Download Core Curriculum Coronary Artery Fistulas

Catheterization and Cardiovascular Interventions 70:110–116 (2007)
Core Curriculum
Coronary Artery Fistulas: How to Manage Them
Larry A. Latson,* MD
Coronary artery fistulas are rare persisting vascular connections from a coronary artery
to a cardiac chamber or major central blood vessel. The true incidence is difficult to discern because at least 75% may be asymptomatic and clinically undetectable until an
echocardiogram or coronary arteriogram is performed. Small coronary artery fistulas
that are not clinically detectable are not clearly associated with significant long term
complications. Medium or large fistulas are associated with significant long term problems including angina, arrhythmias, myocardial infarction, endocarditis, and progressive
dilation. Treatment options include surgical and catheter approaches to significant fistulas. The long term outlook after fistula closure is not well defined and deserves further
study. ' 2007 Wiley-Liss, Inc.
Key words: coronary artery fistula; coronary anomaly; congenital heart defect; catheter
intervention
INTRODUCTION
Coronary artery fistulas are rare congenital malformations that can be defined as direct vascular connections
from a coronary artery to a cardiac chamber or major
central blood vessel without an intervening capillary
bed. The true incidence of coronary artery fistulas is
highly speculative since many may be small and never
detected, or only detected incidentally with imaging for
another indication. Coronary artery fistulas have been
seen in 0.3% of patients with congenital heart disease
[1], 0.06% of children undergoing echocardiography [2],
and 0.13–0.22% of adults undergoing coronary angiography [3–5]. At least 75% of coronary artery fistulas
found incidentally are small and clinically silent.
There is controversy about the best management of
coronary artery fistulas because both spontaneous regression and life threatening complications have been seen.
Data about long term risks of clinically silent fistulas
and the long term effects of interventions are particularly
lacking [6]. Improved methods of detection, treatment,
and follow-up have become available in the last 15
years, but there are no good large, complete, long-term
comparative studies of a significant number of patients
with coronary artery fistulas treated by specific strategies
or left untreated. Our aim in this article is to attempt to
review the limited available information along with our
own experiences and discuss our current management
strategies for patients with this rare malformation.
' 2007 Wiley-Liss, Inc.
ANATOMY AND PHYSIOLOGY OF
CORONARY FISTULAS
Coronary artery fistulas can originate from almost
anywhere in the coronary artery system and terminate in
any of the cardiac chambers, great veins, or pulmonary
arteries. If one looks at older (preechocardiographic)
series in which nearly all patients had large clinically
detectable fistulas, 60% originated from the right
coronary artery, and there were multiple fistulas in only
1% [7]. A more recent literature review that includes
more small fistulas tabulated that 55% arose from the
left coronary artery system and 8% had multiple origins
[8]. Small, asymptomatic fistulas arise much more commonly from the left coronary system (87%) [2]. In nearly
all series, over 90% of fistulas drain to the right side of
the circulation. Drainage to the left ventricle is least
common.
Cleveland Clinic, Center for Pediatric and Congenital Heart
Diseases, Cleveland, Ohio
*Correspondence to: Larry Latson, MD, Cleveland Clinic, Center for
Pediatric and congenital Heart Diseases, 9500 Euclid Ave., Cleveland,
OH 44195. E-mail: [email protected]
Received 12 January 2007; Revision accepted 17 January 2007
DOI 10.1002/ccd.21125
Published online 22 March 2007 in Wiley InterScience (www.interscience.
wiley.com).
Coronary Artery Fistulas
Coronary artery fistulas that drain to the right side of
the circulation create a left-to-right shunt of oxygenated
blood back to the pulmonary circulation. Those that
drain to the systemic veins or right atrium have a circulatory physiology similar to an atrial septal defect. Those
that drain to the pulmonary arteries are similar hemodynamically to a patent ductus arteriosus. A coronary fistula that drains to the left atrium results in no left to right
shunt, but does cause a volume load similar to mitral regurgitation. Similarly, a coronary artery fistula that
drains to the left ventricle produces hemodynamic
changes similar to aortic insufficiency. In addition to
shunts or volume loads on the heart, a coronary artery
fistula can result in the coronary artery \steal" phenomenon with coronary blood preferentially passing through
the fistula instead of into the more distal myocardial
capillaries. Symptoms of angina have been reported in
many patients with large fistulas, and may be aggravated
by distal coronary artery disease. Anginal symptoms
have disappeared with closure of the fistula in most
patients with no distal disease [9].
