Abstract
Single coronary ostium and intramural coronary artery variations in patients with transposition of the great arteries significantly increase the mortality and morbidity after arterial switch operation (ASO). In these patients, the classic coronary button implantation may cause kinking or twisting of the coronary artery which can cause coronary insufficiency. This case series presents two patients, a 15-month-old girl with transposition of the great arteries and a 10-month-old boy with a Taussig–Bing anomaly. Both underwent ASO using the aortocoronary flap technique for coronary button transfer. The coronary augmentation with the right subclavian artery technique compromises the circulation to the arm. Meanwhile, the graft reconstruction needs a tunnel/tube, thus causing risk for thrombosis. Hence, aortocoronary flap technique was preferred in this case report. Postoperative evaluations showed no ST-segment changes and no regional wall motion abnormalities. Intensive care unit (ICU) stay after ASO was 21 days and 14 days, and postoperative length of stay was 26 days and 17 days, respectively. Aortocoronary flap technique is a promising method for coronary button implantation in patient with single ostium and intramural coronary artery to prevent coronary insufficiency due to twisting and kinking of the coronary artery.
Keywords: Aortocoronary flap, arterial switch operation, intramural coronary artery, single coronary ostium, transposition of the great arteries
Introduction
Single coronary ostium and intramural coronary artery variations in patients with transposition of the great arteries (TGA) are the key factors associated with increased mortality risk in arterial switch operation (ASO). These variations pose unique challenges for surgeons during the coronary button transfer, a critical step in the procedure. The classic method of coronary button implantation, which involves mobilizing, rotating, and reattaching the coronary arteries, may not be suitable for these patients. The irregular positioning may increase the chances of kinking and stenosis, which can lead to coronary insufficiency. Therefore, surgeons must be aware, using precise and specialized techniques to avoid this complication.1–3 The aortocoronary flap technique which includes moving the coronary artery with minimal rotation and covering the flap with Contegra patch were used in this case reports. This case report was written and reported according to Surgical Case Report (SCARE) guideline. 4
Case presentation
The first patient is a 15-month-old female, weighted 6 kg, presenting with cyanosis since birth. She had undergone prior cardiac surgery, which are right Blalock–Taussig–Thomas (R-BTT) shunt and pulmonary artery banding (PAB). On physical examination, she exhibited hypoxemia with an oxygen saturation of 80%. Echocardiography revealed transposition of TGA and atrial septal defect (ASD), with adequate left ventricular mass index (LVMI) and posterior wall thickness (LVPWd) for switch. There is no specific significant allergy and drugs. She was subsequently scheduled for surgery 11 days after completing left ventricular training. The cardiopulmonary bypass (CPB) and aortic cross-clamp (AoX) time were 138 and 90 minutes. Following the removal of the aortic cross-clamp, normal sinus rhythm was restored. After surgery, the patient stayed in the ICU for 21 days, and discharged after 26 days.
The second patient is a 10-month-old male, weighted 5.6 kg, diagnosed with Taussig–Bing anomaly and pulmonary hypertension. He initially presented with cyanosis with an oxygen saturation ranging from 30% to 50% and failure to thrive. Chest radiography demonstrated cardiomegaly and pulmonary edema. Echocardiographic findings revealed that both aorta and PA arose from the right ventricle, accompanied by malposed great arteries and large subpulmonic ventricular septal defect (VSD) with a bidirectional shunt. Cardiac catheterization confirmed the echocardiographic findings, along with low flow, high resistance pulmonary hypertension. There is no specific significant allergy and drugs. The patient was urgently scheduled for surgery. The CPB and AoX time was 172 and 120 minutes. After surgery, the patient stayed in the ICU for 14 days, and discharged after 17 days.
Both patients presented with side-by-side positioning of the ascending aorta and PA, with intraoperative findings revealing a single ostium and intramural coronary artery. An ASO with the LeCompte maneuver was performed. The aortocoronary flap technique was employed for coronary button implantation to the neo-aorta in both patients. The implantation technique involves moving the coronary ostium facing outward and upward, without twisting it. Then, a Contegra patch was used to cover the roof. Surgeon must consider and calculate the anatomy thoroughly, so blood flow from neo-aorta can easily flow through the coronary artery. Step-by-step surgery and intraoperative findings are depicted in Figures 1 and 2, respectively.
