Anomalous Aortic Origin of a Coronary Artery: Results from a Single Surgical Team in Spain

Abstract

Objectives

Anomalous aortic origin of a coronary artery is a rare congenital lesion in which a coronary artery arises from an anomalous location within the aorta. Anomalous aortic origin of a coronary artery has been associated with myocardial ischemia and it is considered the second most common cause of sudden cardiac arrest in young athletes. When surgical repair is indicated, surgical unroofing is the most commonly employed technique. Our objective is to describe the outcomes of our surgically treated patients.

Methods

We present a series of 16 adult patients who underwent surgical repair of anomalous aortic origin of a coronary artery. Patients were treated in three different institutions by the same surgeon. Surgical unroofing of the anomalous coronary artery was the surgical technique chosen in the majority of the patients. Follow-up was performed.

Results

Unroofing of an intramural anomalous coronary artery was the procedure performed in 11 patients. Three patients underwent neo-ostium creation; one patient underwent a David procedure with coronary reimplantation; and one patient was treated with coronary bypass grafting due to severe coronary atheromatous lesions. There were no perioperative deaths, and no major postoperative complications. Follow-up period was 73.8 months, the survival rate was 100%, and there were neither ischemia or heart failure reports.

Conclusions

The surgical repair of anomalous aortic origin of a coronary artery by coronary unroofing or neo-ostium creation has demonstrated excellent early and late outcomes. Late survival was excellent. The follow-up period revealed no significant morbidity or complications.

Keywords

anomalous aortic origin of a coronary artery surgical coronary repair long-term outcomes

Introduction

Anomalous aortic origin of a coronary artery (AAOCA) is a rare congenital coronary anomaly described first in the Renaissance period, but it was not considered a clinically significant issue until the mid-1970s.¹ Anomalous aortic origin of a coronary artery occurs in approximately 0.2% to 1.33% of the general population² and it is defined as a congenital malformation in which a coronary artery arises from a place in the aorta other than the normal sinus of Valsalva.³ Anomalous aortic origin of a coronary artery includes a high variability of anatomical variants since the origin can be in the ascending aorta, the opposite sinus of Valsalva, or the noncoronary sinus. Multiple classifications of AAOCA have been proposed, based on anatomy, function, or clinical significance.⁴ In 1999, the nomenclature was standardized by the STS-Congenital Heart Surgery Database Committee and the European Association for Cardiothoracic Surgery.⁵ Anomalous aortic origin of a coronary artery can be subdivided into anomalous aortic origin of the left coronary artery (AAOLCA) and anomalous aortic origin of the right coronary artery (AAORCA).⁶ The relative incidence of AAORCA is estimated to be 3 to 9 times more common than AAOLCA.⁷

The importance of AAOCA is due to its clinical significance: AAOCA has been associated with myocardial ischemia and it is considered the second most common cause of sudden cardiac arrest in young athletes.^8,9 The risk of sudden death is widely acknowledged to be higher in AAOLCA than in AAORCA.⁶ The mechanisms suggested as possible causes for sudden death in AAOCA have been proposed in previously published articles¹⁰: the aortic pressure on an intramural segment, an interarterial segment “squeezed” between the aorta and pulmonary artery, spasm, kinking, or sharp angulation. In addition, the interarterial course of AAOCA has been related to increased risk of ischemia if there is a long intramural segment, acute angulation of the proximal segment or abnormally high take-off from the aorta.^10,11

In older adults, AAOCA typically presents with cardiac symptoms such as chest pain or dyspnea. Some authors have reported an association between AAOCA and coronary artery disease,¹² whereas others concluded there is no association.¹³ Somee authors have reported that AAOCAcontributed to a greater ischemic burden when symptoms appear at younger ages.³

When anatomical restoration treatment is indicated, surgical repair is the gold standard and surgical unroofing is the most common technique. Indications for surgery in AAOCA follow the guidelines of 2017¹⁴ and 2018¹⁵ and, in addition, a systematic review was presented recently by a multidisciplinary group of clinical and interventional cardiologists and cardiac surgeons proposing some recommendations regarding patient management.¹⁶ In general terms, surgery is recommended for individuals with AAOLCA arising from the right sinus of Valsalva and AAORCA arising from the left sinus of Valsalva if there are symptoms, arrhythmias, or ischemia inducible tests are positive. However, in all cases of AAOCA, the decision to perform surgery should be a patient-specific decision.

