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Giant Coronary Artery Aneurysm Associated with Medial Mucoid Degeneration
Ann Thorac Surg 2009;87:933– 4
References 1. Crawford C, Nylin G. Congenital coarctation of aorta and its surgical treatment. J Thorac Surg 1945;14:347– 61. 2. Foster E. Reoperation for aortic coarctation. Ann Thorac Surg 1984;38:81–9. 3. Bergdahl L, Ljungquist A. Long-term results after repair of coarctation of the aorta by patch grafting. J Thor Cardiovasc Surg 1980;80:177– 81. 4. Ala-Kulju K, Heikkinen L. Aneurysms after patch graft aortoplasty for coarctation of the aorta: long-term results of surgical management. Ann Thorac Surg 1989;47:853– 6. 5. DeSanto A, Bills RG, King H, et al. Pathogenesis of aneurysm formation opposite prosthetic patches used for coarctation repair. J Thorac Cardiovasc Surg 1987;94:720 –3. 6. Roos-Hesselink JW, Schölzel BE, Heijdra RJ, et al. Aortic valve and aortic arch pathology after coarctation repair. Heart 2003;89:1074 –7. 7. von Kodolitsch Y, Aydin MA, Koschyk DH. Predictors of aneurysmal formation after surgical correction of aortic coarctation. J Am Coll Cardiol 2002;39:617–24. 8. White CW, Zoller RP. Left aortic dissection following repair of coarctation. The contribution of abnormal hemodynamics to median degeneration. Chest 1973;634:573–7.
Giant Coronary Artery Aneurysm Associated with Medial Mucoid Degeneration Tadahisa Sugiura, MD, Satoshi Saito, MD, Shinichiro Kihara, MD, Wataru Sato, MD, and Hiromi Kurosawa, MD Department of Cardiovascular Surgery, The Heart Institute of Japan, Tokyo Women’s Medical University, Shinjuku, Tokyo, Japan
A 52-year-old man underwent surgical repair for coronary arteriovenous fistula including patch closure of two Accepted for publication July 9, 2008. Address correspondence to Dr Sugiura, Department of Cardiovascular Surgery, The Heart Institute of Japan, Tokyo Women’s Medical University, 8-1 Kawada, Shinjuku, Tokyo, 162-8666, Japan; e-mail: ssugiura@ hij.twmu.ac.jp.
© 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
933
exitus of the fistula, double ligation of the dilated right coronary artery, and coronary bypass grafting to the right coronary artery with saphenous vein graft at the age of 25. Then, twenty-seven years later, a multi-detector computed tomographic scan revealed an aneurysmally dilated right coronary artery (4.8 cm), fistula, and saphenous vein graft. The aneurysm was resected and histologically mucoid degeneration in the media of the aneurysmal wall was detected. (Ann Thorac Surg 2009;87:933– 4) © 2009 by The Society of Thoracic Surgeons
C
oronary artery aneurysm is uncommon. Its cause is mainly atherosclerotic.
A 52-year-old man was first pointed out to have a heart murmur at the approximate age of 20, but he did not consult the doctor. He contracted bacterial endocarditis, which was cured with antibiotics at age 23. Cardiac catheterization was performed the next year to reveal a coronary arteriovenous fistula that originated from the right coronary artery (RCA) and drained into the right atrium; its left-to-right shunt ratio was 56%. A surgical repair was performed at our institute in the same year. Closure of two exitus of the fistula within the RA, double ligation of the aneurysmally dilated RCA (2 cm), and aortocoronary bypass grafting with a saphenous vein graft (SVG) were performed. A follow-up catheterization at the age of 40 revealed a recurrence of arteriovenous fistula at the RCA, an RCA aneurysm with a diameter of 1.5 cm, and grade II aortic regurgitation, but we had not operated because the SVG was patent and the left-toright shunt ratio was not significant (ie, the shunt ratio was 30%). Twelve years later, the patient was admitted to our hospital for an operation at the age of 52, because a multi-detector computed tomographic scan revealed an aneurysmally dilated RCA with a diameter of 4.8 cm. The chest roentgenogram was normal and electrocardiographic measurements showed no ST segment change. Cardiac catheterization showed an aneurysmally dilated RCA after a dilated fistula and a dilated SVG. The pulmonary-to-systemic blood flow ratio was 1.47, the ejection fraction was 52%, and grade II aortic regurgitation was detected. It was revealed that the SVG was patent, but there was 75% stenosis just distal to the anastomosis. Transesophageal echocardiography revealed two shunt flows from distal to the fistula into the RA. A multi-detector computed tomographic scan showed an aneurysmally dilated RCA with a diameter of 4.8 cm after a dilated exitus of the fistula into the right atrium, and a dilated SVG (Fig 1). Under the preparation of femoro-femoral bypass, a median sternotomy was performed. After harvesting the radial artery graft, cardiopulmonary bypass was established with ascending aortic perfusion and bi-caval drainage. The RA was incised and two bleeding points were detected near the previous exitus of the fistula, 0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2008.07.038
FEATURE ARTICLES
monly encountered in patients with previous coarctation repair. Aortic abnormalities may predispose to dilatations and dissection, which necessitates careful lifelong surveillance in all patients with coarctation of the aorta [7]. In a series of 149 patients with coarctation repair, a report by Simoom and colleagues [6] showed dilatation of the ascending aorta in 28% of patients, but they concluded that no relation between dilatation and hypertension [7]. Other series suggested a definite correlation between an ascending aortic aneurysm and hypertension. Possibility of intrinsic abnormality of the media, probably cystic median necrosis was also suggested [8]. Hence, continued clinical and radiological surveillance of coarctation repair is warranted In conclusion, clearly this group requires careful follow-up to identify those patients in whom complications can develop. We believe that the fourth time reoperation for coarctation of the aorta needing a Bentall’s procedure is not reported.
CASE REPORT SUGIURA ET AL GIANT CORONARY ARTERY ANEURYSM
934
CASE REPORT SUGIURA ET AL GIANT CORONARY ARTERY ANEURYSM
which had been closed 27 years before. They were directly closed with 4-0 polypropylene. An aortotomy was performed, and two holes with a diameter of 8 mm were detected in the right coronary cusp. It was presumably attributable to infective endocarditis. Aortic valve replacement was performed with a St. Jude Medical mechanical heart valve (23 mm). The radial artery graft was anastomosed to the middle portion of the RCA. An aneurysmally dilated RCA and SVG were resected together and were closed with a continuous suture. The defect of the aortic wall, including the ostium of the RCA and SVG was closed with a Xenomedica patch (Baxter Healthcare, Chicago, IL). Pathohistologic examination revealed mucoid degeneration in the media of the aneurysmal wall (Fig 2). The multi-detector computed tomographic scan after the operation showed the radial artery graft was patent and the RCA had a good run off.
Ann Thorac Surg 2009;87:933– 4
Fig 2. Microscopical observation of the media of the right coronary artery. Eosin-stained areas are mucoid degeneration. (Hematoxylin and eosin stain, original magnification ⫻100.)
Comment
FEATURE ARTICLES
The incidence of coronary artery aneurysm is reported to be 4.9% based on the Coronary Artery Surgery Study (CASS) registry [1]. Atherosclerosis is the most common cause of coronary artery aneurysm [2]. It can occur secondary to inflammatory or infectious disease (ie, Kawasaki disease, Takayasu disease, systemic lupus erythematosus, or polyarteritis nodosa, endocarditis, and syphilis), connective tissue disease (ie, Marfan’s syndrome, Ehlers-Danlos syndrome), metastatic tumors, or blunt trauma [3]. In this case, the patient did not suffer from systemic connective tissue disease. Histologically
mucoid degeneration of the RCA and SVG is believed to be a cause of aneurysmal dilatation. Bacterial endocarditis develops in patients with coronary artery fistula [4]. Bacterial endocarditis was believed to be a complication of coronary artery fistula in this patient. The combination of coronary artery aneurysm with arteriovenous fistula is rarer (0.2%) [5]. The case of coronary artery aneurysm of the RCA with arteriovenous fistula resulting from high blood flow from a highpressure chamber to a low-pressure chamber was previously reported [6]. Residual or recurrent fistulas were found in 4 of 41 patients (9.8%) who underwent coronary artery fistula operations. The incidence of recurrence was higher in the external plication or division of fistula group compared with the intracardiac closure group [7]. In this case, mucoid degeneration of the aneurysmal wall, as well as high blood flow resulted in giant coronary artery aneurysm.