Figure 1.
Step-by-step technique of the surgery.
Figure 2.
Intraoperative finding of coronary button implantation to NeoAo with aortocoronary flap technique.
Postoperatively, both showed normal sinus rhythm, no ST-segment changes, and no regional wall motion abnormalities (RWMA). A follow-up examination conducted within two weeks after discharge revealed no complications observed. Both patients were admitted to Cipto Mangunkusumo National General Hospital, Indonesia at January and November 2023. Both parents feel very delighted with the surgery conducted. Written informed consents for treatment and this study were obtained from the parents of both patients.
Discussion
The first patient underwent a rapid two-stage ASO. Adequate LVMI and LVPWd were achieved, as confirmed by echocardiographic findings. Studies show that within 1 to 2 weeks after PAB or BTT-shunt, adequate ventricular mass is typically attained. 5 The second patient had a Taussig–Bing anomaly, for which a definitive ASO was required. 6
The classic method for coronary implantation is challenging to perform in patients with single coronary ostium and intramural coronary artery variations, as it often involves moving and rotating the coronary artery, which can compromise the coronary blood flow. To address these challenges, alternative techniques such as the aortocoronary flap, coronary augmentation with the right subclavian artery, and tube graft reconstruction have been suggested for coronary transfer. Among these, the aortocoronary flap technique stands out and was used in this case series without significantly increasing CPB or AoX times, and with favorable postoperative results. This technique includes dissecting out as a single, wide coronary button to avoid damage to the coronary artery as an intramural trajectory. The button is moved to the target with no or minimal rotation. The coronary button is then sutured to the anterior zone of the neo-Aorta, and the flap is covered with Contegra patch.1–3
The aortocoronary flap technique is preferred for several reasons. Unlike the coronary augmentation with the right subclavian artery, it does not compromise circulation to the arm. Additionally, unlike the tube graft reconstruction, the flap does not create any tubular structures, which reduces the risk of thrombosis. This approach maintains coronary patency as well as minimizes the likelihood of complications, creating a safe and effective option for coronary transfer in patients with complex coronary anatomy.7–9
In these cases, postoperative evaluation of both patients with ECG showed no ST-segment changes and echocardiography showed no RMWA. Those patients were discharged after 26 days and 17 days after surgery, respectively.
The case report provides valuable clinical insight into managing complex coronary anatomy, particularly in the context of single coronary ostium and intramural coronary artery variations. It offers guidance for cardiac surgeons on how to approach similar cases and improve patient outcomes. Additionally, the report offers a detailed description of the aortocoronary flap technique, enhancing its educational value for surgeons.
Despite its strengths, this case series is limited by its small sample size. This limits the generalizability of the findings. Additionally, the report focuses mainly on immediate postoperative outcomes and lacks long-term follow-up data, which prevents an assessment of potential late complications, such as long-term graft patency and coronary function.
Conclusion
The aortocoronary flap technique is highlighted as a promising method for coronary button implantation in patients with single ostium and intramural coronary artery variations. This approach can minimize the twisting of coronary artery and prevent coronary insufficiency. By addressing these issues, the technique ensures better surgical outcome. The simplicity and effectiveness of this method offers surgeons a viable alternative to traditional method.
Footnotes
ORCID iDs: Dhama Shinta Susanti https://orcid.org/0009-0005-4977-4335
Matthew Billy https://orcid.org/0000-0002-8000-4751
Informed consent statement: Written informed consent was obtained from the parents of the participants for both participation in the study and the publication of patient information.
Author contributions: DSS contributes to organizing team and writing manuscript; CMT contributes to data collection and writing manuscript. MB contributes to data collection, writing manuscript, and creating step-by-step illustration.
DSS did study design, data analysis, performing the surgery, and writing the manuscript, and validating the manuscript's credibility.
CMP and MB did study design, data collecting, writing the manuscript, reviewing, and editing the manuscript.
Funding: The authors received no financial support for the research, authorship, and/or publication of this article.
Declaration of conflicting interest: The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Data availability statement: The data supporting the findings of this study are included within the article. Additional details or data are available from the corresponding author upon reasonable request.
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