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Ann Thorac Surg 1998;66:1604-1610
© 1998 The Society of Thoracic Surgeons

Repair of ruptured sinus of valsalva aneurysm: Determinants of long-term survival

^a Division of Cardiothoracic Surgery, Department of Surgery, University of Hong Kong, Grantham Hospital, Aberdeen, Hong Kong

Accepted for publication May 7, 1998.

Address reprint requests to Prof He, Cardiothoracic Surgery, University of Hong Kong, Grantham Hospital, 125 Wong Chuk Hang Rd, Aberdeen, Hong Kong

	Abstract

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Background. Ruptured sinus of Valsalva aneurysm is a rare cardiac anomaly and long-term survival after surgical treatment is not well established. This study was designed to investigate the determinants of long-term survival after repair of ruptured sinus of Valsalva aneurysm.

Methods. From April 1978 to April 1996, 53 patients underwent operation for ruptured sinus of Valsalva aneurysm. The incidence among our cardiac surgical population was 0.56%. Long-term survival was investigated in 46 patients (13 to 65 years) who survived the operation, with 96.2% follow-up completeness (mean ± standard deviation, 6.5 ± 4.9 years; maximum, 17.2 years), by univariate and multivariate analyses.

Results. There was no early operative death and no recurrence after the initial repair. Actuarial survival was 83.8% ± 8.4% at 15 years. Reoperation, aneurysm draining into the left ventricle, aortic prosthetic dehiscence, bacterial endocarditis, and aortic cross-clamp time (<70 minutes) were significant factors in long-term survival (p < 0.05). Multivariate analysis revealed that only aortic prosthesis dehiscence was the significant factor influencing late survival (p = 0.0001).

Conclusions. Surgical treatment for ruptured sinus of Valsalva aneurysm is safe and has satisfactory results. Aortic prosthesis dehiscence is the independent determinant for long-term survival. Other factors including bacterial endocarditis, concomitant ventricular septal defect repair, and aortic valve replacement did not independently influence long-term survival.

	Introduction

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Ruptured sinus of Valsalva aneurysm (RSVA) is a rare cardiac abnormality but is relatively more common in the Asian population [1]. The RSVA produces significant left-to-right shunt or left ventricular volume overload, depending on the communicating heart chambers, and will progress to heart failure. Surgical intervention is indicated once the diagnosis is confirmed to prevent further cardiac complications. It has been demonstrated that surgical repair of RSVA carries low morbidity and mortality [2–5] since it was first repaired successfully by Lillehei and colleagues in 1957 [6]. However, the presence of bacterial endocarditis and concomitant cardiac procedures, such as aortic valve replacement (AVR) and closure of ventricular septal defect (VSD), may carry higher surgical risks [2, 4, 5]. Although prognostic factors for RSVA repair have been studied [5, 7], such determinants may not apply to the Asian population as they have a distinct disease pattern compared with the Western series. Also, little has been reported in relation to long-term survival. The present study was designed to investigate the determinants influencing the long-term survival of patients who underwent RSVA repair and the effect of concomitant cardiac procedures on late survival.

	Material and methods

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Between April 1978 and April 1996, 53 patients (35 men and 18 women) with ruptured sinus of Valsalva were operated on at the Division of Cardiothoracic Surgery, Department of Surgery, University of Hong Kong, The Grantham Hospital. During the same period of time, there were 9,514 cardiac operations requiring cardiopulmonary bypass, for an overall incidence of RSVA of 0.56%. The mean age was 35.8 ± 12.2 years (standard deviation) (range, 13 to 65 years). The majority of the patients were Chinese (96.4%) with 1 white patient (1.7%) and 1 Filipino patient (1.7%).

At the time of admission, 50 patients (94.3%) were symptomatic and 17 patients (32%) were in the New York Heart Association functional classes III and IV. The majority had the following symptoms: dyspnea (79.2%), palpitation (50.9%), fatigue (47.2%), angina (26.4%), and syncope (3.8%). Three patients were totally asymptomatic and murmurs were detected during routine physical examination. In this series, 1 patient had lymphoma, 1 had thyrotoxicosis, and 1 patient had ß-thalassemia. Heart murmurs were heard in all patients, and 51 patients (96.3%) presented with loud continuous "machinery-type" murmur. The other 2 patients had loud systolic and diastolic murmurs that were heard best at the left sternal border. Chest roentgenograms showed cardiomegaly (cardiothoracic ratio >0.5) in 39 patients (72.2%) and varying degrees of pulmonary plethora. With regard to electrocardiographic findings in 19 patients with isolated ruptured sinus of Valsalva, 8 were normal and 5 had left ventricular hypertrophy, 1 had biventricular hypertrophy, 1 showed left heart strain, and 1 had right-axis deviation. One patient presented as an emergency with ventricular fibrillation. There were 3 patients (5.7%) in atrial fibrillation and the rest (98.2%) were in sinus rhythm, among them 4 patients with frequent ventricular ectopics.

