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The impact of reverse left ventricular remodeling (r-LVR) on clinical outcomes after surgical correction of anomalous left coronary artery from the pulmonary artery (ALCAPA) remains unclear. This study aims to examine the prognostic significance of r-LVR in patients with ALCAPA after surgery. We prospectively identified 61 patients undergoing surgical correction for ALCAPA; 54 patients had adequate echocardiographic image quality with quantitative biplane analysis performed both at baseline and at 30-day postoperative follow-up. Postoperative r-LVR was defined as a reduction of ≥ 10% in left ventricular end-diastolic volume index during follow-up. Cox proportional-hazards regression was used to investigate the independent association of r-LVR and all-cause mortality. Among 54 patients (age: 21.2 ± 7 months; 37% females), r-LVR occurred in 35 patients (64.8%) after surgery. Compared to patients with r-LVR, patients without r-LVR had significantly higher level of N-terminal pro B-type natriuretic peptide (NT-proBNP) [2176 (711, 4219) vs 998 (623, 2145) P < 0.001] and lower survival rate (47.3% vs 82.9%, HR = 5.72 [1.96 to 17.20], P < 0.001) at 1-year follow-up. NT-proBNP (OR = 2.27 [1.67 to 18.3], P = 0.02) was an independent predictor of r-LVR in multivariate analysis. Moreover, r-LVR was significantly associated with a lower rate of all-cause mortality (HR = 0.27 [0.08 to 0.98], P = 0.03) in multivariate analysis, even after adjustment for clinical and echocardiographic variables. R-LVR occurred in more than half of patients with ALCAPA undergoing surgical correction and it was associated with better clinical outcomes. NT-proBNP is an independent predictor of r-LVR. [ABSTRACT FROM AUTHOR]