Although some large patient series have been published in the last few years from American registries, in Europe less data have been published regarding surgical correction of AAOCA. We aim to report our experience with AAOCA surgery and the long-term results.

Patients and Methods

Patients

This is a retrospective observational study including three centers with the same surgical team.

Sixteen adult patients underwent surgical repair of AAOCA in the last ten years, from 2013 to 2023.

The diagnosis was made based on the results of transthoracic echocardiography, computed tomography angiography, and coronary angiography findings.

The coronary morphology was determined based on a combination of imaging studies, operative reports, and clinical notes. Symptoms, comorbidities, and evidence of ischemia came from records closest to the date of the AAOCA diagnosis. Ischemia was defined based on stress test reports of electrocardiography, echocardiography, and myocardial perfusion imaging.

Follow-up was performed using the hospital database and direct contact with all the patients.

Surgical Technique

All operations included were performed by the same surgeon. Surgical unroofing of an intramural AAOCA was the most frequently employed surgical strategy; however, this technique was adapted to the patient´s variant of AAOCA. Neo-ostium creation and bypass grafting were also employed.

The three specific techniques employed in AAOCA correction are coronary unroofing, neo-ostium formation, and reimplantation. The decision to perform one of the previously mentioned surgical repairs depends on each patient's characteristics and anatomical aspects, the presence of an intramural segment and a subcommissural portion being the main determinants.

Coronary unroofing is a widely used technique which consists of an incision in the intramural segment from the ostium to the take-off of the anomalous coronary artery. Unroofing avoids significant coronary manipulation and reduces the risk of ischemia. Some of the possible complications of this technique are partial compression of the coronary artery, coronary angulation, residual interarterial segment, or aortic valve injury.^17–19 Coronary unroofing is the indicated technique in cases of intramural segment without a subcommissural portion.

In an neo-ostium technique, an unroofing of the intramural portion located at the correct anatomic location is performed without an incision of the entire intramural portion.¹⁷ A circumferential suture is placed into the aortocoronary incision to create the neo-ostium. In cases with intramural segment and a subcommissural portion, the neo-ostium is the appropriate technique.

Coronary reimplantation consists of reattaching the anomalous coronary artery to its respective sinus of Valsalva to restore normal physiology. It is the technique employed in cases of no intramural segment.

Statistical Analyses

Descriptive statistics and analyses were conducted using Stata/IC 16 (Stata Statistical Software: Release 146 College Station, TX: StataCorp LP). Results are summarized reporting the absolute numbers and percentages. For categorical variables, mean with standard deviation (SD), when normally distributed, or median with interquartile range (IQR) is given.

Results

Patient Characteristics

Sixteen adult patients underwent surgical correction of AAOCA. Two of the AAOCA were AAOLCA, anomalous origin of the left coronary artery with origin in the right coronary sinus (Figure 1) and 14 were AAORCA, anomalous origin of the right coronary artery from the left coronary sinus (Figure 2).

Median age was 43.3 years (IQR 13.9), 8 (50%) patients were males and the remaining eight (50%) were females.

All patients had an intramural segment, defined as a part of the coronary artery path within the aorta²⁰ which can be affected by a fixed-anatomical and dynamic stenosis that can vary during the cardiac cycle causing a systolic lateral compression from the aorta.

Two patients were elite athletes, two were nonelite athletes and the remaining 12 patients did not practice any sport. Table 1 summarizes the information about each patient, AAOCA symptoms, diagnostic test, and surgical technique.

Table 1.

Baseline Characteristics of Patient.

