COLLECTIVE REVIEW

Congenital Coronary ArteryCardiac Chamber Fistula Review of Operative Management Edward A. Rittenhouse, M.D., Donald B. Doty, M.D., and Johann L. Ehrenhaft, M.D. ABSTRACT Eight patients who had surgical correction of coronary arterycardiac chamber fistula at our center and 163 from a review of the literature are presented. The patients are usually asymptomatic, and the diagnosis is suspected by observing a continuous cardiac murmur. Electrocardiographic findings are nonspecific. Angina pectoris or electrocardiographic evidence of severe ischemia are surprisinglyuncommon since coronary artery steal syndrome is also rare. Cardiac catheterization with angiocardiography is required to establish the diagnosis and identify the involved coronary artery and the cardiac chamber into which the fistula terminates. Left-to-right shunt flow is usually low (average Qp/QS = 1.5). Indications for operation are not precise. If there should be a large shunt flow (2.0) and symptoms of heart failure are present, the decision to operate is clearly justified. This situation is unusual, and operation is nearly always performed in an asymptomatic patient in whom the fistula is closed to prevent future symptoms or complications. The operation chosen is generally interruption of the fistula by direct ligation. Sometimes cardiopulmonarybypass is required. The results are good, with low morbidity (3.6% myocardial infarction)and low mortality (2%) justifying the operation, to be carried out prophylactically even in asymptomatic patients.

A

fistulous communication between a coronary artery and cardiac chamber was first reported by Krause [62] in 1865. Abbott [l] described the condition more thoroughly in 1906, and Bjork and Crafoord [13] performed the first surgical correction in 1947. Since that time congenital coroFrom the Division of Thoracic and Cardiovascular Surgery, Department of Surgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa. Address reprint requests to Dr. Ehrenhaft, Division of Thoracic and Cardiovascular Surgery, Department of Surgery, University of Iowa Hospitals and Clinics, Iowa City, Iowa 52242.

468

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Coronary Artery -Cardiac Chamber Fistula

nary artery-cardiac chamber fistulas have been reported with increased frequency [21,22,25,33,39,43,44,46,54,56,58,61,64,68,77,78,83,84,93,94]. T h e advent of aortography and selective coronary angiography has more accurately defined the anatomy, thus allowing a precise diagnosis [9, 311. However, this congenital anomaly of the coronary circulation occurs infrequently, and although numerous cases have been reported in the literature, few centers have had an extensive experience with operative correction. T h e patients requiring operation for a coronary artery-cardiac chamber fistula at this institution were analyzed and a review of the literature undertaken to determine the important features of the disease and the current results of surgical management.

Clinical Material At this institution 8 patients have undergone closure of a fistula between the coronary arterial system and cardiac chamber or great vessels (Table 1 ) . T h e ages ranged from 3 to 32 years (average, 12). There were 7 female patients and 1 male. One patient complained of dyspnea on exertion and fatigue, but the remainder were asymptomatic. A continuous grade 2-4/6 murmur was heard in all patients. Electrocardiographic abnormalities were present in 5 patients and included ST-T alterations (2 patients), left ventricular hypertrophy (3 patients), right bundlebranch block ( 1 patient), and left atrial enlargement ( 1 patient). Chest roentgenograms showed increased pulmonary vasculature in all but 2 patients, and 4 individuals had evidence of cardiac enlargement. Cardiac catheterization was performed in 7 patients (Table 2). T h e diagnosis of coronary artery-cardiac chamber fistula was obtained by detection of an oxygen increase in the right side of the heart and angiographically by injection of contrast medium into the aortic root. Right ventricular pressure averaged 26/4 mm Hg, pulmonary artery pressure 23/10, and aortic pressure 94/61. Pulmonary flow averaged 6.8 liters per minute per square meter of body surface area (4.4 to 10.1) and systemic flow 5.1 liters (3.9 to 7.5). Thus, the pulmonary-to-systemic flow ratio (QJQ,)was 1.37. Pulmonary resistance averaged 1.4 mm Hg/Wmin/m2 (0.8 to 2.3) and systemic resistance 14.6 mm Hg/Wmin/mz (9.5 to 18.7). The pulmonary-to-systemic resistance ratio (RJR,) was 0.09. There were no complications as a result of the cardiac catheterization. T h e coronary artery fistula originated from the left coronary artery in 2 patients with termination in the right ventricle in 1 and in the pulmonary artery in the other. In 5 patients a right coronary artery fistula drained into the right ventricle, and in 1 additional patient a fistula from the right coronary artery emptied into the pulmonary artery. Closure of the fistula was accomplished by direct ligation at the site of termination in all patients except 1 who required cardiopulmonary bypass and suture from within the right ventricle. Postoperative complications occurred in 2 patients and consisted of atelectasis and transient myocardial ischemia. There were no deaths, and overall results were excellent. Two individuals were noted to have recurrent or residual fistulas postoperatively that were not considered large enough to require surgical closure.

