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Ann Thorac Surg 2001;71:190-195
© 2001 The Society of Thoracic Surgeons
a Division of Cardiothoracic Surgery, Department of Surgery, The University of Hong Kong, Hong Kong
Accepted for publication April 26, 2000.
Address reprint requests to Dr David L. C. Cheung, The University of Hong Kong, Grantham Hospital, 125 Wong Chuk Hang Rd, Aberdeen, Hong Kong
e-mail: dlcheung{at}netvigator.com
| Abstract |
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Methods. We studied 41 patients with coronary artery fistula operated in our unit in the past 30 years with restudies including coronary angiograms in those who agreed to the investigation.
Results. There was no operative mortality and operative morbidity was low. The mean duration of follow-up was 9.1 years and 96.9% of the patients were asymptomatic. Twenty-one patients had a coronary angiogram. The native coronary artery either remained dilated and tortuous, or more frequently had thromboses with a short proximal stump. (None of these patients had evidence of myocardial ischemia.) Four patients had demonstrable recurrence fistula but without hemodynamic disturbance.
Conclusions. We advocate operation for all patients with coronary artery fistulas and demonstrable shunting in view of minimal operative risks. Small asymptomatic fistulas without demonstrable shunting should be left alone. The relatively high incidence of residual or recurrent fistula makes long-term follow-up mandatory.
| Introduction |
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| Patients and methods |
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| Results |
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There was no operative or hospital mortality in this group of 41 patients. One patient was reexplored for persistent bleeding, which was located at the coronary arteriotomy site. Four patients developed postpericardiotomy syndrome requiring open pericardial drainage. Two patients had supraventricular tachycardia requiring medications for control. Two patients developed a right bundle branch block that reverted to normal on follow-up. Three patients had electrocardiographic evidence of ischemia with transient elevation of cardiac enzymes. No patient had frank evidence of infarction.
Thirty-nine patients were available for follow-up. One patient had emigrated 1 year ago and was well at the time. The mean duration of follow-up ranged from 5.08 months to 28.09 years. Thirteen patients were followed-up for more than 10 years. Thirty-three patients were asymptomatic (80.5%). One patient had persistent palpitation with 24-hour electrocardiogram showing frequent runs of ventricular ectopics similar to the preoperative electrocardiogram. One patient had mild exertional dypsnea and another patient who had a mitral valve replacement had mild congestive heart failure. A soft ejection systolic murmur was heard in 3 patients along the left sternal border. The electrocardiogram of the 3 patients with ischemic changes in the immediate postoperative period showed only nonspecific changes. Exercise stress test results were normal in 2 patients, but was positive in the third patient. The latter patient had the left circumflex artery ligated at its midpoint, which at the time of operation did not show any evidence of ischemia after temporary occlusion of the artery for 15 minutes. Exercise test was also performed in the 2 patients with concomitant valve replacement and target heart rate was reached in both with no ischemic changes.
Twenty-one patients (51.2%) agreed to a second cardiac catheterization. Nine patients had external plication/division and 12 had closure within the recipient cardiac chamber. The angiographic findings are as follows: proximal coronary artery, normal; distal coronary artery, normal (1 patient); proximal coronary artery, dilated; distal coronary artery, normal (10 patients); proximal coronary artery, dilated; distal coronary artery, threadlike/completely thrombosed (4 patients); proximal coronary artery, thrombosed stump; distal coronary artery, filled by collaterals (2 patients); and proximal coronary artery, dilated; distal coronary artery, recurrent/residual fistula (insignificant shunt) (4 patients). The dilated proximal coronary arteries either remained dilated, showing no signs for regression or were thrombosed leaving a short stump at its origin from the aorta. These findings were prevalent in all of those patients studied. The distal coronary arteries remained normal in size in 6 patients; but was threadlike or completely thrombosed in 4, the circulation being maintained by retrograde filling from collaterals of the opposite coronary system. Residual/recurrent fistulas were found in 4 patients (9.8%) and was equally divided between the two group. In 2 patients, the residual fistula was located at the same site as before but with no demonstrable shunting (Fig 1). One patient had a double fistula to start with, the dominant right coronaryright atrial fistula had been dealt with and the small left circumflexright ventricular fistula was left alone. This latter fistula was found to have increased in size over the years with a calculated shunt of 1.2:1 in the recent study. The right coronary system was blocked completely with only a short dilated proximal stump left (Fig 2). The fourth patient had a left circumflexright atrial fistula ligated. A small recurrent fistula was found on follow-up, which was fed by collaterals from the left anterior descending and the right coronary arteries (Fig 3). The residual shunts in these patients were small and insignificant and no patient had been reoperated to date.
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| Comment |
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Symptoms include dypsnea on exertion, easy fatigability, angina pectoris, and other vague nonlocalizing complaints. Sometimes patients presented with complications from congestive heart failure, myocardial infarction, rupture of dilated aneurysmal coronary arteries and hemopericardium, bacterial endarteritis, or peripheral embolization [1, 813]. It is important to note, however, that an estimated 40% to 55% of patients with coronary artery fistula are entirely asymptomatic [1, 2, 8, 10]. In our series, 13 patients (31.9%) were asymptomatic. There is now evidence that the proportion of symptomatic patients increases with age [4, 6, 8]. Liberthson and colleagues [14] reviewed 187 patients with coronary artery fistulas and showed that by using 20 years old as the dividing criteria, the proportion of symptomatic patients increased from 9% to 55%. The incidence of death before operation also increased from 1% in those less than 20 years of age to 14% in those more than 20 years of age. The operative results were also much better with a mortality of 1% and a complication rate of 7% in the young age group compared to the 7% and 23%, respectively, in the older age group. Our series concurred with this observation and 85% of the asymptomatic patients were younger than 20 years, whereas 77% of the symptomatic patients were more than 20 years old.
It is well accepted that all symptomatic patients should be treated with surgical ligation or closure, and the same applies to those with complication [1, 6, 8, 10, 15, 16]. For asymptomatic patients, most surgeons will operate on those with a significant shunt because of the large volume load it places on the recipients cardiac chambers [1, 10, 15, 16]. However, treatment for the asymptomatic fistulas without significant shunting still remains controversial.
All the major studies available in the literature were collective reviews and the majority of the investigators would opt for operation even in those with no symptoms [3, 4, 8, 10, 14, 15]. The arguments for operative treatment in this particular group of asymptomatic patients being that it is a safe procedure with a low mortality and morbidity [1, 4, 10, 15, 17]; that it is effective in immediately correcting the hemodynamic disturbance and thus relieving the extra volume load that the fistula places on the heart [18]; that it is reliable with a negligible recurrence rate [1, 10, 15, 19]; that it is safer to operate at an earlier age [1, 14]; and that by operating early, the chance of subsequent development of complications will be reduced or eliminated [8, 10, 14, 19].
Although the main consensus of opinions are in favor of operation once the condition is diagnosed, there are researchers who question the validity of this policy [20]. The main criticism lies in the fact that the natural history of this disease entity had not been well documented. Cases of spontaneous closure, although rare, had been reported [21]. Francis and colleagues [22] reported on a patient with coronary-to-pulmonary artery fistula with a 2:1 shunt followed for 13 years with no functional, hemodynamic, or angiographic changes. Scattered reports of similar nature were also available showing no progression of disease on long-term follow-up. We did, however, demonstrate in one of our patients that after closing the main fistula, a residual small fistula in the opposite coronary system grew and increased in size on follow-up. In addition, 2 of our patients, who were asymptomatic at the time of diagnosis, became symptomatic in 3 to 5 years duration, strongly suggesting that the pathology does progress with time. The hemodynamic effect of, and the anatomic changes after, operation were also not well shown. Follow-up data from the major review series were always incomplete [10, 15]. Very few investigators had reported on the incidence of recurrent or residual fistula after operation [10, 23]. Acceptance as satisfactory surgical results were usually based on questionnaires, telephone interviews, or clinical examinations alone, and very few patients received a full postoperative workup, including a second cardiac catheterization [10, 15]. Therefore, conclusions based on these data that operation is a reliable method is not quite valid. Among our 21 patients who agreed to a second cardiac catheterization, we discovered 4 with recurrent/residual fistulaa surprisingly high incidence of 19.0%. Although the number of recurrence was the same in the each, the incidence was higher in the external plication/division group (22.2%) as compared to the intracardiac closure group (16.6%). It is now our current policy to close the fistula through ligation within the recipient cardiac chamber. All these patients were asymptomatic and only 1 had a soft ejection systolic murmur along the sternal border that by itself would not have suggested the diagnosis. Our study also showed that the dilated proximal coronary arteries either remained permanently dilated, as already demonstrated by Jaffe [24] and Gasul [4] and their colleagues, or were thrombosed all the way to their aortic origins (the distal coronaries being filled by retrograde circulation). Neither of these entities are desirable, as this means that the complications that one tries to avoid with operation are still potentially possible: the former condition is associated with a risk of rupture and the latter, with a risk of embolization or ischemia. Indeed, Araya and associates [25] have reported on a patient with recurrent coronary aneurysm that ruptured into the mediastinum and who died 6 months after operation for a coronary artery fistula, suggesting that surgical ligation or intracardiac closure of the fistula alone does not prevent subsequent rupture. It had been our policy that if the proximal coronary artery involved with the fistula is very dilated and thin-walled, we would either do a reduction aneurysmectomy by plication using Teflon felt strips or reinforce the thinned out area with a Dacron patch sutured over it for support as an attempt to prevent the potential complication of subsequent rupture, and this was done in 5 of our 32 patients. As for ischemia after operation, temporary occlusion of the coronary artery at the site of intended ligated to check for myocardial ischemia does not appear to be fault proof. Evidence of ischemia after operation developed in 1 of our patients whose circumflex artery fistula was ligated externally, although the temporary occlusion test at operation was negative. Similar experiences had been reported by other workers and evidence of ischemia had been demonstrated to appear as late as 4 hours after ligation [26]. We are fortunate that even in those patients whose coronary artery had thrombosed after operation, none of them showed any evidence of myocardial ischemia either clinically or on electrocardiographic examination, including a stress exercise test. Increased collateral circulation from the opposing coronary system is probably responsible for upkeeping the blood supply and in 3 of our patients, retrograde filling of the distal coronary artery, whose proximal portion had thrombosed by collateral circulation from the opposite coronary system was clearly demonstrated. Whether adequate collaterals can develop in sufficient extent to support the myocardium in all patients is still unknown and theoretically ischemia can result from thrombosis of the involved coronary artery.
Symptoms, complications, and significant shunt still remain the main surgical indications for coronary artery fistula. All symptomatic patients and those asymptomatic ones with moderate to severe shunting could be operated on with minimal morbidity and good surgical results could be expected. In spite of the relatively high incidence of recurrent/residual fistulas, these are usually small and of no hemodynamic significance. However, we have reservations for operating on the asymptomatic patients with no or only mild shunting. The benefits of operation in this group of patients are uncertain and there is a potential, although small, risk involved. These patients, however, should be regularly followed up and operation offered if they become symptomatic or if an increase in the amount of shunting is demonstrated. More long-term studies on larger numbers of patients with coronary artery fistulas both with or without operation is required before this question can be fully answered.
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