A history of bacterial endocarditis was found in 11 patients (20.8%) and 7 patients were treated with full course of antibiotics and had negative blood cultures before operation. Bacteriology of the endocarditis is shown in Table 1. All patients had cardiac catheterization and 46 patients (86.8%) also had echocardiography. The shunt ratios were available in 45 patients (84.9%), with a mean value of 2.4 ± 1.0.

Operation
Surgical repair was carried out by using cardiopulmonary bypass in all patients. The circulatory arrest technique was not necessary in our series and only moderate hypothermia (28° to 30°C) was used with the exception of 1 patient when the patient was cooled down to 25°C. The latter had bacterial endocarditis and biventricular hypertrophies; the operation required closure of VSD, repair of the RSVA, and aortic valve replacement. Cold antegrade crystalloid cardioplegia had been used for all patients. In patients with significant shunting, such as large ruptured sinus of Valsalva aneurysm or moderate to severe aortic incompetence, direct infusion of the cardioplegic solution into the coronary ostia was used. The mean aortic cross-clamp time was 54.4 ± 24.8 minutes (range, 16 to 140 minutes) and the mean bypass time was 72.0 ± 31.6 minutes (range, 31 to 210 minutes).

Surgical findings
The ruptured aneurysms were typically "wind sock" in 50 patients (94.3%). Of 3 patients (5.7%) who had a simple fistula tract, 1 had evidence of bacterial endocarditis and communication between the left coronary cusp and the left ventricle, 1 had communication between the noncoronary cusps and the right atrium, and the last patient had communication between the right coronary cusp and the right ventricle. One patient’s aneurysm originated from the right coronary sinus, which was bisaccular but only one of the saccules ruptured. Ruptured holes were either single or multiple, measuring up to 20 mm in diameter. The origins of the aneurysm of sinus of Valsalva and the communicating cardiac chambers are shown in Table 2. Thirty-four patients (64.2%) had coexistent cardiac lesions, 26 (49.1%) had VSD, 22 (41.5%) had supracristal VSD, and 4 patients (7.5%) had perimembraneous VSD. One patient was found to have VSD, a prolapsed aortic valve, and a patent ductus arteriosus; another patient was found to have mitral incompetence. Twenty-three patients (43.3%) had aortic incompetence, of whom 15 had a prolapsed aortic valve. One patient had pulmonary artery stenosis.

Direct closure of the aneurysmal base after excision was done in 3 patients (5.7%) in the early stage of the series and 2 of them had aortic valve replacement, in which the prosthetic annulus was used as part of the reinforcing material to secure the aneurysmal repair. Twenty-six patients had associated VSDs. Teflon patch was tailored to repair the RSVA defect as well as the VSD in 17 patients (30.2%) (13 supracristal, 4 perimembraneous), but in the other 10 patients with VSD (9 supracristal, 1 perimembraneous), only Teflon-buttressed sutures (PTFE, polytetraflu-oroethylene, Meadox Medicals, Inc.) were used to repair the RSVA defect. Fourteen patients (26.4%) had aortic valve replacement and 6 of them had VSD repaired as well. All 7 patients with severe aortic regurgitation had aortic valve replacement. Six of 16 patients with mild to moderate aortic regurgitation required AVR but in the rest of this group of patients no aortic valvuloplasty was performed. Thirteen patients had mechanical valve prostheses. Other procedures associated with RSVA are as follows: VSD, 20 patients; AVR, 8; AVR plus VSD repair, 6; mitral valve replacement, 1; patent ductus arteriosus ligation, 1; and pulmonary infundibular resection, 1 patient.

Statistical methods
This study was designed to investigate the long-term survival and freedom from complications after surgical repair of ruptured sinus of Valsalva aneurysm. Univariate analysis with regard to actuarial survival was done by the Kaplan-Meier [8] and life table methods (SPSS, Inc, Chicago, IL). Log-rank test is used for statistical analysis and p value less than 0.05 is considered significant. Complication-free survival was also established. For multivariable analyses, we used Cox regression model [9] with risk factors that were significant or those with p value < 0.1 obtained by univariate analysis. Six variables (bacterial endocarditis, aortic cross-clamp time <70 minutes, reoperation, concomitant aortic valve replacement, aortic prosthesis dehiscence, and communication of aneurysm of sinus of valsalva to the left ventricle) were selected and they were entered into a forward stepwise regression analysis and p value of less than 0.05 was considered significant.

	Results

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
There were no early operative deaths after repair of RSVA. There was a total of 5 late deaths (9.4%) during the follow-up period. Two of them were early as a result of reoperation. One patient had aortic valve prosthesis endocarditis with dehiscence and died soon after the reoperation to replace the infected prosthesis, 3 months after the initial operation. The other patient also had aortic valve prosthesis dehiscence but he underwent two further operations to replace the infected prosthesis and died of sepsis and multiorgan failure 6 months after the initial operation. The other late death was in a patient diagnosed with stage IV lymphoma before the operation; he died 7 months later with no evidence of recurrence of ruptured aneurysm. One patient died of sepsis and massive intracranial bleeding 10 months after operation. Diabetes mellitus developed after the operation in another patient with renal failure; she died of end-stage renal failure 12 years later.

Three patients (5.7%) required reexploration in the early postoperative period because of bleeding and all recovered well. A total of 4 patients required reoperations: 2 of the them died because of infected aortic valve prostheses; the third patient, who initially had VSD, patent ductus arteriosus, and ruptured aneurysm of sinus of Valsalva corrected, had development of severe aortic regurgitation and required subsequent aortic valve replacement 9 years later; and the fourth patient, who had aortic valve prosthesis infection and leakage, required a secondary replacement. A cerebral vascular accident developed in 1 patient soon after operation. A cerebral vascular accident developed 6 years after the initial operation in 1 patient with bacterial endocarditis and aortic valve replacement. All except for 1 patient remained in sinus rhythm after operation. That patient, who is the only white patient in this study, had bacterial endocarditis and aortic valve replacement; a complete heart block developed immediately after operation and a permanent pacemaker was inserted. At present there are no recurrences of sinus of Valsalva aneurysm, and patients who had mild aortic incompetence preoperatively remained stable after operation.

Forty-five patients were alive at the completion of the follow-up and all are in functional classes I and II (mean New York Heart Association functional class value was 1.42 compared with the preoperative mean value of 2.26) and the improvement was statistically significant (p < 0.0001).

Long-term survival and complication-free survival
Actuarial survival was 91.8% at 10 years (number of patients at risk, 14), 83.8% at 15 years (n = 1). Complication-free survival was 84.6% at 5 years (n = 25), 75.3% at 10 years (n = 11), and 66.9% at 15 years (n = 1) (Fig 1 ).

View larger version (15K): [in this window] [in a new window]   Fig 1. Total survival and event-free survival graph.

Influence of bacterial endocarditis on long-term survival
The influence of preoperative bacterial endocarditis was shown to decrease survival. In patients without a preoperative history of bacterial endocarditis, the survival was 97.3% ± 2.7% at 10 years (n = 23) and 87.0% ± 9.9% at 15 years (n = 1). In contrast, for patients with a preoperative history of bacterial endocarditis, the survival was 72.7% ± 13.4% at 10 years (n = 3) (p = 0.03) (Fig 2 ).

View larger version (18K): [in this window] [in a new window]   Fig 2. Influence of subacute bacterial endocarditis (BE) on long-term survival after ruptured sinus of Valsalva aneurysm repair. Patients with preoperative subacute bacterial endocarditis had significantly decreased survival (p = 0.03).

View larger version (21K): [in this window] [in a new window]   Fig 3. Influence of prolonged aortic cross-clamp time on long-term survival after ruptured sinus of Valsalva aneurysm repair. Patients who had shorter cross-clamp time (<70 minutes) had better survival (p = 0.037).

View larger version (20K): [in this window] [in a new window]   Fig 4. Influence of aneurysm drainage to cardiac chamber on long-term survival in ruptured sinus of Valsalva aneurysm repair. Patients who had ruptured sinus of Valsalva aneurysm draining to the left ventricle (LV) had poorer long-term survival (p = 0.0006).

View larger version (21K): [in this window] [in a new window]   Fig 5. Influence of reoperation after the initial ruptured sinus of Valsalva aneurysm repair on long-term survival. Patients who underwent reoperation did worse than those who did not require reoperation (p = 0.005).

View larger version (22K): [in this window] [in a new window]   Fig 6. Influence of aortic prosthesis dehiscence on long-term survival after ruptured sinus of Valsalva aneurysm repair and concomitant aortic valve replacement. Patients who had aortic prosthesis dehiscence had very poor survival (p < 0.00001).

Influence of ventricular septal defect on long-term survival
In the present study, presence of VSD did not significantly influence long-term survival. For 26 patients (49.1%) with concomitant VSD who required surgical repair, no statistical differences with regard to the long-term survival were found between the two groups (p = 0.24).

Multivariate analysis (Cox proportional hazard regression)
Six variables with p values less than 0.1 were included in the Cox hazard regression analysis: reoperation, drainage of RSVA to the left ventricle, concomitant AVR, bacterial endocarditis, prolonged aortic cross-clamp time (>70 minutes), and aortic prosthesis dehiscence. The results showed that aortic prosthesis dehiscence was the only independent variable for determining long-term survival after RSVA repair in this series (standard error = 1.1723, p = 0.0001, ß coefficient = 4.495, and Exp (B) = 0.0112).

	Comment

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
Ruptured sinus of Valsalva aneurysm is a rare cardiac anomaly and the incidence ranges from 0.23% to 1.5% [1–3, 5, 10]. Chu and colleagues [1] suggested that the incidence of RSVA in Asian patients is about five times more common than in white populations, although the reason is unclear. The incidence in our series was 0.56%, which was similar to other Asian studies [1, 2]. Surgical repair of RSVA has been shown to be effective to prevent late complications and with acceptable mortality [1, 5]. Factors affecting long-term survival after RSVA repair have not been well established. Although there is a frequent association of RSVA with other cardiac lesions, especially aortic valve incompetence and VSD, little information with regard to their concomitant surgical repair on late survival is available.

The present study has demonstrated that patients who underwent RSVA repair have satisfactory long-term results. Aortic prosthesis dehiscence is the only determinant of late survival. Our study shows that patients with aortic prosthesis dehiscence had poor results as none of them survived more than 1 year with average survival of only 5.8 months. In a recent autopsy study of RSVA, van Son and associates [11] showed that the average area (mean diameter, 1.1 cm) of the defect between the aortic media and the aortic annulus is far larger than the size of the rupture hole at the base of the RSVA (mean diameter, 0.7 cm). This could be one of the reason why the aortic prosthesis dehisced as the anchoring stitches were put on to the weakened annulus where the elastic media was deficient. However, a probably more important factor is infection, as in our series all 3 patients who had prosthetic dehiscence had infections (bacterial endocarditis or sepsis).

Abe and Komatsu [2] suggested that AVR or mitral valve regurgitation could be related to late death. In our multivariate analysis, AVR is not a significant factor for determining long-term survival. Bacterial endocarditis is another common complication of untreated RSVA. About one-fifth of the patients in this series had evidence of bacterial endocarditis. This can be explained by its more turbulent flow across the valve and across the fistula leading to repeated trauma to the valve cusp and the fistula tract. Bacterial endocarditis has been linked to late mortality [2, 4, 5]. Its significance with regard to long-term survival, however, has not been well established. In this study, although all 3 patients with aortic prosthetic dysfunction were found to have bacterial endocarditis, subacute bacterial endocarditis was not an independent factor for survival in the multivariate analysis.

The morphology of the RSVA was described by Edward and Burchell [12] in 1957 showing the absence of normal elastic tissue in the media between the aortic sinus and the hinge line of the aortic annulus. The cause of the sinus of Valsalva aneurysm was believed to be attributable to incomplete fusion of the distal bulbar septum that separates the aortic and pulmonary valves of the bulbus cordis, resulting in aneurysm formation when subjected to prolonged period of high pressure [13]. The left coronary cusp does not usually arise from the bulbar septum, as do the right and noncoronary cusp, therefore explaining the rarity of left RSVA. In the present study, we reported only 1 patient (1.9%) with a left RSVA and this is consistent with other large series [1, 5, 14, 15].

The majority of the patients in this series were symptomatic (94%) and 17 patients (32%) were in functional class III or IV, which is similar to other reports [1, 5, 15]. Intracardiac rupture of sinus of Valsalva aneurysm produce symptoms of heart failure with either significant left-to-right shunt or left ventricular overload, the latter condition mimicking the hemodynamic ill effect of severe aortic incompetence. The traditional wind sock-type lesions were seen in 50 patients (94%) and the majority of RSVA were drained into the right heart chambers (right atrium, 11; right ventricle, 38) and the findings were similar to other series [1, 5, 7, 14].