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None

None

7. 13, F

8. 3. F LVH

LVH, RBBB, & LAE

RVE, pulmonary overcirculation RVE, pulmonary overcirculation Pulmonary overcirculation Pu!monary overcirculation Cardiomegaly, pulmonary overcirculation Normal RCA

RCA

Ligation

Ligation RV

RCA

RV

Ligation

RV

RCA

Ligation

No

Closure from RV

RV

LCA

RV

No

Ligation (invaginate aneurysm)

RV

RCA

No

No

Yes

No

No

Ligation

MPA

RCA

No

Ligation

MPA

Cardiopulmonary Operation Bypass

LCA

Fistula Origin Termination

Excellent (recurrent fistula 6 mo) Excellent

Excellent

Excellent

Excellent

Excellent (minor residual fistula) Excellent

Excellent

Result

LCA = left coronary artery; MPA = main pulmonary artery; RCA = right coronary artery; RV = right ventricle; LVH = left ventricular hypertrophy; RVE = right ventricular enlargement; RBBB = right bundle-branch block; LAE = left atrial enlargement.

Thrill, 3/6 murmur

Thrill, 4/6 murmur

Normal

4/6 murmur

None

6. 14, F

T-wave inversion

3/6 murmur

None

5. 7 , F

Normal

None

4. 10, F

3/6 murmur

None

3. 4, F

LVH

RV heave, systolic murmur

None

2. 32, M

Chest Roentgenogram

ST-T abnor- Cardiomegaly, malities pulmonary overcirculation Normal Normal

ECG

Thrill, 4/6 murmur

2/6 murmur

Dyspnea

Examination

~~

1. 16, F

Patient No., Age (yd, & Sex Symptoms

TABLE 1. CLINICAL DATA ON PATIENTS UNDERGOING OPERATION FOR CORONARY ARTERY -CARDIAC CHAMBER FISTULA

COLLECTIVE REVIEW:

Coronary Artery -Cardiac Chamber Fistula

Review of the Literature Although the pathological condition of congenital coronary artery -cardiac chamber fistula was brought to the attention of the medical profession by Krause [62] in 1865, the surgical treatment began only in 1947 when Bjork and Crafoord [ 131 reported the first successful ligation of a fistula between a branch of the left coronary artery and pulmonary artery. Davis and associates [24] reported closure of a communication between a coronary artery and right ventricle in 1953. Halpert [5 11 in 1930 reported the first description of a coronary artery-coronary sinus fistula diagnosed postmortem, but it was not until 1963 that Haller and Little [49] identified the condition by angiography and carried out surgical correction. Upshaw [loo] in 1961 analyzed 73 patients reported in the literature and found 23 who had undergone surgical correction, with only 1 death. In that same report the fistulas were classified according to site of termination, and it was noted that 89% entered the right side of the heart. Agusti and colleagues [4] in 1967 found only 6 patients with coronary artery-left atrium fistula and reported the first right coronary artery-left atrium fistula. McNamara and Gross [66] reviewed the literature through 1967 and found 97 patients known to have undergone surgical treatment. Fifty-nine percent of the fistulas originated from the right coronary artery, 32% from the left, 2% from both, and 7% from a single coronary artery. T h e fistula terminated in the right ventricle in 52%,right atrium in 24%, pulmonary artery in 14%,left atrium or pulmonary vein in 8%,and left ventricle in 2%. A total of 163 patients who underwent surgical treatment of a coronary artery-cardiac chamber fistula had been reported in detail in the English literature through 1974 [2-5, 7-9, 11-16, 19, 21, 22, 26-28, 31, 32, 34-44, 47, 54-59, 61, 63-74, 76-84, 86, 87, 89, 90, 94, 95, 98, 100, 1051. There were 62 (38%) male and 101 (62%) female patients. T h e average age at the time of operation was 18 years (2 months to 64 years). Most of the patients were in the first decade of life (Fig. 1). T h e majority of patients were asymptomatic. Of the 67 (41%) who had symptoms, 31 (19%) described dyspnea at rest or on exertion, 17 (10%) had fatigue, 14 (9%)had symptoms of congestive heart failure, 16 (10%)had angina or chest pain, 4 had peripheral edema, 4 had palpitations, 4 subacute bacterial endocarditis, 1 hemoptysis, 2 pneumonia, and 2 persistent fever. T h e condition was usually first suspected by the discovery of a cardiac murmur. Eighty-eight (54%) were reported to have electrocardiographic abnormalities, but only 5 (3%)had evidence of a myocardial infarction preoperatively. Eighty-two patients (50%)had evidence of an enlarged heart or increased pulmonary vascular markings on chest roentgenogram. Cardiac catheterization was undertaken in most patients, but detailed results were given for only 42. T h e QJQ averaged 1.53 (1.0 to 2.8). There was no significant difference in magnitude of shunt flow in the group with symptoms (1.7 & 0.15) and the group without (1.5 ? 0.08). T h e diagnosis was usually established by angiography.

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Average

Patient No.

3616 2410 2 716 2212 2818 2515 2014 2614 23/10

36/13 2 119 24/12 2 118 24/14 2319 1515

Pulmonary Right Artery Ventricular Pressure Pressure (mm Hg) (mm Hg)

94/58 100170 95/63 103167 100165 70149 95/60 9416 1

Aortic Pressure (mm Hg)

3.9 7.45 5.3 4.5 4.4 5.1

4.35 7.45 9.4 10.1 4.4 6.8

5.05

Systemic Flow (Umin/mz)

5.05

...

Pulmonary Flow (Umin/m*)

1%

1.37

1.12 1.o 1.79 2.2 1.o

1.o

QP

1.8 0.81 1.27 1.48 2.3 1.4

0.89

Pulmonary Resistance (mm Hg/ Umin/mz)

0.09

0.096 0.085 0.088 0.1 1 0.12 18.7 9.45 14.3 13.2 18.1 14.6

0.065

RpIR,

13.7

...

Systemic Resistance (mm Hg/ Umin/m2)

TABLE 2. CARDIAC CATHETERIZATION DATA O N PATIENTS UNDERGOING CLOSURE OF CORONARY ARTERY -CARDIAC CHAMBER FISTULA

COLLECTIVE REVIEW:

Coronary Artery -Cardiac Chamber Fistula

FIG. I . Age distribution of 163 pa.tients who underwent surgical closure of a congenital coronary artery -cardiac chamber fistula.

Age (yrs.1

The fistula originated from the right coronary artery in 89 (41%),the left coronary artery in 66 (55%),and both coronary arteries in 8 (4%).It terminated in the superior vena cava in 4 patients (2%),coronary sinus in 13 (8%),right atrium in 36 (22%),right ventricle in 7 1 (44%), pulmonary artery in 27 (17%),pulmonary vein in 1 (l%),left atrium in 7 (4%),and left ventricle in 4 (2%)(Fig. 2). Repair was performed by direct ligation in 124 patients (76%),cardiopulmonary bypass in 37 (23%),and hypothermia in 2. Marked postoperative morbidity was recorded in 24 patients (15%)(Table 3). The most frequent complications were myocardial infarction, which occurred in 6 (3.6%),and myocardial ischemia in an additional 4 patients (2.5%).Serious arrhythmias developed postoperatively in 4 individuals (2.4%)and postpericardiotomy syndrome developed in 3 (2%).Two patients had congestive heart failure and another sustained a cardiac arrest. Retrosternal abscess, paralyzed diaphragm, transient coma, and mitral regurgitation were also noted. The overall early mortality was 2%,and these 3 patients died from cardiac tamponade, mitral FIG. 2. Origin and termination of congenital coronary artery -cardiac chamberfistula in 163 patients who underwent surgical correction. (LCA = left coronary artery; RCA = right coronary artery; SVC = superior vena caua; CS = coronary sinus; RA = right atrium; RV = right uentricle; PA = pulmonary artery; PV = pulmonary vein; LA = left atrium; LV = left ventricle.)

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RITTENHOUSE, DOTY, AND EHRENHAFT TABLE 3. MORBIDITY IN 163 PATIENTS FOLLOWING SURGICAL CLOSURE OF CONGENITAL CORONARY ARTERY -CARDIAC CHAMBER FISTULA

Corn plication Myocardial infarction Myocardial ischemia Postpericardiotorny syndrome Arrhythmias Bigeminy Atrial tachycardia Atrial fibrillation Congestive heart failure Cardiac arrest Retroster nal abscess Paralyzed diaphragm Transient coma Mitral regurgitation Total

No. of Patients & Percent 6 (3.6) 4 (2.5) 3 (1.8)

2 (1.2) 1 1 2 1 1 1 1 1

(0.6) (0.6) (1.2) (0.6) (0.6) (0.6) (0.6) (0.6)

24 (15)

regurgitation, and congestive heart failure with arrhythmia, respectively. Three additional patients died late postoperatively from sepsis, myocardial infarction, and cardiac arrhythmia, respectively. FISTULAS TERMINATING IN T H E SUPERIOR VENA CAVA, CORONARY SINUS, OR RIGHT ATRIUM

There were 53 patients whose fistula terminated in the superior vena cava [28,41,76l,coronarysinus[2,11,12,21,31,49,58,61,68,76,77,83,84,87,941, or right atrium [7, 14-16,28,34,35,42,54-56,59,61,78,80,82,83,86,90,94, 981. Of the 4 terminating in the superior vena cava, 3 were from the left coronary artery and 1 from the right coronary artery. Seven of the 13 coronary sinus fistulas originated from the left coronary artery and 6 from the right coronary artery. Of the 36 right atrium fistulas, 10 were from the left coronary artery and 26 from the right coronary artery (see Fig. 2). The average age in this group was 22 years (9 months to 64 years); 16 patients were male and 37 were female. Symptoms of congestive heart failure were present in 26 patients. Twenty-five had cardiac enlargement and increased pulmonary vasculature on chest roentgenogram. In 13 patients whose catheterization data were given in detail, the average QJQS was 1.6 (1.0 to 2.8). Surgical closure was accomplished by direct ligation (4 1 patients), cardiopulmonary bypass ( 1 1 patients), or hypothermia ( 1 patient). Postoperative complications consisted of postpericardiotomy syndrome (2 patients), myocardial infarction (2 patients), and serious arrhythmia (4 patients). Four individuals in this group died; there were 3 early deaths - from cardiac tamponade, arrhythmia with congestive heart failure, and mitral regurgitation - and 1 late death from myocardial infarction.

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FISTULAS TERMINATING IN THE RIGHT VENTRICLE

The coronary artery fistula terminated in the right ventricle in 71 patients [3,

7, 9, 19,22, 27, 28, 31, 37, 40, 43, 47, 54, 58, 61, 65-68, 70, 71, 73, 74, 77-79, 8 1 - 84,86,87,89,94,95,98,105].The site of origin was the left coronary artery in 27, right coronary artery in 41, and both coronary arteries in 3 (see Fig. 2). There were 29 male and 42 female patients with an average age of 13 years (2 months to 56 years). Symptoms included dyspnea (16 patients), chest pain (4 patients), subacute bacterial endocarditis (2 patients), and pneumonia ( 1 patient). The remainder were asymptomatic. The left-to-right shunt was manifested by increased pulmonary vascular markings o r cardiac enlargement on chest roentgenogram in 44 patients. The electrocardiogram showed evidence of ventricular hypertrophy in 40 patients. The QdQ was recorded in 17 patients and averaged 1.6 (1.1 to 2.8). Surgical treatment consisted of direct ligation in 45, closure with cardiopulmonary bypass in 22, tangential arteriorrhaphy in 3, and endoaneurysmorrhaphy under hypothermia in 1 . Complications in this group included myocardial ischemia or infarction (6 patients), retrosternal abscess ( 1 patient), coma ( 1 patient), cardiac arrest (1 patient), and paralyzed diaphragm (1 patient). There were no deaths in this group. FISTULAS TERMINATING IN THE PULMONARY ARTERY

Twenty-seven patients had fistulas terminating in the pulmonary artery [5,8, 28,3 1,36,44,58,63,66,68,70,74,78,82,83,86]. Fourteen fistulas were from the left coronary artery, 8 from the right, and 5 from both (see Fig. 2). There were 8 male and 19 female patients with an average age of 25 years (5 to 58). An increase in heart size or prominent pulmonary vascular markings were noted on chest roentgenogram in at least 8 of the patients. Electrocardiographic abnormalities consisted of left ventricular hypertrophy (8 patients), left axis deviation (2 patients), right bundle-branch block (1 patient), left bundle-branch block (1 patient), myocardial infarction or ischemia (2 patients), and right ventricular hypertrophy (1 patient). In those patients reported in detail the QdQsaveraged 1.3 ( 1 . 1 to 1.5). The surgical approach consisted of direct ligation in 23 patients and suture closure using cardiopulmonary bypass in 4 patients. There were no immediate postoperative complications, but 1 patient died at six weeks from sepsis. FISTULAS TERMINATING IN THE L E R ATRIUM OR PULMONARY VEIN

Of the 7 patients with a coronary artery fistula entering the left atrium, 4 fistulas originated from the left coronary artery and 3 from the right [4,38,69,78, 82,831. One patient had a right coronary artery-pulmonary vein fistula [26]. The average age in this group was 25 years (3 to 64). Six patients were male and 2 were female. Five of the patients had symptoms of congestive heart failure and 4 had cardiac enlargement on chest roentgenogram. The electrocardiogram showed left ventricular hypertrophy (2 patients), old myocardial infarction (2 patients), and atrial fibrillation ( 1 patient). Surgical closure of the fistula was by direct ligation in all patients in this group. Complications consisted of myocardial infarc-

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RITTENHOUSE, DOTY, AND EHRENHAFT tion (1 patient) and congestive heart failure (1 patient). One individual had mitral regurgitation and cardiac arrhythmia that resulted in his death three years postoperatively. FISTULAS TERMINATING IN THE LEFT VENTRICLE

Only 4 patients are reported to have undergone closure of a coronary artery-left ventricle fistula [32,39,66]. T h e age range was4 to 15 years; 2 were male and 2 female. all 4 patients had evidence of left ventricular hypertrophy on electrocardiogram, and 2 had cardiac enlargement on chest roentgenogram. Closure was performed on cardiopulmonary bypass in 3 patients and by direct ligation in 1. T h e only postoperative complication in this group was postpericardiotomy syndrome in 1 patient, and there were no deaths.

Comment EMBRYOLOGY

Coronary artery-cardiac chamber fistulas result from an abnormality in embryonic development of the coronary circulation [2 1, 39, 40, 58, 661. In the primitive heart the coronary veins initially form as endothelial outgrowths that penetrate the myocardium to form trabecular spaces and later terminate on the epicardial surface in capillary networks. T h e coronary arteries originate as an endothelial growth in the base of the aorta and communicate with the capillary network on the surface of the heart. Normally the intramyocardial sinusoids become narrowed and persist only as thebesian vessels in the adult. If the intram yocardial trabecular sinusoids fail to be obliterated, a fistulous communication persists between the coronary arteries and a cardiac chamber [20,45]. Wearn and co-workers [ 1031 described three types of anatomical communication between the coronary arteries and cardiac chambers. In the arterioluminal type the coronary artery enters the cardiac chamber directly, whereas in the arteriosinusoidal type the communication is through the myocardial sinusoidal network; the arteriocapillary fistula drains into the capillaries and then through the thebesian system into the heart chamber. Aneurysmal dilatation of the fistulous tract, which occurs frequently, may result from gradual weakening of the vessel wall from increased blood flow [29]. Histologically the media of abnormal coronary vessels has been shown to be very irregular in thickness with few, fragmented elastic fibers. Sclerosis and lipid accumulations have been found in the subendothelial zones [27, 291. PATHOPHYSIOLOGY

There are several hemodynamic consequences and adjustments to a coronary artery -cardiac chamber fistula, characterized by the size of the communication and chamber of termination [26,27,40,56,58,77,85,96,1041. Fistulas terminating in the pulmonary artery overload the pulmonary circulation and left ventricle as a result of the left-to-right shunt. Those emptying into the right atrium or right ventricle cause a volume overload on the right ventricle as well. Flow in fistulas to the left ventricle occurs almost exclusively during diastole, and thus the left ventricular chamber is overloaded, similar to the situation in aortic valve regurgitation. 476

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Coronary Artery -Cardiac Chamber Fistula

Shunt flow is determined by resistance in the anomalous vessel and the pressure differential between coronary artery and cardiac chamber. Since the abnormal coronary artery is usually markedly dilated, the most important determinant of flow in the vessel is the size of the opening into the recipient chamber. An additional factor is the changing size of the orifice during the cardiac cycle in those fistulas terminating in a ventricular cavity. Systole may partially obliterate the fistula aperture. It has been suggested [40] that flow may be less in fistulas terminating in ventricular cavities than in those emptying into the atrium, since with the latter there is a larger pressure gradient throughout most of the cardiac cycle. However, in our review of patients coming to operative correction, the average left-to-right shunt in fistulas entering the right atrium and right ventricular cavity was essentially the same (average Qp/Qs= 1.6). Furthermore, the magnitude of the shunt did not correlate with the presence of symptoms in our study. Ogden [75] found that fistulas terminating in the pulmonary artery often have multiple ostia o r a single small opening with low shunt flow. However, in all cases reviewed, Qp/Qs in the pulmonary artery fistulas averaged 1.3, only slightly below the average of those terminating in the right atrium or right ventricle. Although the left-to-right shunt flow through a coronary artery-cardiac chamber fistula may be large, a marked increase in pulmonary vascular resistance with reversal of flow from right to left has not been reported. Pulmonary hypertension has been described but is quite unusual [ 15, 231. In the 7 patients in our series undergoing cardiac catheterization, the preoperative study indicated an average pulmonary artery pressure of 23/10 mm H g (15/5 to 36/13 mm Hg). During systole there may be a tendency for greater shunting of flow through the fistula and away from the myocardium in left than in right coronary artery fistulas. T h e flow pattern in the right coronary artery is somewhat different because the major distribution of the vessel supplies the right ventricle, which has a relatively low systolic pressure [40,581. Very few direct measurements of coronary artery fistula flow have been made, and little is known of the phasic flow and pressure relationships. Dedichen and associates [26] recorded 1,800 ml per minute flow through a right coronary artery-pulmonary vein fistula. T h e instantaneous flow tracing documented a rather even flow during most of the cardiac cycle but with a definite short systolic peak increase. Hudspeth and Linder [55] recorded a flow of 735 ml per minute through a right coronary artery-right atrium fistula and distal flow of 50 ml supplying the myocardium. Closure of the fistula resulted in effective myocardial flow increasing to 122 ml per minute. Their study was an excellent demonstration of the “coronary steal” phenomenon [261. COMPLICATIONS OF CORONARY ARTERY-CARDIAC CHAMBER FISTULAS

T h e clinical manifestations of ischemia or infarction as a result of the coronary steal phenomenon are unusual [40]. Ghosh and colleagues [43] noted only 10 patients with angina included in the literature to 1969 and described 1 additional case. Morgan and co-workers [68] reported angina in 1 child and were among the first to describe electrocardiographic evidence of infarction in children with coronary artery-cardiac chamber fistulas. Oldham and associates [78] found that VOL. 20, NO. 4, OCTOBER, 1975

477

RITTENHOUSE, DOTY, AND EHRENHAFT only 7% of 150 reported patients had chest pain interpreted as angina pectoris. Bishop and associates [ 121 in 1974 noted that 6 patients with myocardial infarction had previously been reported and described 1 additional case. In our review only 3% of the 163 patients who underwent surgical closure of the fistula had evidence of a myocardial infarction on their preoperative electrocardiogram. It has been postulated that the relatively low incidence of myocardial ischemia and infarction is probably due to adequate collateral circulation that developed over a long period. A large shunt flow through the fistula may eventually result in congestive heart failure. Daniel and associates [221 reviewed 150 patients and found 2 1 (14%) who had congestive heart failure [3, 5, 10, 14, 17, 1 8 , 2 3 , 3 0 , 4 9 , 5 3 , 6 0 , 7 1 ,1011. It was noted that SO% were over the age of 20 years. Other authors have also noted that congestive heart failure may develop during infancy [861. Furthermore, all patients who had congestive heart failure had fistulas to the right side of the heart, and in the 4 patients who became decompensated during infancy the fistula terminated in the right ventricle. In our review of the literature, congestive heart failure was specifically described in only 14 patients (9%).However, an additional 43 patients (26%) had fatigue, shortness of breath, or paroxysmal nocturnal dyspnea that could be interpreted as early cardiac decompensation. Subacute bacterial endocarditis has been reported in approximately 10% of patients with a coronary artery-cardiac chamber fistula [40, 66, 86, 87, 92, 97, 991, but only 4 of the 163 patients who underwent operation were definitely stated to have had endocarditis preoperatively. Taber and associates [98] noted that subacute bacterial endocarditis preceded operative closure in 4 of 46 reported patients. Frequently the development of endocarditis was the first clue to the existence of a congenital cardiac lesion. Rupture of the aneurysmal coronary artery prior to operation is quite rare [6, 48, 521 and was not encountered in any of the patients included in our review. Even after closure of the fistula by distal ligation or intracardiac repair, the risk of rupture postoperatively still exists [6]. More proximal ligation of the vessel may reduce the risk but has been abandoned because of the high incidence of myocardial ischemia and infarction [56]. Distal closure of the fistula also does not affect the marked aneurysmal dilatation of the sinus of Valsalva at the origin of the coronary artery. The ultimate fate of these remaining aneurysms is unknown. Either resection or invagination of the terminal aneurysm may be indicated if it can be performed without compromising myocardial perfusion [27, 371. One potential complication of coronary artery-cardiac chamber fistulas is the development of premature atherosclerosis. Shear-induced intimal damage resulting from high flow through the vessel may be a precursor to premature coronary obliteration. Jaffe and associates [56] have analyzed shear stress and Reynold’s number in patients with coronary artery fistulas and concluded that both were increased at the narrow communication between the coronary artery and cardiac chamber, Further observations over a long period will have to be carried out before definite conclusions can be reached. Occasionally the recipient chamber of a coronary artery fistula may greatly 478

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COLLECTIVE REVIEW:

Coronary Artery -Cardiac Chamber Fistula

enlarge, impinging on surrounding structures. Floyd and co-workers [38] have reported a patient who developed obstruction of the superior vena cava from a markedly enlarged left atrium caused by a coronary artery-left atrium shunt. CLINICAL MANIFESTATIONS

Many of the reports in the literature have indicated that the majority of patients were asymptomatic. Gasul and colleagues [40] noted that even in the presence of obvious cardiomegaly, most patients remained active and unlimited. Only 1 of our 8 patients had mild dyspnea; the others had normal exercise tolerance despite evidence of cardiac overload in all 7. Of the 163 previously reported patients undergoing surgical correction, 4 1% described symptoms including dyspnea on exertion, angina, paroxysmal nocturnal dyspnea, subacute bacterial endocarditis, congestive heart failure, fatigue, upper respiratory infection, hemoptysis, edema, palpitations, pneumonia, and chest pain. T h e diagnosis is usually suspected from detection of a continuous murmur in an unusual location for a patent ductus arteriosus. However, at times it may be difficult to differentiate coronary artery-cardiac chamber fistula from patent ductus arteriosus, ventricular septa1 defect with aortic valve incompetence, sinus of Valsalva fistula, or aortopulmonary window. Several authors have commented on the characteristics of the coronary artery fistula murmur [7,40,43, 721. T h e diastolic component of the murmur may be louder in fistulas draining to the right ventricle since most flow is believed to occur then [40]. In a fistula to the atrium a systolic accentuation is often noted, and those draining to the left ventricle have a murmur only in diastole. Differentiation from patent ductus arteriosus can often be made on the basis of the murmur characteristics as well as location [72]. Phonocardiography may be a useful means of documenting the type of murmur and area of maximum intensity. Neufeld and associates [72] stated that the site of maximum intensity may vary according to the chamber in which the fistula terminates. In fistulas draining to the right ventricle the murmur is heard best along the left sternal border at the fourth or fifth intercostal space. For right atrium fistulas the point of maximum intensity is the second intercostal space at the right sternal border. For left ventricle fistulas the murmur is heard loudest along the right fourth and fifth intercostal space. And for fistulas draining to the pulmonary artery or left atrium the second intercostal space along the right sternal border is the area of maximum intensity. The roentgenographic features of coronary artery-cardiac chamber fistula to the right side of the heart are, in general, characteristic of any left-to-right shunt. Those terminating in the left heart chambers may be associated with enlargement of the left side of the heart, and sometimes diminished pulmonary vascular markings may be noted [40,69]. In patients with a small shunt the chest roentgenogram may be entirely normal. Some patients may have a dilated ascending aorta [40, 721, and occasionally an unusual cardiac silhouette has been described. Coronary artery aneurysms protruding beyond the borders of the heart are responsible for this unusual configuration [14, 88, 1011. Colbeck and Shaw [181 had a patient with cardiac calcifications from coronary aneurysm and arteriovenous fistula. VOL. 20, NO. 4, OCTOBER, 1975

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RITTENHOUSE, DOTY, AND EHRENHAFT Although 54% of the patients included in this review had electrocardiographic changes, they were not specific or diagnostic. The electrocardiogram in many patients with this lesion is entirely normal [26, 1021. McNamara and Gross [661 noted that 61% of 130 patients reviewed had evidence of right or left ventricular overload. Sakakibara and associates [86] found a high incidence of atrial fibrillation in patients with a fistula draining into the right atrium. Electrocardiographic changes of myocardial infarction are unusual in these patients despite the potential for myocardial ischemia from a coronary steal. Only 5 of the 163 patients included in this review had evidence of a myocardial infarction on their preoperative electrocardiogram. Morgan and co-workers [68] had 2 patients who developed myocardial ischemia during exercise. CARDIAC CATHETERIZATION

Cardiac catheterization with angiography is undoubtedly the best method for arriving at a precise diagnosis and determining the degree of shunt flow and cardiac decompensation. Only 1 of our patients who underwent catheterization had a significantly elevated pulmonary artery pressure (see Table 2). Gasul and colleagues [401 also noted that most patients with coronary artery-cardiac chamber fistula have normal pulmonary artery pressure, although isolated cases of mild [40,95, 1021 or moderate elevation have been reported [ 1 5 , 2 3 , 7I]. In a review of several cases, Gasul and associates [40] noted pulmonary shunt flow varying from 22 to 189%of systemic flow. Our patients had Q d Q ratios ranging from 1 to 2.2. The QdQs in the patients reviewed from the literature averaged 1.53 ( 1 to 2.8). The magnitude of the shunt did not seem to correlate with the presence of symptoms in either our patients or those reported in the literature. Retrograde aortography at the aortic root or selective coronary angiography usually defines the origin and termination of the shunt. In addition, it provides a qualitative estimate of shunt flow. INDICATIONS FOR OPERATION

In those patients who develop congestive heart failure, angina, or recurrent subacute bacterial endocarditis, the need for surgical closure of the fistula is clear. The indication for operation in asymptomatic patients or those with small fistulas is less well defined. Several authors have suggested that all fistulas should be closed once they are diagnosed [2 1,28,38,40,43]. When the lesion is discovered in infancy, most believe the operation should be performed electively during childhood [28,40]. It has also been argued that many patients have lived a normal lifespan with their coronary fistula detected only at postmortem examination [271. Based on the latter finding, it has been recommended that some patients who are asymptomatic not be subjected to closure of the fistula. Jaffe and associates [561 described 6 patients followed an average of ten years with stable hemodynamics. In 1 the fistula eventually closed spontaneously. They concluded that there may be little anatomical or functional change in these patients with a small to moderate shunt followed over a long period. The risk of death or substantial morbidity following closure of a coronary artery-cardiac chamber fistula has been shown to be very low. It therefore seems reasonable to proceed with surgical treatment in patients without symptoms in 480

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order to prevent the potential complications as well as relieve the chronic circulatory overload. OPERATIVE T R E A T M E N T

T h e operative approach in most patients has been direct ligation [64]. In the literature reviewed, 124 patients (76%)had their fistula closed by this method. T h e remainder required cardiopulmonary bypass (37 patients) or hypothermia (2 patients). In many of the earlier reports the coronary fistula was ligated proximal to its entrance into the cardiac chamber [21; however, a high incidence of myocardial ischemia resulted [3, 4, 14, 541. More recently most authors have advised ligation at the point of entrance to the cardiac chamber, thus avoiding the hazard of myocardial ischemia [2 1, 271. A period of temporary occlusion with constant electrocardiographic monitoring would seem warranted to determine if myocardial ischemia had resulted. Horiuchi and co-workers [54] pointed out the potential danger of ligating a right coronary artery fistula proximal to the fistula orifice since the atrioventricular nodal artery may be injured during ligation of fistulas communicating to the right atrium or superior vena cava. Cooley, Hallman, and their associates 150,641 have recommended tangential arteriorrhaphy in those patients who have multiple communications between the coronary artery and cardiac chamber. With that technique several horizontal mattress sutures are passed beneath the coronary artery at the point of maximal thrill, which is obliterated when the sutures are tied. Some fistulas must be approached utilizing cardiopulmonary bypass [54,59, 66,78,97,98]. Liotta and associates [64] stated that cardiopulmonary bypass may be required for successful closure of fistulas to the posterior aspect of the right ventricular inflow tract, especially in children. T h e opening into the right ventricular chamber in those patients was frequently near the ventricular septum and tricuspid annulus. In some cases of atrial communication the site of entry may be so far posterior that it is inaccessible without the use of bypass [98]. McNamara and Gross [66] have advocated using extracorporeal circulation for lesions in a particularly difficult location, angiomatous fistulas with multiple communications (usually to the right ventricle), and fistulas with large aneurysms requiring aneurysmorrhaphy. RESULTS OF OPERATION

T h e overall results following surgical ligation of coronary artery-cardiac chamber fistula have been very good. The 3.6% incidence of postoperative myocardial infarction in the patients reviewed may be reduced even further by proper placement of sutures to close the fistula and avoid vessels supplying the myocardium. Those fistulas terminating in the right ventricle have a higher incidence of postoperative myocardial infarction because there is a greater risk of impairing myocardial blood flow during interruption. Residual or recurrent fistulas have been alluded to in the literature, but the true incidence is unknown. Furthermore, many of these fistulas may be hemodynamically insignificant and not require closure. An early mortality of 2% was not excessive and should improve with further experience. T h e reason 4 of the 6 deaths occurred in the group with fistulas to the VOL. 20, NO. 4, OCTOBER, 1975

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RITTENHOUSE, DOTY, AND EHRENHAFT right atrium is unknown; however, the 1 early death from cardiac tamponade could possibly have been prevented. T h e low operative mortality associated with closure of a coronary artery fistula seems tojustify operation, even in asymptomatic patients, to prevent the complications, which have been well described.

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