Patient characteristics

A total of 111 patients were classified into five groups based on the type of definitive operations: the atrial switch operation (n = 20), ASO (n = 12), Nikaidoh (n = 7), Rastelli (n = 48), and REV operations (n = 24). The ASO group had the youngest mean operative age of 0.3 years and the Rastelli group had the oldest one of 5.3 years. Forty-six balloon atrial septostomy and 61 palliative operations were performed before the definitive operation. The category of LVOTO was valvular in 34 patients (31%), subvalvular in 30 (27%), atresia in 15 (14%), and subvalvular/valvular in 32 (29%). The most common type of LVOTO was small pulmonary diameter with valvular stenosis and left deviation of the infundibular septum with subvalvular stenosis. Preoperative median Z-scores of pulmonary diameter in valvular LVOTO for atrial switch operation, ASO, Nikaidoh operation, Rastelli operation, and REV operation groups were − 0.20 (range, − 0.4–0.6), 0.73 (range, − 0.5–1.7), − 0.64 (range, − 0.3–− 1.3), − 3.84 (range, − 14.0–− 1.2), and − 1.99 (range, − 6.4 to − 0.4), respectively. Preoperative left ventricular shortening fraction was not notably different between the groups. Preoperative patient data were summarized in Table 1.

Table 1 Patient characteristics

Surgical technique

In atrial switch group, Senning (n = 18) or Mustard (n = 2) technique was used. In ASO group, the neo-PA was reconstructed by the original method (n = 2) or the Lecompte maneuver (n = 10), and the VSD was enlarged for intraventricular baffle in 5 TGA-type DORV patients. In Nikaidoh group, the RVOT was reconstructed using an autologous pericardial transannular patch. In Rastelli group, the RVOT was reconstructed with a xenograft roll conduit bearing handmade xenograft pericardial valves (1980–1996; n = 34), an autologous pericardial roll conduit bearing handmade autologous pericardial valves (1992–2012; n = 11), or an expanded polytetrafluoroethylene (e-PTFE) conduit with bulging sinuses and fan-shaped valves (2012–2017; n = 3) [7]. The conal flap method [8] was used in 20 patients with structural abnormalities of the tricuspid valve and the VSD was enlarged in 34 patients in this group. In REV group, the RVOT was reconstructed with a xenograft pericardial patch bearing a handmade xenograft pericardial valve (1980–1986; n = 6) or an autologous pericardial patch bearing a handmade autologous pericardial valve (1987–1999; n = 18). The mean perfusion and aortic cross-clamp times were longer in ASO (248 and 120 min) and Nikaidoh groups (242 and 125 min) and shortest in atrial switch group (138 and 79 min). In all groups, no patients were converted to other types of procedures. Operative results were summarized in Table 2.

Table 2 Operative results

Overall survival

During the median follow-up of 18.2 (7.3–25.0) years, 74 patients were alive, 28 died, and 9 were lost to follow-up. There were 8 early mortalities (1 in atrial switch operation group [5%], 1 in ASO group [8%], 2 in Nikaidoh group [29%], and 4 in Rastelli group [8%]), which were all recorded in the 1980s or 1990s. Extracorporeal membrane oxygenation was introduced in four of these patients. There was no early mortality in REV group. The most frequent cause of these early mortalities was low output syndrome. The causes of death are shown in Table 3.

Table 3 The causes of death

Survival at 20 years after definitive operation was 78.5 ± 9.6% in atrial switch group, 75.0 ± 12.5% in ASO group, 42.9 ± 18.7% in Nikaidoh group, 75.5 ± 6.5% in Rastelli group, and 76.7 ± 10.8% in REV group (Fig. 2a). The deaths within three years after definitive operation were notable in ASO and Nikaidoh groups, however, there were no long-term mortalities in these groups. Mortalities more than10 years after definitive operation were recorded in the Rastelli and REV groups. The most frequent cause of late death was sudden/unknown reason, followed by infection/sepsis.

Fig. 2
figure 2

Kaplan–Meier survival curve. Kaplan–Meier survival curve is shown in the atrial switch operation (red), ASO (green), Nikaidoh (blue), Rastelli (orange), and REV (purple) groups. ASO, arterial switch operation; REV, Réparation à l’Etage Ventriculaire. a Survival. Survival at 20 years after definitive operation was 78.5 ± 9.6% in atrial switch operation group, 75.0 ± 12.5% in ASO group, 42.9 ± 18.7% in Nikaidoh group, 75.5 ± 6.5% (67.6 ± 7.9% at 30 years) in Rastelli group, and 76.7 ± 10.8% in REV group. b Freedom from reoperation. Freedom from reoperation at 20 years after definitive operation was 87.7 ± 8.3% (76.7 ± 12.6% at 30 years) in atrial switch operation group, 61.1 ± 15.7% in ASO group, 100% in Nikaidoh group, 32.1 ± 8.4% in Rastelli group (24.5 ± 8.4% at 30 years), and 85.2 ± 8.0% (42.6 ± 30.4% at 25 years) in REV group. c Freedom from RVOT-related reoperation. Freedom from RVOT-related reoperation at 20 years after definitive operation was 33.1 ± 8.9% in Rastelli group (28.4 ± 8.6% at 30 years), 90.2 ± 6.7% in REV group (42.6 ± 30.4% at 25 years), and 100% in the other groups. d. Freedom from LVOT-reoperation. Freedom from LVOT-related reoperation at 20 years after surgery was 76.4 ± 15.5% in ASO group, 76.6 ± 10.1% in Rastelli group (57.5 ± 13.9% at 30 years), and 100% in the other groups

