrielle, Ding Tao, Field Ella, Ziólkowska Lidia, Olivotto Iacopo, Limongelli Giuseppe, Anastasakis Aristides, Weintraub Robert, Biagini Elena, Ragni Luca, Prendiville Terence, Duignan Sophie, McLeod Karen, Ilina Maria, Fernández Adrián, Bökenkamp Regina, Baban Anwar, Kubuš Peter, Daubeney Piers E F, Sarquella-Brugada Georgia, Cesar Sergi, Marrone Chiara, Bhole Vinay, Medrano Constancio, Uzun Orhan, Brown Elspeth, Gran Ferran, Castro Francisco J, Stuart Graham, Vignati Gabriele, Barriales-Villa Roberto, Guereta Luis G, Adwani Satish, Linter Katie, Bharucha Tara, Garcia-Pavia Pablo, Rasmussen Torsten B, Calcagnino Margherita M, Jones Caroline B, De Wilde Hans, Toru-Kubo J, Felice Tiziana, Mogensen Jens, Mathur Sujeev, Reinhardt Zdenka, O'Mahony Constantinos, Elliott Perry M, Omar Rumana Z, Kaski Juan P
Аннотация
OBJECTIVE: To develop and validate an SCD risk prediction model that provides individualized risk estimates.
DESIGN, SETTING, AND PARTICIPANTS: A prognostic model was developed from a retrospective, multicenter, longitudinal cohort study of 1024 consecutively evaluated patients aged 16 years or younger with HCM. The study was conducted from January 1, 1970, to December 31, 2017.
EXPOSURES: The model was developed using preselected predictor variables (unexplained syncope, maximal left-ventricular wall thickness, left atrial diameter, left-ventricular outflow tract gradient, and nonsustained ventricular tachycardia) identified from the literature and internally validated using bootstrapping.
MAIN OUTCOMES AND MEASURES: A composite outcome of SCD or an equivalent event (aborted cardiac arrest, appropriate implantable cardioverter defibrillator therapy, or sustained ventricular tachycardia associated with hemodynamic compromise).
RESULTS: Of the 1024 patients included in the study, 699 were boys (68.3%); mean (interquartile range [IQR]) age was 11 (7-14) years. Over a median follow-up of 5.3 years (IQR, 2.6-8.3; total patient years, 5984), 89 patients (8.7%) died suddenly or had an equivalent event (annual event rate, 1.49; 95% CI, 1.15-1.92). The pediatric model was developed using preselected variables to predict the risk of SCD. The model's ability to predict risk at 5 years was validated; the C statistic was 0.69 (95% CI, 0.66-0.72), and the calibration slope was 0.98 (95% CI, 0.59-1.38). For every 10 implantable cardioverter defibrillators implanted in patients with 6% or more of a 5-year SCD risk, 1 patient may potentially be saved from SCD at 5 years.
CONCLUSIONS AND RELEVANCE: This new, validated risk stratification model for SCD in childhood HCM may provide individualized estimates of risk at 5 years using readily obtained clinical risk factors. External validation studies are required to demonstrate the accuracy of this model's predictions in diverse patient populations.