Id	Sex	Age (years)	Athlete^a	Symptoms	Ischemia detection test	AAOCA	Surgery
1	Male	47	No	Atypical chest pain	Positive ECG	AAORCA	Unroofing
2	Male	18	Elite	Chest pain during exercising	Negative	AAORCA	Unroofing
3	Male	44	No	Chest pain during exercising	Negative	AAORCA	Neo-ostium
4	Female	35	No	Atypical chest pain	Clinically positive	AAORCA	Unroofing
5	Male	56	No	Dyspnea, chest pain	None performed	AAORCA	Unroofing
6	Male	38	No	No symptoms	None performed	AAORCA	Neo-ostium
7	Female	38	No	Syncope, chest pain	None performed	AAOLCA	Neo-ostium
8	Male	53	No	No symptoms	None performed	AAORCA	Reimplantation
9	Male	20	Elite	No symptoms	MIBI-Spect positive	AAORCA	Unroofing
10	Male	43	Nonelite	Chest pain during exercising	Positive ECG	AAORCA	Unroofing
11	Female	73	No	Sudden cardiac arrest	Negative	AAOLCA	CABG
12	Female	43	Nonelite	Sudden cardiac arrest	MIBI-Spect positive	AAORCA	Unroofing
13	Female	39	No	Chest pain during exercising	Negative	AAORCA	Unroofing
14	Female	47	No	Atypical chest pain	Positive ECG	AAORCA	Unroofing
15	Female	29	No	No symptoms	Negative	AAORCA	Unroofing
16	Female	55	No	Atypical chest pain	Clinically positive	AAORCA	Unroofing

Abbreviations: AAOCA, anomalous aortic origin of a coronary artery; AAOLCA, anomalous aortic origin of the left coronary artery; AAORCA, anomalous aortic origin of the right coronary artery; CABG, coronary artery bypass grafting; ECG, electrocardiography; .

Athlete: No athlete, Elite athlete, or Nonelite (athlete but not in an elite competition).

Four patients were asymptomatic when diagnosed (25%) whereas 12 (75%) patients presented with cardiovascular symptoms: 9 (56.3%) suffered from exertional chest pain, syncope was present in 1 (6.25%) patient and 2 (12.5%) patients had aborted sudden cardiac death.

Of the 16 patients, 7 (43.8%) had ischemic manifestations or electrocardiogram changes on a stress test, whereas in 4(25%) patients the test was not performed.

The four asymptomatic patients were diagnosed incidentally after undergoing computed tomography angiography for other clinical reasons. One of them, the 20-year-old elite athlete, underwent a specific test as part of the process to participate in an international sports competition.

Surgical Procedures

Unroofing of an intramural AAOCA was performed in 11 patients. Figure 3 shows a case before and after unroofing of AAORCA, and Figure 4 shows a real image of coronary unroofing.

Three patients underwent neo-ostium creation (Figures 5 and 6). The remaining two patients underwent more complex surgeries. One patient underwent a David procedure with aortic valve sparing and coronary reimplantation; and the other patient was treated by coronary bypass grafting due to coronary atheromatous lesions in the anomalous aortic origin of a coronary artery (Figure 7).

In two patients, a concomitant procedure was performed: replacement of the pulmonary valve due to an associated congenital pulmonary valve stenosis in one patient and an atrial communication closure due to an atrial septal defect in the other.

Outcomes

The mean length of stay was 8.1 days (SD 4.7), median length of stay was 7 days (IQR, 7-8). There was no operative mortality.

All patients were discharged home without complications (neither ischemia or heart failure). Oral treatment with 100 mg per day of Aspirin was scheduled for three months after the surgical procedure. No exercise restrictions were recommended after the first three months, regarding the recommendations in the Guidelines on Sports, Cardiology and Exercise in Patients with Cardiovascular Disease.²¹

Follow-up period was 73.8 months, there was no late mortality.

In the follow-up echocardiography, none of the patients showed significant aortic regurgitation (considering significant more than mild regurgitation).

Two patients reported chest pain during the follow-up period. In both of them, ischemia detection testing was negative; echocardiography showed no alterations, and coronary angiography was performed showing no lesions and an intact surgical correction. These two patients also suffered from anxiety and nervousness; therefore, an anxiety disorder was suspected. The chest pain disappeared with anxiety treatment in both cases.

