ABDOMINAL aortic aneurysms (AAA) are frequently associated with clinically

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1 Case Report Multiple Coronary Artery Aneurysms Combined with Abdominal Aortic Aneurysm Oktay PEKER, MD, Kanat ÖZISIK, MD, Fatih ISLAMOGLU, MD, Hakan POSACIOGLU, MD, and Metin DEMIRCAN, MD ˆ ˆ SUMMARY Coronary artery aneurysm (CAA) is defined as coronary dilatation which exceeds the diameter of a normal adjacent segment or the diameter of the patients largest coronary vessel by as much as 1.5 times. It is an uncommon pathology with a frequency of 1-4% in routine autopsies or coronary angiographies. Atherosclerosis plays an important role in the development of CAA, and it may be a predominant cause in the majority of patients. However, the timing of surgical intervention and the treatment options for CAA are still controversial. In this report, we present a patient who had multiple CAAs of all main coronary arteries and abdominal aortic aneurysm. Different treatment modalities and indications are also discussed. (Jpn Heart J 2001; 42: ) Key words: Coronary artery aneurysm, Abdominal aortic aneurysm, Treatment ABDOMINAL aortic aneurysms (AAA) are frequently associated with clinically significant coexisting coronary artery disease. Therefore, this clinical fact has led several authors to recommend cardiac evaluation and coronary revascularization when indicated before or during AAA repair. 1) But, combined aneurysmal involement of the coronary arteries and the abdominal aorta is a rarely encountered matter in Turkey. The major etiologic factor for coronary artery aneurysm (CAA) is usually atherosclerosis. 2) The other less frequent etiologic factors are congenital abnormalities, Kawasaki disease, trauma, Ehlers Danlos syndrome, Marfan syndrome, poliarteritis nodosa,takayasu disease and syphilis. Moreover, CAA formation can be seen after percutaneous transluminal coronary angioplasty and directional coronary atherectomy due to medial damage or subintimal damage respectively, resulting from either dissection or overexpansion, or both processes leading to weakness and subsequent dilatation. 3) This rare pathology has a significant importance. Because the presence of atherosclerotic CAA is not always considered to be an operative indication it is not familiar to most surgeons. 3) There is no concensus From Department of Cardiovascular Surgery, Sevgi Hospital, Ankara, Turkey. Address for correspondence: Fatih Islamoglu, MD, Ege Universitesi Tip Fakültesi, Kalp ve Damar Cerrahisi Anabilim Dali Izmir, Turkey. Received for publication August 10, Revised and accepted September 13,

2 136 PEKER, ET AL Jpn Heart J January 2001 regarding the optimal approach to the CAA. It may differ from one patient to another. We present a patient who had multiple CAAs in all main coronary arteries and AAA with beta thalasemia minor. CASE REPORT A 60 year-old hypertensive, obese Turkish male was referred for repair of AAA to our unit. An epigastric throbbing pain was the only presenting symptom. Physical examination of the abdomen revealed a pulsatile abdominal mass. Preoperative laboratory tests were normal except microcytic and hypochromic morphology of the red blood cells. Elevated HbA2 levels in hemoglobin study revealed the laboratory diagnosis of beta thalassemia minor in this patient. Hypercholesterolemia was also detected. Preoperative total cholesterol level was 372 mg / dl (HDL=37 mg / dl and LDL=217 mg / dl). The patient had not any previous episodes of Kawasaki disease or syphilis. Further diagnostic evaluations with computerized tomography (Figure 1) and digital subtraction angiography (Figure 2) demonstrated large abdominal aortic aneurysm, with a maximal cm in diameter, extending from below the renal arteries to iliac bifurcation. His Figure 1. Computerized tomographic (CT) appearance of the abdominal cavity demonstrating severe aneurysmal dilation of the abdominal aorta and common iliac arteries.

3 Vol 42 No 1 COMBINED CORONARY AND ABDOMINAL AORTIC ANEURYSMS 137 Figure 2. Digital subtraction angiographic appearance of fusiform aneurysmal dilation of the abdominal aorta and iliac arteries. electrocardiographic (ECG) and echocardiographic evaluations were normal. Our policy with patients of 60 years and above having AAA is to evaluate the coronary arteries by routine coronary angiograpy even though they may have a normal ECG and good ejection fraction and wall motions. Coronary angiography showed multiple coronary artery aneurysms involving all main coronary arteries without any significant stenosis (Figures 3 and 4). Therefore, we decided to perform a surgical intervention for only AAA. The exposure of the AAA was performed through a transperitoneal approach. After systemic heparinization, aortic cross-clamp was applied below the renal arteries. The abdominal aortic aneurysm was incised longitudinally, and thrombus and debris were removed. There was no evidence of dissection. A bifurcated vascular graft (18 9 cm Bard Albumin coated graft) was interposed in end to end fashion between the abdominal aorta and common iliac arteries. Histologic study of a specimen taken from the aneurysm wall revealed remarkable atherosclerotic change. The postoperative course was uneventful. The patient was discharged on the 6th postoperative day. Na-warfarin and acetylsalicylic acid were started to protect against potential complications of coronary aneurysm, such as

4 138 PEKER, ET AL Jpn Heart J January 2001 Figure 3. Coronary angiography demonstrating multiple atherosclerotic aneurysmal dilations of the left anterior descending coronary artery. Figure 4. Coronary angiographic appearance of multiple aneurysmal dilations of the right coronary artery.

