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Aneurysm of the Sinus of Valsalva with Coexistent Coronary Atherosclerosis
Aneurysm of the Sinus of Valsalva with Coexistent Coronary Atherosclerosis Probal K. Ghosh, F.R.C.S.(E), M.Ch., Hylton I. Miller, M.D., and Bernard0 A. Vidne, M.D.
ABSTRACT Unruptured aneurysm of the sinus of Valsalva coexistent with extensive coronary atherosclerosis was noted in a 65-year-old man. He underwent transaortic patch repair of the aneurysm and quadruple aortocoronary bypass. The essential features of adequate management of this association are discussed. The incidence of aneurysm of the sinus of Valsalva (ASV) among all congenital heart diseases ranges from 2% in the Orient [l]to 0.31% in the western world [2]. Anginal pain has been reported as a feature of unruptured ASV with dissection of the interventricular septum [3]. Kerber and colleagues [4] described a patient with unruptured type I ASV with right ventricular outflow tract obstruction mimicking a myocardial infarction. This patient's angina was thought to be due to increased right ventricular work secondary to the outflow tract obstruction or interference with the right coronary flow by the aneurysm, or both. We report the surgical treatment of a patient with extensive coronary atherosclerosis coexistent with a large unruptured ASV partially dissecting the interventricular septum and protruding into the right ventricle. In reviewing the English-language literature, we could not find another report of coexistence of these two conditions. In 1978, a 65-year-old man was admitted to another hospital with chest pain. Results of investigations at that time were inconclusive. Five years later he experienced chest pain radiating to the neck, shortness of breath on mild exertion, and a nonproductive cough. His condition was diagnosed as angina pectoris, and he was treated with oxprenolol hydrochloride, isosorbide dinitrate, dipyridamole, and nitroglycerin. O n admission, his blood pressure was 140/80 mm Hg and his heart rate was 70 beats per minute and regular. First heart sound was diminished with a fourth sound at the apex. A grade 3/6 midsystolic murmur was heard at the apex, radiating to the axilla and the left sternal border. The electrocardiogram showed left ventricular hypertrophy. An ergometric stress test was positive at From the Departments of Cardiology and Thoracic and Cdrdiovascular Surgery, lchilov Hospital, Tel-Aviv Medical Center and Sacklcr School of Medicine, Tel Aviv University, Tel Aviv, Israel. Accepted for publication July 2, 1984. Address reprint requests to Dr. Vidne, Dcyartmcnl o f Thoracic and Cardiovascular Surgery, lchilov Hospital, 6 Weizmann St, Tel Aviv 64239, Israel.
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maximum effort with dyspnea and ST depression of 1.5 mm in leads V5 and V6 at a heart rate of 130 bpm. Mmode and two-dimensional echocardiography indicated normal left ventricular cavity and aneurysmal dilatation of the anterior aortic sinus encroaching into the right ventricular outflow tract. Results of hemodynamic studies were unremarkable. Digital subtraction aortography demonstrated an aneurysm of the right sinus of Valsalva with a wide neck and a long-axis dimension of 5 cm. No aortic regurgitation was seen (Fig 1). Coronary angiography revealed marked stenoses in the left main coronary, left anterior descending, circumflex, and right coronary arteries (Figs 2, 3). Repair of ASV and aortocoronary bypass was performed on cardiopulmonary bypass using hypothermia to 23°C and crystalloid cardioplegia with topical cooling. The heart looked normal except for the origin of the right coronary artery, which was dilated, tortuous, and distorted. The aneurysm could not be seen or felt from the outer aspect. Quadruple distal coronary arterysaphenous vein anastomoses were performed to the first obtuse marginal branch, the left anterior descending coronary artery sequential to the diagonal branch, and the posterior descending arteries. An oblique aortotomy revealed a wide-necked type I1 ASV originating from the middle of the right aortic sinus. The outer margin ran very close to the inferior border of the right coronary ostium. The ASV had partially dissected through the interventricular septum and protruded into the right ventricle. There was no prolapse or fibrotic change of the aortic cusps nor any feature of past endocarditis. The neck of the aneurysm was oval and measured 2.5 x 1.5 cm (Fig 4). To close the neck, an oval Dacron patch was stitched circumferentially with 4-0 Prolene to the margin; this prevented distortion of the cuspal anatomy. Interrupted sutures were used on the cuspal border of the patch. Its annular border was stitched with continuous suture skirting the lower border of the right coronary ostium, which lay above the suture line and appeared patent. Proximal aortosaphenous anastomoses were performed above the aortotomy suture line. The patient was easily weaned off cardiopulmonary bypass. Postoperative recovery was uneventful. Seven months postoperatively he remained asymptomatic. Transvenous digital subtraction aortography showed a normal aortic root with no valvular regurgitation. Echocardiography demonstrated an echogenic patch in the anterior aortic wall. The shrunken wall of the obliterated aneurysm was seen in the right ventricular outflow tract, which was of normal size.
580 The Annals of Thoracic Surgery Vol 39 No 6 June 1985
Fig I , Digital subtraction nortograin (right anterior oblique uie7u) demonstrates a large aneurysm of the sinus of Valsalva from the riglit coronary sinrrs.
Fig 3 . Selective left coronary angiogram (right anterior oblique view) shows a 50% occlusion of the left main coronary artery, two stenoses in the proximal and the middle thirds of the left anterior descending arteny, and high-grade serial stenoses in the proximal arid middle circ i n n ~ e artery. x
LC A
RCA
Fig 2 . Selective right coronary angiograin (40-degree left anterior oblique v i m ) demonstrates high-grade stenoses in its proximal and distal thirds. The contrast mediugi spill outlines the aneurysm of the sinus of Valsalva and shows its relationship to the right coronary system.
Comment Hope first described the aneurysm of the sinus of Valsalva in 1839 [ 5 ] .Until recently, surgeons preferred not to repair an unruptured, asymptomatic ASV [6, 71. Although there are few clinical manifestations before the aneurysm ruptures, the importance of repairing an unruptured ASV has been recognized and recommended [4, 81. The natural history of unruptured ASV is unknown. The reported associations are dissection of the interventricular septum [3], aortoventricular discontinuity [9], ventricular tachycardia [lo], atrioventricular
A B Fig 4. (A) Coronal plane view showing the course of the aneurysm of the sinus of Valsalva dissecting through the interventricular septum and encroaching into the right ventricle. fB) Vie70 tlirorrgh the aortotomy showing an oval Dacron patch stitched circirmferenfially to the neck of the aneurysm of the sinus of Valsalva. (LCA = left coronary artery; RCA = right coronar~artey.)
or bundle-branch block [ll],and emboli [12]. We agree with Sakakibara and Konno [13], who advise elective repair of an unruptured asymptomatic ASV, even when detected incidentally. The right coronary sinus is the most common site of origin of ASV. Its incidence varies from 64 to 89% [6, 111. Probable etiological factors and associated lesions have been extensively discussed in the literature [14-16]. Surprisingly, the coexistence of coronary atherosclerosis with ASV has not been noted, even among the patients in the age group prone to this condition. Taguchi and coworkers [ll]anatomically classified ASVs into 6 main types and 16 subtypes. Our patient belonged to type 11. The anatomy, direction, and dimensions of the ASV constitute important technical determinants in choosing
581
Case Report: Ghosh, Miller, and Vidne: ASV with CAD
between patch repair and direct closure with interrupted mattress sutures. Aortocoronary bypass has been used sparingly in the treatment of ASV [lo, 171. In both of these reports, the coronary flow was compromised proximally by the aneurysm. Although the origin of the right coronary artery was distorted in our patient, atherosclerotic narrowing of its middle and distal third mandated saphenous vein bypass. In general, we agree with others [7, 181 that the transaortic approach is the safest route o f closing the neck of the aneurysm. Tanabe and colleagues [l]have recommended a double approach through the involved cardiac chamber and the aorta. In most instances, simple stitch closure of the neck is inadequate and is likely to give way to recurrence, possibly caused by weakness of the supporting tissue of the annulus. A patch repair has the further advantage of not distorting the cuspal anatomy; thus, it prevents the possibility of postoperative aortic regurgitation. In the presence of prolapsing cusps and fibrotic changes, especially in older patients, aortic valve replacement has been advocated [ 11J .
References 1. Tanabe T, Yokota A, Sugie 5: Surgical treatment of aneu2. 3. 4. 5.
rysms of the sinus of Valsalva. Ann Thorac Surg 27:133, 1979 Danielson GK: Discussion of Pan-Chih, Tsao C-H, ChenChun, Liu C-F: surgical treatment of the ruptured aneurysm of the aortic sinuses. Ann Thorac Surg 32:162, 1981 Chen WWC, Tai YT: Dissection of interventricular septum by aneurysm of sinus of Valsalva. Br Heart J 50:293, 1983 Kerber RE, Ridges ID, Kriss JP, et al: Unruptured ancurysm of the sinus of Valsalva producing right ventricular outflow obstruction. Am J Med 53:775, 1972 Oram S, East T: Rupture of aneurysms of aortic sinus (of
Valsalva) into the right side of the heart. Br Heart J 17:541, 1955 6. Meyer J, Wukasch DC, Hallman GL, Cooky DA: Aneurysm and fistula of the sinus of Valsalva. Ann Thorac Surg 19:170, 1975 7. Howard RJ, Moller J, Castaneda AR, et al: Surgical correction of sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg 66:420, 1973 8. Mayer JH, Holder TM, Canent RV: Isolated unruptured sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg 69:429, 1975 9. Yarnoz MD, Weber DO, Richman A, Del Mastro P: Repair of sinus of Valsalva aneurysm associated with aortoventricular discontinuity. Ann Thorac Surg 33:290, 1982 10 Raizes GS, Smith HC, Vliestra RE, Puga FJ: Ventricular tachycardia secondary to aneurysm of sinus of Valsalva. J Thorac Cardiovasc Surg 78:110, 1979 11 Taguchi K, Sasaki N, Matsuura Y, Uemura R: Surgical correction of aneurysm of the sinus of Valsalva. Am J Cardiol 23:180, 1969 12 Bjark VO, Edhag 0: Aortic sinus aneurysm: report of an unusual case. Scand J Thorac Cardiovasc Surg 6:136, 1972 13 Sakakibara S, Konno S : Congenital aneurysms of the sinus of Valsalva: a clinical study. Am Heart J 63:708, 1962 14 Spooner EW, Dunn JM, Behrendt DM: Aortico-left ventricular tunnel and sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg 75:232, 1978 15 Edwards JE, Burchell HB: Pathologic anatomy of deficiencies between the aortic root and the heart, including aortic sinus aneurysms. Thorax 12:125, 1957 16. Van Praagh R, McNamara JJ: Anatomic types of ventricular septa1 defects with aortic insufficiency. Am Heart J 75:604, 1968 17. Williams TG, Williams BT: Isolated unruptured aneurysm of the left coronary sinus o f Valsalva. Ann Thorac Surg 35:556, 1983 18. Henze A, Huttunen H, Bjdrk VO: Ruptured sinus of Valsalva aneurysms. Scand J Thorac Cardiovasc Surg 17:249, 1983
ABSTRACT Unruptured aneurysm of the sinus of Valsalva coexistent with extensive coronary atherosclerosis was noted in a 65-year-old man. He underwent transaortic patch repair of the aneurysm and quadruple aortocoronary bypass. The essential features of adequate management of this association are discussed. The incidence of aneurysm of the sinus of Valsalva (ASV) among all congenital heart diseases ranges from 2% in the Orient [l]to 0.31% in the western world [2]. Anginal pain has been reported as a feature of unruptured ASV with dissection of the interventricular septum [3]. Kerber and colleagues [4] described a patient with unruptured type I ASV with right ventricular outflow tract obstruction mimicking a myocardial infarction. This patient's angina was thought to be due to increased right ventricular work secondary to the outflow tract obstruction or interference with the right coronary flow by the aneurysm, or both. We report the surgical treatment of a patient with extensive coronary atherosclerosis coexistent with a large unruptured ASV partially dissecting the interventricular septum and protruding into the right ventricle. In reviewing the English-language literature, we could not find another report of coexistence of these two conditions. In 1978, a 65-year-old man was admitted to another hospital with chest pain. Results of investigations at that time were inconclusive. Five years later he experienced chest pain radiating to the neck, shortness of breath on mild exertion, and a nonproductive cough. His condition was diagnosed as angina pectoris, and he was treated with oxprenolol hydrochloride, isosorbide dinitrate, dipyridamole, and nitroglycerin. O n admission, his blood pressure was 140/80 mm Hg and his heart rate was 70 beats per minute and regular. First heart sound was diminished with a fourth sound at the apex. A grade 3/6 midsystolic murmur was heard at the apex, radiating to the axilla and the left sternal border. The electrocardiogram showed left ventricular hypertrophy. An ergometric stress test was positive at From the Departments of Cardiology and Thoracic and Cdrdiovascular Surgery, lchilov Hospital, Tel-Aviv Medical Center and Sacklcr School of Medicine, Tel Aviv University, Tel Aviv, Israel. Accepted for publication July 2, 1984. Address reprint requests to Dr. Vidne, Dcyartmcnl o f Thoracic and Cardiovascular Surgery, lchilov Hospital, 6 Weizmann St, Tel Aviv 64239, Israel.
579
maximum effort with dyspnea and ST depression of 1.5 mm in leads V5 and V6 at a heart rate of 130 bpm. Mmode and two-dimensional echocardiography indicated normal left ventricular cavity and aneurysmal dilatation of the anterior aortic sinus encroaching into the right ventricular outflow tract. Results of hemodynamic studies were unremarkable. Digital subtraction aortography demonstrated an aneurysm of the right sinus of Valsalva with a wide neck and a long-axis dimension of 5 cm. No aortic regurgitation was seen (Fig 1). Coronary angiography revealed marked stenoses in the left main coronary, left anterior descending, circumflex, and right coronary arteries (Figs 2, 3). Repair of ASV and aortocoronary bypass was performed on cardiopulmonary bypass using hypothermia to 23°C and crystalloid cardioplegia with topical cooling. The heart looked normal except for the origin of the right coronary artery, which was dilated, tortuous, and distorted. The aneurysm could not be seen or felt from the outer aspect. Quadruple distal coronary arterysaphenous vein anastomoses were performed to the first obtuse marginal branch, the left anterior descending coronary artery sequential to the diagonal branch, and the posterior descending arteries. An oblique aortotomy revealed a wide-necked type I1 ASV originating from the middle of the right aortic sinus. The outer margin ran very close to the inferior border of the right coronary ostium. The ASV had partially dissected through the interventricular septum and protruded into the right ventricle. There was no prolapse or fibrotic change of the aortic cusps nor any feature of past endocarditis. The neck of the aneurysm was oval and measured 2.5 x 1.5 cm (Fig 4). To close the neck, an oval Dacron patch was stitched circumferentially with 4-0 Prolene to the margin; this prevented distortion of the cuspal anatomy. Interrupted sutures were used on the cuspal border of the patch. Its annular border was stitched with continuous suture skirting the lower border of the right coronary ostium, which lay above the suture line and appeared patent. Proximal aortosaphenous anastomoses were performed above the aortotomy suture line. The patient was easily weaned off cardiopulmonary bypass. Postoperative recovery was uneventful. Seven months postoperatively he remained asymptomatic. Transvenous digital subtraction aortography showed a normal aortic root with no valvular regurgitation. Echocardiography demonstrated an echogenic patch in the anterior aortic wall. The shrunken wall of the obliterated aneurysm was seen in the right ventricular outflow tract, which was of normal size.
580 The Annals of Thoracic Surgery Vol 39 No 6 June 1985
Fig I , Digital subtraction nortograin (right anterior oblique uie7u) demonstrates a large aneurysm of the sinus of Valsalva from the riglit coronary sinrrs.
Fig 3 . Selective left coronary angiogram (right anterior oblique view) shows a 50% occlusion of the left main coronary artery, two stenoses in the proximal and the middle thirds of the left anterior descending arteny, and high-grade serial stenoses in the proximal arid middle circ i n n ~ e artery. x
LC A
RCA
Fig 2 . Selective right coronary angiograin (40-degree left anterior oblique v i m ) demonstrates high-grade stenoses in its proximal and distal thirds. The contrast mediugi spill outlines the aneurysm of the sinus of Valsalva and shows its relationship to the right coronary system.
Comment Hope first described the aneurysm of the sinus of Valsalva in 1839 [ 5 ] .Until recently, surgeons preferred not to repair an unruptured, asymptomatic ASV [6, 71. Although there are few clinical manifestations before the aneurysm ruptures, the importance of repairing an unruptured ASV has been recognized and recommended [4, 81. The natural history of unruptured ASV is unknown. The reported associations are dissection of the interventricular septum [3], aortoventricular discontinuity [9], ventricular tachycardia [lo], atrioventricular
A B Fig 4. (A) Coronal plane view showing the course of the aneurysm of the sinus of Valsalva dissecting through the interventricular septum and encroaching into the right ventricle. fB) Vie70 tlirorrgh the aortotomy showing an oval Dacron patch stitched circirmferenfially to the neck of the aneurysm of the sinus of Valsalva. (LCA = left coronary artery; RCA = right coronar~artey.)
or bundle-branch block [ll],and emboli [12]. We agree with Sakakibara and Konno [13], who advise elective repair of an unruptured asymptomatic ASV, even when detected incidentally. The right coronary sinus is the most common site of origin of ASV. Its incidence varies from 64 to 89% [6, 111. Probable etiological factors and associated lesions have been extensively discussed in the literature [14-16]. Surprisingly, the coexistence of coronary atherosclerosis with ASV has not been noted, even among the patients in the age group prone to this condition. Taguchi and coworkers [ll]anatomically classified ASVs into 6 main types and 16 subtypes. Our patient belonged to type 11. The anatomy, direction, and dimensions of the ASV constitute important technical determinants in choosing
581
Case Report: Ghosh, Miller, and Vidne: ASV with CAD
between patch repair and direct closure with interrupted mattress sutures. Aortocoronary bypass has been used sparingly in the treatment of ASV [lo, 171. In both of these reports, the coronary flow was compromised proximally by the aneurysm. Although the origin of the right coronary artery was distorted in our patient, atherosclerotic narrowing of its middle and distal third mandated saphenous vein bypass. In general, we agree with others [7, 181 that the transaortic approach is the safest route o f closing the neck of the aneurysm. Tanabe and colleagues [l]have recommended a double approach through the involved cardiac chamber and the aorta. In most instances, simple stitch closure of the neck is inadequate and is likely to give way to recurrence, possibly caused by weakness of the supporting tissue of the annulus. A patch repair has the further advantage of not distorting the cuspal anatomy; thus, it prevents the possibility of postoperative aortic regurgitation. In the presence of prolapsing cusps and fibrotic changes, especially in older patients, aortic valve replacement has been advocated [ 11J .
References 1. Tanabe T, Yokota A, Sugie 5: Surgical treatment of aneu2. 3. 4. 5.
rysms of the sinus of Valsalva. Ann Thorac Surg 27:133, 1979 Danielson GK: Discussion of Pan-Chih, Tsao C-H, ChenChun, Liu C-F: surgical treatment of the ruptured aneurysm of the aortic sinuses. Ann Thorac Surg 32:162, 1981 Chen WWC, Tai YT: Dissection of interventricular septum by aneurysm of sinus of Valsalva. Br Heart J 50:293, 1983 Kerber RE, Ridges ID, Kriss JP, et al: Unruptured ancurysm of the sinus of Valsalva producing right ventricular outflow obstruction. Am J Med 53:775, 1972 Oram S, East T: Rupture of aneurysms of aortic sinus (of
Valsalva) into the right side of the heart. Br Heart J 17:541, 1955 6. Meyer J, Wukasch DC, Hallman GL, Cooky DA: Aneurysm and fistula of the sinus of Valsalva. Ann Thorac Surg 19:170, 1975 7. Howard RJ, Moller J, Castaneda AR, et al: Surgical correction of sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg 66:420, 1973 8. Mayer JH, Holder TM, Canent RV: Isolated unruptured sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg 69:429, 1975 9. Yarnoz MD, Weber DO, Richman A, Del Mastro P: Repair of sinus of Valsalva aneurysm associated with aortoventricular discontinuity. Ann Thorac Surg 33:290, 1982 10 Raizes GS, Smith HC, Vliestra RE, Puga FJ: Ventricular tachycardia secondary to aneurysm of sinus of Valsalva. J Thorac Cardiovasc Surg 78:110, 1979 11 Taguchi K, Sasaki N, Matsuura Y, Uemura R: Surgical correction of aneurysm of the sinus of Valsalva. Am J Cardiol 23:180, 1969 12 Bjark VO, Edhag 0: Aortic sinus aneurysm: report of an unusual case. Scand J Thorac Cardiovasc Surg 6:136, 1972 13 Sakakibara S, Konno S : Congenital aneurysms of the sinus of Valsalva: a clinical study. Am Heart J 63:708, 1962 14 Spooner EW, Dunn JM, Behrendt DM: Aortico-left ventricular tunnel and sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg 75:232, 1978 15 Edwards JE, Burchell HB: Pathologic anatomy of deficiencies between the aortic root and the heart, including aortic sinus aneurysms. Thorax 12:125, 1957 16. Van Praagh R, McNamara JJ: Anatomic types of ventricular septa1 defects with aortic insufficiency. Am Heart J 75:604, 1968 17. Williams TG, Williams BT: Isolated unruptured aneurysm of the left coronary sinus o f Valsalva. Ann Thorac Surg 35:556, 1983 18. Henze A, Huttunen H, Bjdrk VO: Ruptured sinus of Valsalva aneurysms. Scand J Thorac Cardiovasc Surg 17:249, 1983