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Ann Thorac Surg 2002;74:1621-1624
© 2002 The Society of Thoracic Surgeons

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Original article: cardiovascular

Ruptured sinus of valsalva aneurysm: a Beijing experience

^a Department of Surgery, Cardiovascular Institute and Fu Wai Hospital, Peking Union Medical College, Chinese Academy of Medical Sciences, Beijing, China

Accepted for publication July 2, 2002.

* Address reprint requests to Dr Dong, Department of Surgery, Fu Wai Hospital, 167 North Li-Shi Rd, Beijing 100037, P.R. China.
e-mail: dr_dong_chao{at}yahoo.com.cn

	Abstract

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BACKGROUND: Ruptured sinus of Valsalva aneurysm (RSVA) is relatively common in oriental patients. We retrospectively analyzed 67 patients receiving repair of RSVA in a Beijing hospital over 5 years.

METHODS: Between October 1, 1996 and September 30, 2001, at Fu Wai Hospital, 67 patients with RSVA underwent surgical repair, 0.78% of all congenital open-heart operations. Forty-four were male and 23 female. Age ranged from 2 to 57 years old (mean 32 ± 10 years). The RSVA originated in the right (n = 52) or noncoronary (n = 15) sinus. Rupture into the right ventricle was most common (n = 39) with 26 going to the right atrium and two to the left ventricle. Associated cardiovascular lesions were ventricular septal defect (n = 32) and aortic valve incompetence (n = 12). Repair was achieved through an incision in the cardiac chamber of the fistula exit in 61 patients. Aortotomy was used in three patients and both routes were used in three patients. The sinus of Valsalva was repaired with either a patch (n = 63) or direct sutures (n = 4). The aortic valve was replaced in 12 patients.

RESULTS: All but 1 patient (n = 66) survived the 30-day operative interval. One patient died of an anticoagulation complication 2 months after the operation. Late complications included residual shunt (n = 2), peri-prosthetic leakage (n = 1), and aortic incompetence (n = 1).

CONCLUSIONS: In this relatively high-risk population, repair of RSVA can be achieved with satisfactory early results.

	Introduction

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Ruptured sinus of Valsalva aneurysm (RSVA) is more common in the oriental population [1]. Uncorrected these intracardiac shunts and the frequently associated cardiac lesions cause a preventable deterioration in heart function. We retrospectively analyzed 67 patients receiving repair of RSVA in a Beijing Hospital over 5 years.

	Material and methods

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Between October 1, 1996 and September 30, 2001, 67 Chinese patients with RSVA underwent operation at Fu Wai Hospital. This experience constitutes 0.42% of all the heart operations (n = 15,994) and 0.78% of the congenital open-heart operations (n = 8,621) during this 5-year interval. Forty-four were male and 23 were female. Age ranged from 2 to 57 years old (mean 32 ± 10 years; Table 1), and body weight ranged from 13 to 96 kg (mean 62 ± 12 kg). Preoperative signs and symptoms with their duration are shown in Table 2. Acute symptoms occurred after a motor vehicle accident in one patient and during exertion in one. Preoperative New York Heart Association (NYHA) Class is shown in Table 3. Preoperative left ventricular end diastolic dimension (LVEDD) is shown in Figure 1.

View larger version (31K): [in this window] [in a new window]   Fig 1. Preoperative (Pre) and postoperative (Post) left ventricular end-diastolic dimension (LVEDD) of 66 patients with repaired ruptured sinus of Valsalva aneurysm in Beijing. *The last postoperative echocardiographic LVEDD was measured at 0–42 months (4.5 ± 8.2) after operation. Student’s t-test, n = 66, t = 12.247, and p < 0.001.

Preoperative systemic pulse pressure ranged from 40 to 170 mm Hg (mean 76 ± 25 mm Hg). Before operation, a complete diagnosis was made in 42 patients (62.7%). RSVA was diagnosed by echocardiography in 60 patients (90%). In the remaining 7 patients RSVA was not suspected but found during operation. Ventricular septal defect (VSD) was not diagnosed preoperatively in 18 of 32 patients (56%). Confirmatory cardiac angiogram was performed in one patient. Patients more than 50 years old by protocol undergo preoperative coronary angiography. One of these three patients was in such critical condition that coronary angiogram was not done.

At operation cardiopulmonary bypass (39 to 176 minutes., mean 82 ± 30 minutes.) was used with moderate hypothermia (25.9° to 33.9°C, mean 29.9° ± 1.7°C). After aortic cross clamping (15 to 120 minutes., mean 54 ± 25 minutes.), the RSVA fistula was visualized through the cardiac chamber receiving fistula blood flow, i.e., the right atrium (RA) or the right ventricular out-flow tract (RVOT). When the aortic valve was competent and the aneurysm long enough, the fistula was clamped before cardioplegia, cold crystalloid (n = 13) or blood (n = 54) cardioplegia, was administered. Otherwise, cardioplegia was infused directly into the coronary ostia via an aortotomy. The aortic valve, sinuses of Valsalva, and coexisted lesions were inspected and repaired.

Table 4 shows the frequency of RSVA origin, exit site and presence of coexistent VSD. When the fistula exited into the RVOT (n = 38), 29 had subarterial VSD whereas 2 had a conoventricular VSD. When the aneurysm ruptured into the RA, only one perimembranous VSD coexisted. No VSD was found when the aneurysm ruptured into either the left ventricle (LV) or the ventricular membranous septum. In all 29 patients with subarterial VSD, the VSD was separated from the fistula by an aortic valve annulus fibrous band. In both patients with conoventricular VSD, the VSD was separated from the fistula by a narrow myocardial bundle. When the fistula entered the right atrium (RA) and associated with a perimembranous VSD, the noncoronary aortic leaflet hinge line separated the fistula and the VSD.

Aortic valve leaflet abnormalities were found in 15 patients. Two patients had a bicuspid aortic valve. The right leaflet was abnormal in 15 patients, while the left leaflet was abnormal in seven and the noncoronary leaflet was abnormal in five. The RSVA originated from the right sinus of Valsalva in all 15 of these patients and exited into the RVOT in 14 and into the LV in a single patient.

Aortic valve incompetence (AI) was present in 12 patients: one mild, eight moderate, one severe, and two extremely severe. The fistula exited into the RVOT in 11 of these 12 patients. Eleven patients had a long history of symptoms. Eight patients, having a mean age of 30.1 ± 6.3 years, had a coexisting subarterial VSD. A heart murmur was the principle finding 18 and 20 years before operation in two patients, while sudden onset of exertional palpitation and shortness of breath occurred 20 years before operation in one patient. The remaining patient with a RSVA into the LV had symptoms for 11 months.

Associated cardiovascular lesions included: RVOT muscular stenosis (n = 4), bicuspid aortic valve (n = 2), discrete subaortic stenotic membrane (n = 2), sinus of Valsalva aneurysm of right sinus (n = 1), aortic stenosis (n = 1), prior VSD repair (n = 1), mitral regurgitation (n = 1), and tricuspid regurgitation (n = 1).

Repair of the RSVA fistula was done through an incision in the heart chamber of fistula exit. An aortotomy was made in 29 patients, 12 of whom had aortic valve replacement (AVR). In 34 patients, after excising the aneurysm and reinforcing the sinus wall from outside with a patch, the base of the fistula was closed: pledged interrupted sutures in 21, continuous suture in 12, and a purse string suture in one. In 24 patients, the base of the fistula was closed with a patch from the outside. Simply suture closure from the outside was used in three patients. A patch from the outside and suture closure from the inside was done in three patients. Repair of the fistula base from the inside alone was done in three patients. A Dacron patch was employed in 58 patients, autogenous pericardium lined Dacron patch in four, and autogenous pericardial patch in one patient. When a VSD was present, a single patch was used to cover both the fistula base and the VSD. Simultaneous procedures included RVOT stenosis resection (n = 4), discrete subaortic stenotic membrane resection (n = 2), mitral valve repair (n = 1), and tricuspid valve repair (n = 1).

Infectious endocarditis was present in four patients with a fistula into the RVOT and a subarterial VSD. Vegetation on the aortic valve leaflets was found in two patients, in the aneurysm sac with an aortic valve leaflet perforation in one, and in the aneurysm sac in one. Repair of aneurysm, VSD closure and AVR were done in the three patients who had aortic valve leaflet abnormalities.

In all patients receiving AVR, a mechanical valve prosthesis was implanted including: six St. Jude (St. Jude Medical, St. Paul, MN), four Medtronic-Hall (Medtronic, Minneapolis, MN), and two Sorin disk (Sorin Biomedica Cardio S.P.A., Saluggia, Italy).

If periodic postoperative hospital visits by us were not available, follow-up telephone calls were made to the patient by the Outpatient Department.

	Results

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Sixty-six patients survived operation. In the patient who succumbed, an aneurysm ruptured acutely into the RA leading to heart failure and chronic renal dysfunction. Successful emergent repair was achieved with an uneventfully recovery for the first two postoperative days. Five days postoperatively, acute peritonitis, septic shock and acute renal failure developed. The patient died on the eighth postoperative day.

Postoperative complications included: bleeding (n = 2), skin wound infection (n = 2), acute renal failure (n = 2), acute peritonitis (n = 1), blood transfusion reaction (n = 1), and persistent VSD (n = 1). Postoperative hospital stay was 7 to 28 days (mean 10.3 ± 4.6 days). Change in LVEDD for each patient is shown in Figure 1.

Fifty-one of the survivors had follow-up 1 to 58 months (mean 25.5 ± 18.1 months) postoperatively. We examined 36 patients and 15 patients were reached by telephone. NYHA Class at the last following-up is shown in Table 3.

Late complications in one patient each included: residual VSD, residual fistula, mild AI, and mild peri-prosthesis leakage. At 19 months after operation, the patient with a residual VSD was NYHA Class III. Reoperation has been recommended. The remaining 3 patients are NYHA Class I at 20, 25 and 29 postoperative months respectively. No fistulas have recurred and no additional operations have been done.

One patient developed atrial fibrillation 20 months after RSVA closure. Radial frequency ablation has effectively treated this arrhythmia. A patient with chronic renal failure is NYHA Class I, has shown no recovery of renal function.

Eleven of 12 AVR patients have been followed with one death from an anticoagulation complication two months after operation. No additional prosthesis related complications have been noted.

	Comment

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The incidence of acute onset of the symptoms in patients with RSVA is about 35% [2]. In our series, however, we believe the sudden development of palpitation or chest pain in 58% represent a higher percentage of acute onset. The origin, exit, coexistent VSD and coexistent AI do not help us explain this higher percentage.

When an aneurysm ruptures into the RVOT, a coexistent subarterial VSD is likely [1, 3]. Exceptions can be explained by the following. If the coexistent subarterial VSD is small and the diameter of the aneurysm wall increases, it may contact the opposite margin of the VSD. The VSD is then closed by adhesion. This rationale may hold true if a patient has a long history of a heart murmur and a fistula entering the RVOT is the only operative finding. This occurred in two of our patients.

We did not encounter any patient having the fistula enter the ventricular septum, a special situation for which the strategy and technique of surgical repair has been different [4].

We routinely performed repairs through an incision in the heart chamber of fistula exit. Good exposure can be achieved with repair of the RSVA and correction of coexistent lesions easily achieved. This study did not reveal that the side of the repair/reinforcement coronary sinus is important. Residual shunts and aneurysm recurrence can be eliminated by patch reinforcement of the sinus wall with sutures place in normal tissue. Aortotomy may be necessary when abnormalities of the aortic valve leaflets are present pre- or intraoperatively following repair.

Principles of repairing RSVA include the following. Sutures should always be placed in normal tissue. Sutures in thin and degenerative sinus or aneurysmal wall should be avoided. The patch should be large enough to reinforce the entire involved coronary sinus wall. One patch may be used to repair both the fistula and the coexistent VSD, if present. The hinge line of an involved aortic valve cusp should always be kept at its proper functional location on the patch. Aortic valve distortion should be avoided. The possibility of injury to the aortic valve leaflets or coronary ostia must be kept in mind and avoided. Direct suture closure of the RSVA does not meet these criteria [5, 6].

The probability of coexistent AI increases if the aneurysm ruptures into the RVOT, particularly when a subarterial VSD exists [7, 8]. We did not see AI when the aneurysm ruptured into the RA. When a RSVA and a VSD coexistent, the sinus wall and aortic valve annulus are deprived of endocardial muscular support. Hemodynamically, the Bernoulli effect of the interventricular shunt and aorto-ventricular shunt may explain [1, 8, 9] an adverse influence on the movement of the aortic valve. As we have observed, the subarterial VSD may be partially obscured by a prolapsing aortic leaflet. During systole, the Bernoulli effect of flow across the VSD may pull an aortic valve leaflet, causing it to prolapse. This may partially explain the development of VSD with the subsequent occurrence of AI syndrome [9]. Infective endocarditis may also play an important role.

Aortic valve replacement using a prosthesis was done in this series instead of employing aortic valvuloplasty techniques for patients with AI. After prolonged hemodynamic trauma resulting from the VSD and the fistula, the involved aortic leaflets had become fibrotic, retracted and deformed. This precluded adequate repair and necessitated prosthesis implantation. Impaired preoperative heart function reduces cardiac tolerance for postoperative residual AI. Reoperation for a failed aortic valve repair exceeds our patients’ tolerance for postoperative long-range outcomes. As greater experience with aortic valve repair is gathered, our economy improves, and patients seek the help of physicians earlier in their disease process, valve repair for AI in RSVA patients will likely become more common.

Follow-up was the most difficult aspect of this series. Family practitioners are currently not available in China. Although the expense for a patient’s outpatient follow-up is not large, the economic and logistic burden for patients living thousands of miles from Beijing is limiting. Telephone service is not always available in the countryside. Many mailing addresses and telephone numbers may have changed due to rapid economic development and transitory nature of the population in the last decade and a half. Most important, long term followed up as a concept and necessary step after resolution of the acute process has not been well established in our population.

Conclusion
Repair of the sinus of Valsalva aneurysm rupture can be achieved with satisfactory early results in this population having a relatively high incidence in a developing economy.

	Acknowledgments

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The authors thank Dr Donald C. Watson for language editing this manuscript.

	References

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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. Kirklin J.W. Congenital aneurysm of the sinus of Valsalva. In: Kirklin J.W., Barratt-Boyes B.G., eds. Cardiac surgery, 2nd ed. Edinburgh, United Kingdom: Churchill Livingstone Inc, 1993:825-839.
3. Choudhary S.K., Bhan A., Sharma R., et al. Sinus of Valsalva aneurysms. 20 years’ experience. J Card Surg 1997;12:300-308.[Medline]
4. Wu Q.-Y. Surgical treatment of dissecting aneurysm of the interventricular septum. Ann Thorac Surg 1997;63:545-547.[Abstract/Free Full Text]
5. Abe T., Komatsu S. Surgical repair and long-term results in ruptured sinus of Valsalva aneurysm. Ann Thorac Surg 1988;46:520-525.[Abstract]
6. Barragry T.P., Ring W.S., Moller J.H., et al. 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]
7. Taguchi K., Sasaki N., Matsuura Y., Mura R. Surgical correction of aneurysm of the sinus of Valsalva: a report of 45 consecutive patients, including 8 with total replacement of the aortic valve. Am J Cardiol 1969;23:180-191.[Medline]
8. van Son J.A.M., Danielson G.K., Schaff H.V., et al. Long-term outcome of surgical repair of ruptured sinus of Valsalva aneurysm. Circulation 1994;90(Part 2):II20-II29.
9. Tatuno K., Konno S., Ando M., et al. Pathogenetic mechanisms of prolapsing aortic valve and aortic regurgitation associated with ventricular septal defect: anatomical, angiographic, and surgical considerations. Circulation 1973;48:1028-1037.[Abstract/Free Full Text]

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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 Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Dong, C. Articles by Tang, Y. Search for Related Content PubMed PubMed Citation Articles by Dong, C. Articles by Tang, Y. Related Collections Congenital - acyanotic