5 Vol 42 No 1 COMBINED CORONARY AND ABDOMINAL AORTIC ANEURYSMS 139 thrombosis. DISCUSSION Abdominal aortic aneurysms are frequently associated with various forms of coronary artery disease. Aproximately 80% of patients with AAA will have angiographic evidence of atherosclerosis in at least one coronary vessel and nearly 20% percent of all AAA patients will have surgically treatable coronary artery disease. 1) This is the main reason for evaluation of coronary arteries directly with coronary angiography in our patients, especially in elderly ones. A coronary artery aneurysm is defined as a coronary artery dilatation that exceeds by 1.5 times the diameter of normal adjacent segments or the diameter of the patient's largest coronary vessel. 4) Coronary artery aneurysms were first recognized in postmortem studies. More recently they have been recognized in antemortem and angiographic studies and according to the literature, the incidence varies from 1.5% to 4.9% in the presence of coronary artery disease. 4,5) In spite of a close correlation between coronary artey disease and AAA, and a frequent incidence of CAA, there have only been a few reports of combined multiple aneurysmal involvement of all main coronary arteries and AAA. Davi and colleagues reported a patient admitted to hospital for chest pain who died suddenly after 24 hours due to ventricular fibrillation and cardiac arrest. Chest X ray, echocardiogram and computed tomography had shown a very large paracardiac mass. Upon autopsy, three large saccular aneurysms were found, one for each coronary artery and lumen completely filled with thrombi. 2) LaMendola and colleagues reported a case of multiple coronary artery aneurysms in a patient with an abdominal aortic aneurysm and bilateral popliteal artery aneurysms. 6) Fuyama and colleagues also reported a 2- month old boy with Kawasaki disease who developed the rare complication of abdominal aortic aneurysm. He was followed up for 7 years and calcification was noted 33 months after the onset of the disease but the aneurysm did not decrease in size. The authors indicated that computed tomography was the imaging method of choice for the evaluation of obstructive or calcific changes. 7) Our case was an obese, hypertensive patient with hypercholesterolemia. Although there was no significant stenosis of coronary arteries, we concluded that the main causative factor leading to coronary artery aneurysms in this patient was a hypercholesterolemia based atherosclerotic process. The presence of an atherosclerotic CAA does not always warrant sur-

6 140 PEKER, ET AL Jpn Heart J January 2001 gical intervention. 3,4) However, progressively enlarged CAAs in a relatively short follow-up period necessitates surgical intervention, since they may have potential hazardous complications, such as rupture or embolism. 3,8) Furthermore, there is no concensus regarding the optimal surgical approach. Four different types of surgical interventions are usually performed by surgeons. The first is aneurysm ligation and distal bypass: The most important complication of this technique is the remarkably high risk of inadvertent occlusion of important coronary branches which may lead to hemodynamic instability and deterioration. In addition, ligation of a coronary artery aneurysm with only mild or moderate stenosis constitutes a larger and more immediate demand on the flow reserve of the bypass conduit to the distal coronary artery, and unlike a vein graft, an arterial bypass graft may not be able to meet this immediate demand. This may lead to "hypoperfusion syndrome" and increased mortality. 3) The second is isolated coronary artery bypass grafting: This surgical intervention does not eliminate the potential complications of coronary artery aneurysm such as expansion, rupture, thrombosis and embolization. Also, "streak sign" or early graft occlusion can be seen due to a competitive flow pattern. The third is plication of the aneurysm: If the aneurysm wall is smooth, the procedure can be easly performed but it does not reduce the risk of later complications, such as CAA development. 9) The fourth and final is saphenous vein patch plasty repair of aneurysm: This alternative approach may be especially suitable for true sacciform type aneurysms. The advantages of this procedure are maintaining of antegrade flow and elimination of the need for any bypass grafting to the distal segment. 9) In the present case, we were not able to use different treatment modalities, which are explained above, because the aneurysm had a multiple and atherosclerotic nature causing no significant stenosis in coronary arteries. The optimal way to handle the aneurysm is still unknown, and the types of surgical interventions should be individualized depending on the patient's pathology. REFERENCES 1. King RC, Parrino PE, Hurst JL, Shockey KS, Tribble CG, Kron IL. Simultaneous coronary artery grafting and abdominal aneurysm repair decreases stay and costs. Ann Thorac Surg 1998; 66: Davi R, Marchese F, Borghesi MR, Romanelli R. Multiple coronary aneurysms of an atherosclerotic nature in a patient with an abdominal aortic aneurysm; presentation of an anatomo-clinical case. Pathologica 1997; 89:

7 Vol 42 No 1 COMBINED CORONARY AND ABDOMINAL AORTIC ANEURYSMS Dralle JG, Turner C, Hsu J, Replogle RL. Coronary artery aneurysms after angioplasty and atherectomy. Ann Thorac Surg 1995; 59: Robertson T, Fisher L. Prognostic significance of coronary artery aneurysm and ectasia in the Coronary Artery Surgery Study (CASS) registry. Prog Clin Biol Res 1987; 250: Barettella MB, Bott-Silverman C. Coronary artery aneurysms: an unusual case report and a review of the literature. Cathet Cardiovasc Diagn 1993; 29: LaMendola CL, Culliford AT, Harris LJ, Amendo MT. Multiple aneurysms of the coronary arteries in a patient with systemic aneurysmal disease. Ann Thorac Surg 1990; 49: Fuyama Y, Hamada R, Uehara R, et al. Long-term follow up of abdominal aortic aneurysm complicating Kawasaki disease: comparison of the effectiveness of different imaging methods. Acta Paediatr Jpn 1996; 38: Tunick PA, Slater J, Kronzon I, Glassman E. Discrete atherosclerotic coronary artery aneurysms: a study of 20 patients. J Am Coll Cardiol 1990; 15: Moriyama Y, Hisatomi K, Shimokawa S, Taira A, Arima S. Coronary artery aneurysm repaired with saphenous vein patch plasty. Ann Thorac Surg 1998; 65: