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Ann Thorac Surg 2006;81:729-732
© 2006 The Society of Thoracic Surgeons

Case report

Off-Pump Surgery for Multiple Coronary Artery Fistulas With Aneurysm

Ju-Chi Liu, MD a , Paul Chan, MD, PhD a , Tsung-Hao Chang, MD b , Robert Fu-Chean Chen, MD b , *

a Division of Cardiovascular Medicine, Taipei Medical University—Wan Fang Medical Center, Taipei, Taiwan
b Division of Cardiothoracic Surgery, Taipei Medical University—Wan Fang Medical Center, Taipei, Taiwan

Accepted for publication December 20, 2004.

* Address correspondence to Dr Chen, Division of Cardiothoracic Surgery, Taipei Medical University—Wan Fang Medical Center; No. 111, Hsing-Lung Road, Section 3, Wen Shan District, Taipei City, Taiwan 116 (Email: cvchen{at}wanfang.gov.tw).


   Abstract
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 Abstract
Introduction
 Comment
 References
 
A 78-year-old woman presented with acute myocardial infarction, anterior wall, Killip III, with congestive heart failure. The finding of coronary angiographic examination was multiple congenital coronary artery fistulas with a huge aneurysm, with fistulas originating from both the right coronary artery and left anterior descending artery. The patient received surgery successfully without cardiopulmonary bypass. The finding of the pathologic examination revealed hyaline change in the aneurysmal vessel wall. In a two-year follow-up, the patient was found to be asymptomatic clinically with improved left ventricular function.


   Introduction
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 Abstract
 Introduction
Comment
 References
 
Congenital coronary artery fistulas with aneurysm formation are rare, particularly with acute myocardial infarction as clinical presentation [1]. The indications for surgical intervention have not been well-defined [2]. We report a good long-term outcome for a patient with acute myocardial infarction caused by multiple congenital coronary artery fistulas with an aneurysm, which was treated successfully by surgery without cardiopulmonary bypass support.

The 78-year-old woman complained of chest tightness suddenly one day before admission. Besides her persisting chest tightness, she also had nausea, cold sweating and dyspnea. Thus, she presented at our emergency room more than 12 h after the onset of symptoms occurred. She denied any history of major systemic disorders, such as hypertension, diabetes mellitus or hyperlipidemia.

In the physical examination, her blood pressure was 179/102 mm Hg and her heart rate 107 beats per minute. Coarse crackles were noted on auscultation of bilateral lung fields. Initial resting electrocardiogram (ECG) showed Q wave with ST elevation in V1-3 and reciprocal ST segment depression in leads II, III, aVF, and V4~6. The chest X-ray film revealed cardiomegaly, acute pulmonary edema, and left-sided pleural effusion. Her diagnosis was acute myocardial infarction, anteroseptal wall, Killip III with acute congestive heart failure. She received intravenous nitroglycerin, heparin, diuretics, oral aspirin, and angiotensin converting enzyme inhibitor. The peak serum creatinine kinase was 414 units/L and its MB form 102 IU/L. The echocardiographic finding showed that she had severe anteroseptal wall hypokinesis, left ventricular dysfunction (ejection fraction, 45%), moderate amount of pericardial effusion, and mild mitral regurgitation. The result of the thallium-201 myocardial perfusion study demonstrated a moderate fixed defect in the anteroapical wall and a mild fixed defect in the apex.

As indicated in Fig. 1, the coronary angiographic finding showed normal coronary blood flows without significant stenosis or thrombosis in the left anterior descending artery (LAD). However, a huge spherical aneurysm about 3 cm in diameter (Fig. 1) was found to have connected with multiple coronary fistulas originating from both the right coronary artery (RCA) and LAD.


Figure 1
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  		Fig 1. The pictures of coronary angiography. (A) A coronary aneurysm connected with multiple fistulas from LCA. (B) Coronary fistulas originating from RCA and draining blood flow into the aneurysm, follow-up after two years. (C) Indicated LCA. (D) Indicated RCA.

 
She received surgical intervention with the usual median sternotomical approach under general anesthesia. A Swan-Ganz catheter was inserted for hemodynamic monitoring. Cardiopulmonary bypass machine was standby in the operating room. Patient had autotransfusion with a cell saver but did not receive anticoagulant. We found a spherical coronary artery aneurysm (3 cm in diameter), which was located near right ventricle outflow tract between RCA and LAD. We also saw multiple coronary fistulas from RCA and LAD, which were draining into the aneurysm with several tortuous feeding vessels of about 3 mm in diameter. We tied those feeding vessels individually, then opened and partially resected the aneurysm for pathological examination. We checked and identified every entry point in the inner surface of the aneurysm (the receiving chamber). Having carefully traced the routes of these fistulas from all drainage sites in the aneurysm, we made sure that all fistulas were tied well before our resecting the aneurysm. The entire procedure was performed without the use of cardiopulmonary bypass support (off-pump). She was medicated with esmolol, a short-acting beta-blocker, for reduction of heart rate and myocardial contractility during the procedure. Her ECG tracing did not show any ST-T segment change or cardiac arrhythmia. She did not receive any blood transfusion or inotropic agents during the operation.

The finding of the pathological specimen revealed that the aneurysmal vessel wall had hyaline change and focal fibrin deposition (Fig 2). The patient recovered well and was discharged to clinic follow-up. In a two-year follow-up, this patient was clinically free of symptom with improved left ventricular function with ejection fraction up to 61% (per echocardiography) and decreased extent of myocardial ischemia (per Thallium-201 myocardial perfusion scans).


Figure 2
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  		Fig 2. The finding of the pathological specimen (magnification, x400). Hyaline change with focal fibrin deposition in the media of the vessel wall.

 

   Comment
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 Abstract
 Introduction
 Comment
References
 
After our literature review, we found that this case appears to be very rare: presentation of acute myocardial infarction due to multiple congenital coronary artery fistulas (origin, RCA and LCA) with an aneurysm. The patient recovered well after off-pump coronary artery surgery.

Coronary artery fistula is a rare congenital anomaly with a prevalence of 0.1% to 0.3% in adult population undergoing coronary artery angiography [3]. The majority of the coronary fistula is single [3]. Some clinicians reported that fistulas originate primarily from the left coronary arteries (75%–85%) [1]. Multiple fistulas originating from both RCA and LCA are rare [3]. Only three cases of fistulas with aneurysm formation were identified in one study of 4,300 adults undergoing coronary angiography [4].

The majority of the coronary artery fistulas are congenital, and they are not related to other cardiac anomalies [5]. Fistulas may develop during enlargement of the coronary capillary network between the 6th and the 8th weeks of embryogenesis. According to the model of Holman and Penniston, pathogenesis of aneurysm formation may be associated with hydraulic forces beyond a stenotic site, resulting in an injury to the vessel wall and leading vessel weakening and dilatating [6]. However, the actual pathophysiological mechanism in this case is still unclear.

Natural histories of patients with coronary artery fistulas are variable. Patients with isolated coronary artery fistula may not have any symptom throughout the life [3]. But some develop problems after a long asymptomatic period. The most frequent clinical manifestations were dyspnea or fatigue, which occurred in approximately 25% of patients. Congestive heart failure or angina each occurred in approximately 20%. Myocardial infarction was very rare; the reported incidence was about 4% [5]. Increased turbulence in the fistulas may damage endothelium and cause myocardial ischemia. Thromboembolism may be related myocardial infarction.

There is general agreement that surgical intervention is necessary for the symptomatic coronary artery fistulas. However, the indications for surgery in asymptomatic patients are less well-defined. Some surgeons recommend that nearly all patients be treated surgically as soon as the diagnosis was made because the incidence of symptoms seems to increase with age and complications eventually develop [5]. In contrast, some series showed the natural courses of coronary artery fistulas are benign in those who were discovered incidentally at angiography [4, 5]. Therefore, they do not appear to need surgical treatment immediately. It seems reasonable to recommend careful follow up in asymptomatic and uncomplicated patients.

Conventional coronary arterial surgery under cardiopulmonary bypass is still a high-risk procedure that may result in numerous complications, including intraoperative ischemia, impaired hemostatsis, mechanical trauma to blood cells, and activation of complement [7]. Off-pump procedures may avoid the complications of cardiopulmonary bypass, save operative time, reduce cardiac invasions, and keep better myocardial function [8]. In our case, we chose the off-pump approach to avoid further myocardial injury. At the two-year follow-up, our patient's myocardial ischemia had decreased (per Thallium 201 scan) and her left ventricular function had improved (per echocardiography). We think that increased postoperative coronary blood flow, avoidance of intraoperative myocardial injury with the off-pump procedure, and beneficial effects from the angiotensin converting enzyme inhibitor on myocardial remodeling would have contributed to the improvement of our patient's myocardial performance.

In summary, we presented a patient who had multiple coronary fistulas and an aneurysm complicated with acute myocardial infarction, was treated successfully by off-pump surgery. After two years, this patient was found doing well under regular medications.


   References
Top
 Abstract
 Introduction
 Comment
 References
 

  1. Gillebert C, Van Hoof R, van de Werf F, Piessens J, Geest HD. Coronary artery fistulas in adult population Eur Heart J 1986;7:437-443.[Abstract/Free Full Text]
  2. Lowe JE, Oldham HN, Sabiston DC. Surgical management of congenital coronary artery fistulas Ann Surg 1981;194:373-380.[Medline]
  3. Vavuranakis M, Bush CA, Boudoulas H. Coronary artery fistulas in adultsincidence, angiographic characteristics, natural history. Cathet Cardiovasc Diagn 1990;21:28-40.[Medline]
  4. Liu PY, Chan SH, Chao TH, et al. Adult coronary artery fistula—a retrospective analysis of 14 cases Acta Cardio Sin 2003;19:221-228.
  5. Sapin P, Frantz E, Jain A, Nichols TC, Dehmer GJ. Coronary artery fistulasan abnormality affecting all age groups. Medicine 1990;69:101-113.[Medline]
  6. Holman E, Peniston W. Hydrodynamic factors in the production of aneurysms Am J Surg 1995;90:200-209.
  7. Magovern JA, Mack MJ, Landreneau RJ, et al. The minimally invasive approach reduces the morbidity of coronary artery bypass Circulation 1996;94(Suppl I):I-51.
  8. Akins CW, Boucher CA, Pohost GM. Preservation of interventricular septal function in patients having coronary artery bypass grafts without cardiopulmonary bypass Am Heart J 1984;107:304-309.[Medline]



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