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Ann Thorac Surg 2007;84:156-160
© 2007 The Society of Thoracic Surgeons

Original Articles: Cardiovascular

Surgical Repair of Sinus of Valsalva Aneurysm in Asian Patients

Zheng-jun Wang, PhD, Cheng-wei Zou, MD, De-cai Li, MD, Hong-xin Li, MD, An-biao Wang, MD, Gui-dao Yuan, MD, Quan-xin Fan, MD*

Department of Cardiac Surgery, Shandong Provincial Hospital, Shandong University, Jinan, China

Accepted for publication March 9, 2007.

* Address correspondence to Dr Fan, Department of Cardiac Surgery, Shandong Provincial Hospital, Jingwu Rd 324, Jinan, 250021, PR China (Email: fanquanxin512{at}sohu.com).


   Abstract
Top
 Abstract
Introduction
 Patients and Methods
 Results
 Comment
 References
 
Background: Sinus of Valsalva aneurysm is a rare cardiac anomaly, and the difference between Asian and Western countries in its occurrence is not well established. This study was designed to investigate the difference between Asian and Western patients.

Methods: Between September 1988 and February 2006, 83 patients with sinus of Valsalva aneurysm underwent surgical repair in our institute. The aneurysms originated from the right and noncoronary sinus in 74 and 9, respectively, and ruptured into the right ventricle in 52 patients, the right atrium in 30, and the left ventricle in 1. Ventricular septal defect (n = 38), aortic regurgitation (n = 21), and bicuspid aortic valve (n = 4) were the common coexisting anomalies. To compare the differences between Asian and Western patients in sinus of Valsalva aneurysm, 1049 cases (654 Asian patients versus 395 Western) were collected from the literature.

Results: Sixty-six patients were followed up for 9.6 ± 3.8 years. The cardiac function of 15 patients with aortic regurgitation was worse than that of those with no aortic regurgitation (p < 0.05). There was no difference between the direct closure and the patch closure (p > 0.05). Analysis of all collected cases revealed that aneurysm of the sinus of Valsalva in Asian patients compared with Western series is characterized by a higher incidence, more aneurysms originating from the right coronary sinus (85.8% versus 67.9%), more aneurysm rupture into the right ventricle (72.5% versus 60%), a higher incidence of association with ventricular septal defect (52.4% versus 37.5%), and lower incidence of association with bicuspid aortic valve (0.6% versus 7.8%). However, both Asian and Western patient series have similar incidence of combination with aortic regurgitation (33.6% versus 32.7%).

Conclusions: Long-term results of ruptured sinus of Valsalva aneurysm are associated with preoperative aortic regurgitation. The difference between Asian and Western patients with ruptured aneurysm of the sinus of Valsalva is significant.


   Introduction
Top
 Abstract
 Introduction
Patients and Methods
 Results
 Comment
 References
 
Sinus of Valsalva aneurysms (SVAs) are relatively rare anomalies, most often caused by the congenital absence of muscular and elastic tissue in the aortic wall of the sinus of Valsalva [1]. Males are more often affected than females (ratio 3 to 4:1) [2, 3], and the incidence is higher in the Eastern than in the Western population [4, 5].

In recent years, improvements in diagnostic techniques have resulted in increasing numbers of these patients undergoing surgical repair. A variety of various surgical methods have been reported with good results; however, the long-term results after operation remain unclear. In this report, we review our 18-year experience with SVAs to analyze the determinants influencing the long-term results after surgical repair and the relevant literature to reveal the difference between Asian and Western countries in ruptured aneurysm of the sinus of Valsalva.


   Patients and Methods
Top
 Abstract
 Introduction
 Patients and Methods
Results
 Comment
 References
 
Patient Profile
During the 18-year period from September 1988 to February 2006, 83 patients (55 males and 28 females) underwent surgical repair for aneurysm of the sinus of Valsalva, which comprised 1.2% of the 6831 open heart operations performed in Shandong Provincial Hospital during this period. The Ethics Committee approved this study and waived the need for individual patient consent.

The mean age of patients at repair was 30.7 ± 12.1 years (range, 3 to 69 years). At the time of admission, 71 patients (86%) were symptomatic and 22 (27%) were in New York Heart Association (NYHA) functional classes III and IV. Major symptoms were dyspnea in 59 (71%), palpitation in 45 (54%), fatigue in 40 (48%), chest pain in 15 (18%), swoon in 3 (4%), fever in 1 (1%), and twitch in 1 (1%).

A continuous "machinery-type" murmur was heard at the left sternal border in 67 patients (81%). Chest roentgenogram findings included an increased cardiothoracic ratio of 0.56 ± 0.08 (range, 0.38 to 0.8). Electrocardiographic findings were normal in 26 patients, and 28 had left ventricular hypertrophy, 2 had biventricular hypertrophy, 6 had right-axis deviation, and 1 had atrial fibrillation. All patients were diagnosed through echocardiogram except 6, who were diagnosed by aortography.

Operative Procedure
Surgical repair was done under cardiopulmonary bypass (CPB) with moderate hypothermia through median sternotomy in all patients. The left ventricle was vented. Before ventricular fibrillation, the aorta was cross-clamped and cardioplegia was infused. In the patients with no aortic regurgitation and those with no rupture of the aneurysm, the cardioplegia was infused directly into the aortic root; otherwise, coronary ostial cardioplegia infusion or retrograde cardioplegia infusion was used. Cold crystalloid cardioplegia (n = 27), cold blood cardioplegia (n = 49), and warm blood cardioplegia (n = 7) were used for myocardial protection. The mean aortic cross-clamp time was 42 ± 19 minutes (range, 16 to 111 minutes), and the mean CPB time was 70 ± 25 minutes (range, 24 to 164 minutes).

Repairs were achieved through the chamber of termination in 49 (right atrium in 10; right ventricle in 39), aortotomy in 5, or double-chamber (right atrial/ventricle and aortic) in 29. The origins of the SVAs and the cardiac chambers into which they ruptured are summarized in Table 1. Ruptured SVAs (RSVA) were found in 66 patients during operation. Single or multiple ruptured holes were found, measuring 0.70 ± 0.22 cm in diameter (range, 0.2 to 1.5 cm).


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  		Table 1 Sinus of Origin and Cardiac Chamber Exit of the Ruptured Aneurysm
 
After excising the aneurysm tissue, the defect in sinus of Valsalva was closed. The techniques used for closure of SVA orifice and ventricular septal defect (VSD) are summarized in Table 2. Direct suture closure of RSVA was done in 46 patients (pledged interrupted sutures in 43, continuous suture in 3), and patch closure was used in 37 patients in whom the diameter of the defect in sinus of Valsalva exceeded 0.8 cm.


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  		Table 2 Closure of Ruptured Sinus of Valsalva Aneurysm and Ventricular Septal Defect
 
Coexisting cardiac lesions are listed in Table 3. Thirty-eight patients (46%) had VSD, of which 25 VSDs were subarterial. In these patients, the VSD was closed directly with pledgeted mattress sutures in 14 patients and with a Dacron patch (DuPont, Wilmington, DE) in 24. Among the 21 patients (25%) with aortic regurgitation, 12 patients had VSD, 6 patients required aortic valve replacement for severe and irreparable aortic regurgitation, and 4 patients required valvuloplasty. Other malformations included bicuspid aortic valve in 4 (5%), right ventricular outlet stenosis in 2 (2%, right ventricular outlet enlarged), tricuspid insufficiency in 5 (6%), among which 2 patients required tricuspid valve repair for severe insufficiency; and 1 patient (1%) each with a double-chambered right ventricle, pulmonary stenosis (pulmonary valve was incised), and patent ductus arteriosus (direct closure with a pledgeted mattress suture).


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  		Table 3 Associated Lesions and Corresponding Procedures
 
Patients were followed up at our outpatient department. Telephone calls were made and questionnaires were sent to patients for data collection, when necessary. Heart function was appraised according to the echocardiogram and reported symptoms.

Published reports on RSVA from all over the world were reviewed to compare the differences between Asian (China, Japan) and Western countries (North America and Europe) in this rare cardiac lesion.

Statistical Analysis
This study was designed to investigate the determinants of long-term results after surgical repair of sinus of Valsalva aneurysm and to distinguish the difference between Asian and Western population. The {chi}2 test was used for statistical analysis and values of p < 0.05 were considered significant. Data are presented as the mean ± standard deviation.


   Results
Top
 Abstract
 Introduction
 Patients and Methods
 Results
Comment
 References
 
Surgical Results of the Present Series
In the present series, there was no hospital mortality. The postoperative hospital length of stay was 14.3 ± 6.4 days before 1997 and 10.8 ± 2.5 days after 1997. The postoperative mechanical ventilation support time was 9.2 ± 4.8 hours. Postoperative complications included skin wound infection in 4 patients, arrhythmia in 4, bleeding in 3 (cardiac tamponade in 1 patient), and 1 patient each with VSD residual shunt, right pneumothorax, and acute left-sided heart failure for severe aortic valve incompetence after operation.

A total of 66 patients (79.5%) patients were followed up. The average follow-up period for survived patients was 9.6 ± 3.8 years (range, 2 months to 16 years). None of these patients had any residual fistula. One late postoperative death occurred as a result of the rupture of a dissecting aortic aneurysm 7 years after operation.

All survivors were found to be in NYHA functional classes I–III. Among the 66 follow-up patients, the heart function in 15 with preoperative aortic regurgitation is less satisfactory, with 6 patients (40%) in functional class I. The heart function in 51 patients without preoperative aortic regurgitation is much better, with 45 patients (88%) in functional class I. The difference between the two groups was statistically significant ({chi}2 = 4.46, p < 0.05). Of the 38 patients who underwent direct closure, 29 (76%) are in functional class I, and 25 (89%) of 28 who underwent patch closure are in functional class I. There was no difference between these two groups ({chi}2 =1.06, p > 0.05).

Differences Between Asian and Western Populations
The incidences of RSVA are much higher in Asian populations (about 1.2% to 4.94%) than in Western populations (about 0.5% to 1.5%). In a collected review of 1049 patients (654 Asian versus 395 Western) in reported series [2, 6–21], the differences between the Asian and the Western patients in the origin of the SVA, the cardiac chamber into which the SVA ruptured, and the incidences of association with VSD and congenital bicuspid valve are all statistically significant, as listed in Tables 4, 5, and 6. Go Go The incidence of association with aortic regurgitation is similar in both Asian and Western series (Table 6), however.


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  		Table 4 Difference Between Asian and Western Population in Origin of Ruptured Sinus of Valsava Aneurysm
 

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  		Table 5 Difference Between Asian and Western Population in Chambers Into Which Sinus of Valsalva Aneurysm Ruptured
 

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  		Table 6 Associated Congenital Cardiac Anomalies in Ruptured Sinus of Valsava Aneurysm
 

   Comment
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
References
 
Aneurysm of the sinus of Valsalva is a rare congenital heart disease that constitutes about 0.14% to 1.5% of congenital heart repairs [22]. It is uncommonly associated with endocarditis, atherosclerosis, syphilis, or aortic dissection [23]. Because the aortic valve occupies a central position in the base of the heart, an SVA can rupture into any of the four heart chambers, especially into right atrium and right ventricle. Occasionally, it ruptures into the pulmonary artery [24, 25] or interventricular septum [8].

Compared with Western patients, Asian patients have a higher incidence of RSVA, with a remarkable tendency to protrude and rupture into right ventricle rather than right atrium (Table 5), and a high incidence of origin from the right coronary sinus (Table 4). In addition to being more evenly distributed between the right ventricle and right atrium, a few cases of RSVA showed rupture into the left ventricle [8, 12, 13, 20, present series], the left atrium [12, 15, 16, 20], and the interventricular septum [8, 13]. No patient had fistula formation into the pulmonary artery.

Coexisting lesions are common in patients with congenital, ruptured SVA. Subarterial and perimembranous VSDs occur in Western patients with an incidence of 30% to 50% [13]. In comparison, Asian patients have a higher incidence of VSD, and in our collected 654 patients from China and Japan, VSD occurred in 343 patients (52.4%), whereas only 37.5% of Western patients had associated VSD, the difference between them is statistically significant (p < 0.005).

Next to ventricular septal defect, aortic regurgitation in RSVA is also a commonly associated lesion, with 33.6% in the Asian group and 32.7% in the Western group (Table 6). Anatomically, SVA deprives the aortic sinus and aortic annulus of endocardial muscular support. Hemodynamically, the flow of aortic root run-off produces the Bernoulli effect, a tendency for the related aortic cusp to pull away from closure. These two mechanisms induce aortic regurgitation, and aortic regurgitation begets aortic regurgitation and produces some secondary changes, making surgical treatment of aortic regurgitation necessary in some patients in addition to repairing the RSVA. In our present series, 6 patients (28.6%) required aortic valve replacement for severe and irreparable aortic regurgitation, and 4 patients required valvuloplasty. The aortic valve replacement rate in Asian patients is higher than in Western countries (7% to 20%) [13].

In addition to VSD and aortic regurgitation, recent reports have indicated that aortic valve abnormalities are common in patients with RSVA. Ring [26] reported a 10% incidence of bicuspid aortic valve, and Azakie and associates [13] reported a 20.59% incidence, the latter authors suggested there was a trend to late aortic valve repair failure in the presence of a bicuspid aortic valve. In our collected series, a bicuspid aortic valve was present in 31 Western patients (7.8%) but in only 4 Asian patients, a statistically significant difference (p < 0.005).

Surgical repair is the most effective treatment of RSVA. With patients who have no associated VSD, transcatheter closure is feasible and effective [27]. The RSVA or unruptured SVA but with VSD or aortic valve regurgitation, or both, a huge SVA causing mitral valve incompetence [28], right ventricular outflow obstruction [29], and myocardial ischemia [30] should be surgically repaired as soon as the diagnosis is confirmed.

We prefer to use a combination of approaches through the chamber of termination and aortotomy, although most fistula repair can be accomplished solely through the chamber of termination. This allows us to observe the situation of the aortic root and aortic valve simultaneously with the intention of preventing aortic valve twisting and residual shunt of RSVA.

The present study has demonstrated that patients who underwent RSVA repair have satisfactory long-term results. Survival is 90% ± 7% [26] at 10 years and 93% [15] at 20 years. Residual or progressive aortic regurgitation is the important factor influencing prognosis [11, 15]. Our study also suggests that aortic regurgitation is a very important factor influencing cardiac function after operation. The cardiac function of 2 patients who underwent aortic valvuloplasty and 6 patients with mild aortic valve regurgitation who were not treated during operation decreased because of deteriorated aortic regurgitation. However, the relationship between methods of repair and prognosis is not close. So we definitely think that aortic valve replacement, but not aortic valvoplasty, should be performed in patients with moderate-to-severe aortic valve regurgitation; furthermore, some patients in our country cannot afford the second operation.

In conclusion, the RSVA and unruptured SVA but with VSD or aortic regurgitation, or both, should be repaired surgically as early as possible. The outcome of operation and long survival is encouraging. In some aspects, the difference between Asian and Western patients is statistically significant.


   References
Top
 Abstract
 Introduction
 Patients and Methods
 Results
 Comment
 References
 

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