Twenty-six patients (49%) had VSDs, which was the most common cardiac lesion associated with the sinus of Valsalva aneurysms and 85% were supracristal. Chu and colleagues [1] suggested that Asian patients with RSVA have a higher incidence of VSD with the majority being supracristal compared with white patients. However, although van Son and associates [5] found some correlation between subarterial VSD and late complications, especially for the risk of reoperation, we are unable to correlate VSD with significant late death in this series.

We used the combined approach for RSVA repair in which the involved heart chamber and the aorta were opened in 49 patients. Aortotomy has been widely performed in RSVA repair, first suggested by Lillehei and colleagues in 1957 [6] and its advantages have been fully appreciated since. More recently with the aid of echocardiographic assessment of aortic valve preoperatively, 4 patients had their RSVA repair only through their involved heart chambers without aortotomy. None of them had aortic incompetence after operation and remain well in the postoperative period.

We have reported no recurrence of RSVA (cases with aortic prosthetic dehiscence were excluded); however, the rate of recurrence was reported to be as high as 20% by Barragry and colleagues [7]. We believe that one of the reasons is that some of their RSVA lesions were closed by simple sutures without reinforcement. Van Son and associates [11] recommended patch closure rather than simple suture closure of RSVA even if the RSVA appears small. However, we recommend the routine use of Teflon buttress to close the RSVA, preferably from both ends of the lesions, as Teflon felts are easier to apply especially in the aortic sinus where patch may be difficult to use when the ruptured hole is small.

We conclude that surgical treatment of RSVA is safe and has satisfactory results. Aortic prosthesis dehiscence is the independent determinant for long-term survival. Other factors, including bacterial endocarditis, concomitant VSD repair, and aortic valve replacement, did not independently influence long-term survival.

	Acknowledgments

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 
We thank Dr Jian-An Yang for his invaluble help in data collection.

	References

Top Abstract Introduction Material and methods Results Comment Acknowledgments References

 

1. Chu S.H., Hung C.R., How S.S., et al. Ruptured aneurysms of the sinus of Valsalva in Oriental patients. J Thorac Cardiovasc Surg 1990;99:288-298.[Abstract]
2. Abe T., Komatsu S. Surgical repair and long-term results in ruptured sinus of Valsalva aneurysm. Ann Thorac Surg 1988;46:520-525.[Abstract]
3. Mayer E.D., Ruffmann K., Saggau W., et al. Ruptured aneurysms of the sinus of Valsalva. Ann Thorac Surg 1986;42:81-85.[Abstract]
4. Hamid I.A., Jothi M., Rajan S., Monro J.L., Cherian K.M. Transaortic repair of ruptured aneurysm of sinus of Valsalva: fifteen-year experience. J Thorac Cardiovasc Surg 1994;107:1464-1468.[Abstract/Free Full Text]
5. Van Son J.A.M., Danielson G.K., Schaff H.V., Orszulak T.A., Edwards W.D., Seward J.B. Long-term outcome of surgical repair of ruptured sinus of Valsalva aneurysm. Circulation 1994;90(Suppl 2):20-29.
6. Lillehei C.W., Stanley P., Varco R.L. Surgical treatment of ruptured aneurysms of the sinus of Valsalva. Ann Surg 1957;146:459-472.
7. Barragry T.P., Ring W.S., Moller J.H., Lillehei C.W. 15- to 30-year follow-up of patients undergoing repair of ruptured congenital aneurysms of the sinus of Valsalva. Ann Thorac Surg 1988;46:515-519.[Abstract]
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11. Van Son J.A.M., Sim E.K.W., Starr A. Morphometric features of ruptured congenital sinus of Valsalva aneurysm: implication for surgical treatment. J Cardiovasc Surg 1995;36:433-436.[Medline]
12. Edwards J.E., Burchell H.B. The pathological anatomy of deficiencies between the aortic root and the heart, including aortic sinus aneurysms. Thorax 1957;12:125-139.
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14. Pan-Chih, Ching-Heng T., Chen-Chun, Chieh-Fu L. Surgical treatment of the ruptured aneurysm of the aortic sinuses. Ann Thorac Surg 1981;32:162-166.
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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): David Cheung Guo-Wei He Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Au, W.-K. Articles by He, G.-W. Search for Related Content PubMed PubMed Citation Articles by Au, W.-K. Articles by He, G.-W.