References
Fig 1. Multi-detector computed tomographic image shows aneurysmally dilated right coronary artery, fistula, and saphenous vein graft. (RCA ⫽ right coronary artery; SVG ⫽ saphenous vein graft.)
1. Swaye PS, Fisher LD, Litwin P, et al. Aneurysmal coronary artery disease. Circulation 1983;67:134 – 8. 2. Moriyama Y, Hisatomi K, Shimokawa S, et al. Coronary artery aneurysm repaired with saphenous vein patch plasty. Ann Thorac Surg 1998;65:561–2. 3. Aqel RA, Zoghbi GJ, Iskandrian A. Spontaneous coronary artery dissection, aneurysm, and pseudoaneurysms: a review. Echocardiography 2004;21:175– 82. 4. Sapin R, Frantz E, Jain A, et al. Coronary artery fistula: an abnormality affecting all age groups. Medicine 1990;69:101–13. 5. Hurst JW, Schlant RC, Rackley CE, et al. The heart: arteries and veins, 7th ed. New York, NY: McGraw-Hill Inc, 1990: 1233– 4. 6. Sexena P, Konstantinov IE, Burstow D, et al. Surgical repair of a large coronary artery aneurysm with arteriovenous fistula. J Thorac Cardiovasc Surg 2006;131:1167– 8. 7. Cheung DL, Au WK, Cheung HH, et al. Coronary artery fistulas: long-term results of surgical correction. Ann Thorac Surg 2001;71:190 –5.
References 1. Crawford C, Nylin G. Congenital coarctation of aorta and its surgical treatment. J Thorac Surg 1945;14:347– 61. 2. Foster E. Reoperation for aortic coarctation. Ann Thorac Surg 1984;38:81–9. 3. Bergdahl L, Ljungquist A. Long-term results after repair of coarctation of the aorta by patch grafting. J Thor Cardiovasc Surg 1980;80:177– 81. 4. Ala-Kulju K, Heikkinen L. Aneurysms after patch graft aortoplasty for coarctation of the aorta: long-term results of surgical management. Ann Thorac Surg 1989;47:853– 6. 5. DeSanto A, Bills RG, King H, et al. Pathogenesis of aneurysm formation opposite prosthetic patches used for coarctation repair. J Thorac Cardiovasc Surg 1987;94:720 –3. 6. Roos-Hesselink JW, Schölzel BE, Heijdra RJ, et al. Aortic valve and aortic arch pathology after coarctation repair. Heart 2003;89:1074 –7. 7. von Kodolitsch Y, Aydin MA, Koschyk DH. Predictors of aneurysmal formation after surgical correction of aortic coarctation. J Am Coll Cardiol 2002;39:617–24. 8. White CW, Zoller RP. Left aortic dissection following repair of coarctation. The contribution of abnormal hemodynamics to median degeneration. Chest 1973;634:573–7.
Giant Coronary Artery Aneurysm Associated with Medial Mucoid Degeneration Tadahisa Sugiura, MD, Satoshi Saito, MD, Shinichiro Kihara, MD, Wataru Sato, MD, and Hiromi Kurosawa, MD Department of Cardiovascular Surgery, The Heart Institute of Japan, Tokyo Women’s Medical University, Shinjuku, Tokyo, Japan
A 52-year-old man underwent surgical repair for coronary arteriovenous fistula including patch closure of two Accepted for publication July 9, 2008. Address correspondence to Dr Sugiura, Department of Cardiovascular Surgery, The Heart Institute of Japan, Tokyo Women’s Medical University, 8-1 Kawada, Shinjuku, Tokyo, 162-8666, Japan; e-mail: ssugiura@ hij.twmu.ac.jp.
© 2009 by The Society of Thoracic Surgeons Published by Elsevier Inc
933
exitus of the fistula, double ligation of the dilated right coronary artery, and coronary bypass grafting to the right coronary artery with saphenous vein graft at the age of 25. Then, twenty-seven years later, a multi-detector computed tomographic scan revealed an aneurysmally dilated right coronary artery (4.8 cm), fistula, and saphenous vein graft. The aneurysm was resected and histologically mucoid degeneration in the media of the aneurysmal wall was detected. (Ann Thorac Surg 2009;87:933– 4) © 2009 by The Society of Thoracic Surgeons
C
oronary artery aneurysm is uncommon. Its cause is mainly atherosclerotic.
A 52-year-old man was first pointed out to have a heart murmur at the approximate age of 20, but he did not consult the doctor. He contracted bacterial endocarditis, which was cured with antibiotics at age 23. Cardiac catheterization was performed the next year to reveal a coronary arteriovenous fistula that originated from the right coronary artery (RCA) and drained into the right atrium; its left-to-right shunt ratio was 56%. A surgical repair was performed at our institute in the same year. Closure of two exitus of the fistula within the RA, double ligation of the aneurysmally dilated RCA (2 cm), and aortocoronary bypass grafting with a saphenous vein graft (SVG) were performed. A follow-up catheterization at the age of 40 revealed a recurrence of arteriovenous fistula at the RCA, an RCA aneurysm with a diameter of 1.5 cm, and grade II aortic regurgitation, but we had not operated because the SVG was patent and the left-toright shunt ratio was not significant (ie, the shunt ratio was 30%). Twelve years later, the patient was admitted to our hospital for an operation at the age of 52, because a multi-detector computed tomographic scan revealed an aneurysmally dilated RCA with a diameter of 4.8 cm. The chest roentgenogram was normal and electrocardiographic measurements showed no ST segment change. Cardiac catheterization showed an aneurysmally dilated RCA after a dilated fistula and a dilated SVG. The pulmonary-to-systemic blood flow ratio was 1.47, the ejection fraction was 52%, and grade II aortic regurgitation was detected. It was revealed that the SVG was patent, but there was 75% stenosis just distal to the anastomosis. Transesophageal echocardiography revealed two shunt flows from distal to the fistula into the RA. A multi-detector computed tomographic scan showed an aneurysmally dilated RCA with a diameter of 4.8 cm after a dilated exitus of the fistula into the right atrium, and a dilated SVG (Fig 1). Under the preparation of femoro-femoral bypass, a median sternotomy was performed. After harvesting the radial artery graft, cardiopulmonary bypass was established with ascending aortic perfusion and bi-caval drainage. The RA was incised and two bleeding points were detected near the previous exitus of the fistula, 0003-4975/09/$36.00 doi:10.1016/j.athoracsur.2008.07.038
FEATURE ARTICLES
monly encountered in patients with previous coarctation repair. Aortic abnormalities may predispose to dilatations and dissection, which necessitates careful lifelong surveillance in all patients with coarctation of the aorta [7]. In a series of 149 patients with coarctation repair, a report by Simoom and colleagues [6] showed dilatation of the ascending aorta in 28% of patients, but they concluded that no relation between dilatation and hypertension [7]. Other series suggested a definite correlation between an ascending aortic aneurysm and hypertension. Possibility of intrinsic abnormality of the media, probably cystic median necrosis was also suggested [8]. Hence, continued clinical and radiological surveillance of coarctation repair is warranted In conclusion, clearly this group requires careful follow-up to identify those patients in whom complications can develop. We believe that the fourth time reoperation for coarctation of the aorta needing a Bentall’s procedure is not reported.
CASE REPORT SUGIURA ET AL GIANT CORONARY ARTERY ANEURYSM
934
CASE REPORT SUGIURA ET AL GIANT CORONARY ARTERY ANEURYSM
which had been closed 27 years before. They were directly closed with 4-0 polypropylene. An aortotomy was performed, and two holes with a diameter of 8 mm were detected in the right coronary cusp. It was presumably attributable to infective endocarditis. Aortic valve replacement was performed with a St. Jude Medical mechanical heart valve (23 mm). The radial artery graft was anastomosed to the middle portion of the RCA. An aneurysmally dilated RCA and SVG were resected together and were closed with a continuous suture. The defect of the aortic wall, including the ostium of the RCA and SVG was closed with a Xenomedica patch (Baxter Healthcare, Chicago, IL). Pathohistologic examination revealed mucoid degeneration in the media of the aneurysmal wall (Fig 2). The multi-detector computed tomographic scan after the operation showed the radial artery graft was patent and the RCA had a good run off.
Ann Thorac Surg 2009;87:933– 4
Fig 2. Microscopical observation of the media of the right coronary artery. Eosin-stained areas are mucoid degeneration. (Hematoxylin and eosin stain, original magnification ⫻100.)
Comment
FEATURE ARTICLES
The incidence of coronary artery aneurysm is reported to be 4.9% based on the Coronary Artery Surgery Study (CASS) registry [1]. Atherosclerosis is the most common cause of coronary artery aneurysm [2]. It can occur secondary to inflammatory or infectious disease (ie, Kawasaki disease, Takayasu disease, systemic lupus erythematosus, or polyarteritis nodosa, endocarditis, and syphilis), connective tissue disease (ie, Marfan’s syndrome, Ehlers-Danlos syndrome), metastatic tumors, or blunt trauma [3]. In this case, the patient did not suffer from systemic connective tissue disease. Histologically
mucoid degeneration of the RCA and SVG is believed to be a cause of aneurysmal dilatation. Bacterial endocarditis develops in patients with coronary artery fistula [4]. Bacterial endocarditis was believed to be a complication of coronary artery fistula in this patient. The combination of coronary artery aneurysm with arteriovenous fistula is rarer (0.2%) [5]. The case of coronary artery aneurysm of the RCA with arteriovenous fistula resulting from high blood flow from a highpressure chamber to a low-pressure chamber was previously reported [6]. Residual or recurrent fistulas were found in 4 of 41 patients (9.8%) who underwent coronary artery fistula operations. The incidence of recurrence was higher in the external plication or division of fistula group compared with the intracardiac closure group [7]. In this case, mucoid degeneration of the aneurysmal wall, as well as high blood flow resulted in giant coronary artery aneurysm.
References
Fig 1. Multi-detector computed tomographic image shows aneurysmally dilated right coronary artery, fistula, and saphenous vein graft. (RCA ⫽ right coronary artery; SVG ⫽ saphenous vein graft.)
1. Swaye PS, Fisher LD, Litwin P, et al. Aneurysmal coronary artery disease. Circulation 1983;67:134 – 8. 2. Moriyama Y, Hisatomi K, Shimokawa S, et al. Coronary artery aneurysm repaired with saphenous vein patch plasty. Ann Thorac Surg 1998;65:561–2. 3. Aqel RA, Zoghbi GJ, Iskandrian A. Spontaneous coronary artery dissection, aneurysm, and pseudoaneurysms: a review. Echocardiography 2004;21:175– 82. 4. Sapin R, Frantz E, Jain A, et al. Coronary artery fistula: an abnormality affecting all age groups. Medicine 1990;69:101–13. 5. Hurst JW, Schlant RC, Rackley CE, et al. The heart: arteries and veins, 7th ed. New York, NY: McGraw-Hill Inc, 1990: 1233– 4. 6. Sexena P, Konstantinov IE, Burstow D, et al. Surgical repair of a large coronary artery aneurysm with arteriovenous fistula. J Thorac Cardiovasc Surg 2006;131:1167– 8. 7. Cheung DL, Au WK, Cheung HH, et al. Coronary artery fistulas: long-term results of surgical correction. Ann Thorac Surg 2001;71:190 –5.