Discussion

AAOCA is a rare congenital lesion with high importance in clinical practice due to the association with myocardial ischemia and sudden cardiac arrest/death in young patients. Although AAOCA was commonly diagnosed in patients with ischemic symptoms, in recent years, it is frequently identified in screening imaging studies in asymptomatic patients.^2,22 When surgical repair is indicated, the most commonly used technique is surgical unroofing. Large series in the USA have been published showing excellent early outcomes,^18,23–25 however, in Europe less has been published in reference to surgical approach of AAOCA.^26,27

Our study reviewed 16 patients treated with surgical correction of AAOCA who had an excellent outcome. Although it is a small sample of patients with AAOCA, as far as we know, it is the only reported series of cases in which all patients underwent a surgical correction of AAOCA by the same surgeon. In addition, isolated case reports were published in Spain,^28–30 and a few studies were published in Europe^26,27 without any follow-up period.

The largest series in Europe,²⁷ published by Padalino et al, reviewed data from 16 cardiac centers in Europe including 156 patients who underwent surgical repair, 22.4% with AAOLCA and 67.3% with AAORCA. The authors reported a low incidence of operative deaths (1.3% due to two emergent cases) and morbidity. However, the authors did not find differences in terms of early survival comparing with patients who had not undergone surgery. Surgery provided benefits in terms of returning to normal activity when comparing patients with medical treatment alone.

Previously Blomjous et al²⁶ reported 40 cases of AAOCA in adults from which only one underwent surgery. Anomalous origin of the right coronary artery was diagnosed in 90% of the patients and AAOLCA in the remaining 10%. Sudden cardiac death was reported in just one patient, the one who underwent cardiac surgery. The authors were more conservative than other studies as their series showed a better prognosis in adults that did not undergo surgery. Additionally, the authors performed a systematic review of the literature including 20 studies and 1,193 patients. Cardiac surgery was performed in 32.6% of patients. The reported mortality rate was 6.3% during a follow-up of four years, higher than the nonsurgical patients. The systematic review did not demonstrate a protective role of cardiac surgery in AAOCA; however, a long-term follow up was not performed.

In our experience, surgical repair of AAOCA can be performed safely and should be considered after a careful risk assessment. Surgical correction could even be offered to asymptomatic patients if there is a risk of ischemia or sudden cardiac arrest, always taking into account the patient-sharing decision.

Type of Surgical Repair

Unroofing of the intramural segment is the more widely used technique since it avoids significant coronary manipulation.^6,17 Some limitations described for this technique included that it does not always remove the interarterial segment of the coronary artery, and the risk of aortic regurgitation.^17,31 However, unroofing has proven to be a feasible and simple surgical technique with excellent outcomes.^7,23,32

Other surgical approaches have been proposed. Law et al in a study of 16 cases in Australia³³ reported that surgical reimplantation of the anomalous right coronary artery can be performed safely with excellent medium to long-term outcomes. Bonilla-Ramirez et al¹⁷ in a very good quality review of the different surgical techniques, also compared unroofing, reimplantation, and neo-ostium in AAOCA correction. We believe that the surgical repair technique should be individualized depending on each patient's anatomy, and that the neo-ostium technique has better outcomes than unroofing in cases with a subcommisural course of the AAOCA. Although good results are published regarding all types of surgical approaches, it is important to note the significant technical challenges associated with coronary artery repair.

Unroofing of the coronary artery was the procedure we chose in most of our patients since it is a safe surgical technique that allows a satisfactory surgical correction. However, it is important to adequately match the treatment strategy to each patient to offer the best correction strategy, and for that reason we also performed complex procedures and neo-ostium correction in indicated cases.