Reintervention

Twenty-five patients (24%) required postoperative catheter intervention: 2 in atrial switch, 5 in ASO, 1 in Nikaidoh, 14 in Rastelli, and 3 in REV. The most common catheter intervention for each group was pacemaker implantation in atrial switch operation group (2/2: 100%), percutaneous RVOT dilatation in Rastelli group (8/14: 57%), and percutaneous distal PA angioplasty in ASO (5/5: 100%), Nikaidoh (1/1: 100%), and REV (2/3: 67%) groups. Catheter ablation was performed for supraventricular tachycardia in 3 patients in Rastelli group.

A total of 40 reoperations were conducted on 36 patients. Freedom from reoperation at 20 years after definitive operation was 87.7 ± 8.3% in atrial switch operation group, 61.1 ± 15.7% in ASO group, 100% in Nikaidoh group, 32.1 ± 8.4% in Rastelli group, and 85.2 ± 8.0% in REV group (Fig. 2b).

Thirty RVOT-related reoperations were conducted on 23 patients in Rastelli group (3 patients required twice) and 4 in REV group. In Rastelli group, conduit stenosis was repaired by conduit replacement (n = 20) or direct right ventricle-to-PA anastomosis with patch angioplasty (n = 2), and infectious endocarditis was treated by bioprosthetic pulmonary valve replacement (n = 1). In REV group, RVOT was reconstructed with an autologous pericardial patch bearing a handmade autologous pericardial valve (n = 4). Overall, freedom from RVOT-related reoperation at 20 years after definitive operation was 33.1 ± 8.9% in Rastelli group, 90.2 ± 6.7% in REV group, and 100% in other groups (Fig. 2c). A xenograft pericardial conduit/patch for RVOT reconstruction had a higher chance to require RVOT-related reoperation than an autologous pericardial conduit/patch in Rastelli and REV groups (62% vs. 18% in Rastelli group, and 50% vs. 6% in REV group).

Ten LVOT-related reoperations were conducted in 10 patients: 3 in ASO group and 7 in Rastelli group. Aortic valve replacement for neo-aortic regurgitation was required for 3 patients in ASO group. Seven LVOT-related reoperations in Rastelli group comprised 5 cases of re-intraventricular rerouting for intraventricular route stenosis and 2 of aortic valve replacement for aortic regurgitation. Of these 7 reoperations, 5 were performed concomitantly with RVOT-related reoperations. Conversely, there were no LVOT-related reoperations in REV group. Freedom from LVOT-related reoperation at 20 years after definitive operation was 76.4 ± 15.5% in ASO group, 76.6 ± 10.1% in Rastelli group, and 100% in other groups (Fig. 2d).

Other types of reoperations included 2 mitral valvuloplasties (1 in ASO and 1 in REV), 1 residual VSD closure with concomitant tricuspid annuloplasty (ASO), and 3 tricuspid valve replacement for systemic atrioventricular valve regurgitation (atrial switch operation).

Echocardiographic data and clinical condition

Late echocardiography data were obtained from 73 of 83 survivors (88%). The median Z-score of valve diameter in the systemic ventricular outflow tract in atrial switch operation, ASO, Rastelli and REV groups was 2.0 (1.4–2.6), 1.7 (1.3–1.9), 0.2 (0.2–0.4), 2.5 (1.4–3.6), and 2.5 (1.4–3.6) respectively and the median flow velocity in the systemic ventricular outflow tract was 1.3 (1.1–1.4), 2.0 (1.6–2.1), 1.0 (0.9–1.1), 1.3 (0.9–2.0), and 1.3 (0.9–1.7) m/s respectively. A high incidence of substantial neo-aortic regurgitation was found in ASO group (2/7: 29%). A proportion of the substantial pulmonary regurgitation was comparable among Nikaidoh, Rastelli, and REV groups (67% vs. 58% vs. 63%). The median systemic ventricular shortening fraction in atrial switch operation, ASO, Nikaidoh operation, Rastelli operation, and REV operation groups was 0.20 (0.17–0.22), 0.37 (0.31–0.38), 0.31 (0.26–0.33), 0.28 (0.24–0.34), and 0.27 (0.26–0.29), respectively. No patients had Class III or IV on the New York Heart Association functional classification.

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