Indication of Cardiac Surgery in AAORCA

One interesting point is the surgery indication of AAORCA. Anomalous origin of the right coronary artery was considered for many years to be a benign type of AAOCA. While AAOLCA was always considered a high-risk entity due to its well-reported association with ischemia and sudden cardiac death, AAORCA seemed to be less associated with symptoms.¹⁸ Anomalous origin of the right coronary artery is six to ten times more common than AAOLCA and, regarding previous articles, it is less associated with cardiac ischemia.³⁴ Brothers et al³⁵ calculated a cumulative risk of death over a 20-year period in athletes to be 6.3% for AAOLCA and 0.2% in AAORCA. Moreover, Jegatheeswaran et al³⁶ in a large cohort of 560 patients found that AAORCA is also associated with ischemia and sudden cardiac death. The authors reported that AAORCA might not always be benign, since it was present in half of the patients with ischemia and one-third of those with sudden cardiac events. These results are consistent with a series from Doan et al of 220 AAORCA patients,³⁷ in which 56 (26%) patients were identified as having high-risk features of sudden death and consequently surgical repair was performed. However, management of asymptomatic AAORCA in a young, active person is challenging,²³ since the surgical intervention has the potential risk of complications.³⁶ The risk of developing aortic regurgitation is about 10% in the published series^17,36 and up to 20 to 50% of patients reported chest pain after surgery.^19,36 Cardiac arrest after surgery, although a rare entity, has also been described,³⁸ and restenosis of the coronary ostia and progressive aortic valve regurgitation have also been reported as late complications after surgical unroofing.^32,39

We operated on 14 patients diagnosed with AAORCA, 25% asymptomatic, and all of them had an excellent outcome without surgical complication. In our opinion unroofing is a feasible, safe, and reproducible procedure that should be considered in these patients.

Results in Large Series

Recently, the Mayo Clinic²³ published their experience with a protocolized approach to AAOCA treatment in 148 consecutive patients who underwent surgical unroofing; 87.8% of the cases were AAORCA and the remaining 11.5% were AAOLCA. The authors reported a 1% incidence of early deaths, all due to reoperations for aortic root or valve replacement. After a follow-up period, late survival after AAOCA repair at 10 and 15 years was 94.5% and 94.5%. This is the largest series published regarding AAOCA surgery, and the conclusions were that surgical unroofing is a safe procedure with excellent outcomes.

Similarly, Mainwaring et al⁴⁰ published the results regarding AAOCA surgery from Lucile Packard Childrens Hospital in Stanford, California. One hundred and fifteen patients were operated; unroofing was the surgical technique employed in 86 patients, reimplantation in 9, and pulmonary artery translocation in 20, all with excellent outcomes. No early or late mortality were reported by the authors.

Regarding the Cleveland Clinic results,²⁵ 167 patients were diagnosed with AAOCA: 57% involving AAORCA, 23% AAOLCA, and 19% other type of AAOCA. Only 36 (22%) patients were treated surgically and unroofing was the most employed technique, being used in a higher proportion of AAORCA than AAOLCA; 47% of the patients were treated with unroofing technique, however, within those cases the authors included patients in which a neo-ostium was created. Although the surgical approaches varied, outcomes were favorable for the patients repaired, without any postoperative deaths, and complications were limited to new atrial arrhythmias.

Poynter et al⁷ reported 113 cases of surgical repair for AAOCA in patients younger than 30 years at 29 Congenital Heart Surgeons Society institutions. Unroofing was the surgical technique employed in 88% of the cases.

Conclusions

Although our series has fewer patients when compared with the large series published in the United States, little has been published in Spain and in Europe regarding AAOCA. As we mentioned, Blomjous et al²⁶ have published the largest series in Europe and only one patient underwent cardiac surgery. We operated on 16 patients and all of them had excellent outcomes. In addition, as far as we know, this is the only study published in our continent that includes a long follow-up period.

Our study has the limitations inherent to retrospective studies; however, no patient was missing. This is an observational study at a tertiary center with a selected population of patients and therefore subject to referral bias. Although the follow-up period is longer than several other previously published studies, even longer follow-up is clearly desirable. Similarly, a higher sample size is necessary to obtain more robust results.

Despite all these limitations, our clinical experience has shown that surgical repair of AAOCA can be performed safely without surgical mortality or complications.

In our patients, the surgical repair of AAOCA by unroofing or neo-ostium creation has been shown to have a very good outcome with excellent late survival.

The follow-up period, essential to accurately balance the risks of repairing AAOCA, showed the absence of morbidity and complications.

Surgical procedures for the treatment of AAOCA in are an effective strategy in adult patients with AAOCA.

Footnotes

Abbreviations

Author Contributions

All authors have contributed to the article.

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

ORCID iD

Laura Varela Barca

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