TYPICAL CLINICAL PRESENTATIONS AND
NATURAL HISTORY OF CORONARY FISTULAS
Patients with a coronary artery fistula can present in
several common ways. Patients with the largest fistulas
(relative to body size) may present with signs of congestive heart failure in infancy. Other neonates or infants
with a fistula may be diagnosed during evaluation for
separate congenital heart defects or because of a murmur. The typical murmur of a moderate or large coronary artery fistula is continuous with diastolic accentuation, but isolated systolic or diastolic murmurs are often
described. Pediatric patients with moderate or large fistulas are most often brought to medical attention because
of a murmur, but as many as 19% may have either a
complication of the fistula or symptoms possibly related
to the fistula [9]. Small, incidentally detected fistulas
were not associated with symptoms or complications
over a mean follow-up period of 9.3 years in the largest
study of this patient subgroup [2]. Although symptoms
and complications of medium or large sized coronary artery fistulas are not common in children, they become
increasingly apparent in patients diagnosed after 20 years
of age. In the literature review and analysis by Liberthson et al. in 1979, (which would likely include only
moderate or larger fistulas) 63% of patients over age
20 years had either symptoms or a complication of the
fistulas [9]. The most common symptoms and complications in adults include angina (especially if there is concomitant coronary artery disease), myocardial infarction,
heart failure, arrhythmias, and endocarditis. A very small
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Fig. 1. Volume rendered view of an ECG-gated cardiac CT scan
beautifully demonstrating a very large and tortuous right coronary artery (arrowheads) secondary to multiple fistulous connections to the coronary sinus. This fistula is classified as a
large fistula because the right coronary is more than three times
the expected diameter. Closure of this type of distal fistula is
associated with stasis and possibly late thrombosis in the very
dilated coronary artery. [Color figure can be viewed in the online
issue, which is available at www.interscience.wiley.com.]
number of large fistulas have been documented to
expand aneurysmally and even dissect or rupture [9–11].
FACTORS TO CONSIDER IN THE
MANAGEMENT DECISION
The best management of a patient with a coronary artery fistula would provide the greatest probability of
event-free survival over a lifetime. One must consider
what is known about the risks of nonintervention versus
the risks of any available intervention in the location and
timeframe in which a decision is made. For any given
patient there are many factors which may enter into the
decision. Some of the more important ones in our opinion include the size of the fistula, presence of symptoms
or complications likely related to the fistula, the age of
the patient, the relative size of the fistula, the anatomy of
the fistula, and whether the patient has other indications
to be undergoing an invasive procedure.
Defining the relative size of coronary artery fistulas
has not been systematically done, but it is clear that
many of the symptoms and complications of the fistulas
are greatly affected by size. Absolute size is a poor measure since a 3-mm fistula in a neonate may have far
more detrimental effect than a similar diameter fistula in
an adult. Left to right shunt ratios (QP/QS) are routinely
measured at catheterization, but are not large (over 2:1)
except in the most extreme cases. Aneurysmal dilation
of portions of large, thin walled fistulas is common, but
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Latson
Fig. 2. (A) Angiogram of a fistula arising from the very proximal
right coronary artery. The fistula has an aneurysmally dilated
mid portion and two exit sites (arrows) near the junction of the
superior vena cava and right atrium. To demonstrate this anatomy, the contrast injection was initiated in the fistula and
continued as the catheter was withdrawn to opacify the right
coronary artery. This is classified as a large fistula even though
the actual openings are relatively small (see text) because of the
aneurysmal dilation of the fistula itself in this 5-year-old. (B) An
Amplatzer vascular plug (large arrows) was placed proximally in
the fistula (at its origin from the right coronary artery) through a
delivery sheath advanced from the femoral vein over an arteriovenous guidewire rail. A second Amplatzer vascular plug (small
arrows) was placed in the largest exit in hopes of reducing the
chance of propagation of any thrombus that could form in
the aneurysm sack. A small amount of contrast is still seen
in the aneurysm from a previous angiogram.
the time-course of this dilation is not well understood.
Fistulas are often irregularly shaped and tortuous. The
smallest diameter of a fistula, frequently near the exit
point or at a sharp bend of a tortuous vessel, is often
considerably smaller than the largest portion. The excess
flow through a fistula tends to be associated with dilation
of the proximal portion of its associated coronary artery.
Very distal coronary artery fistulas cause (or are associated with) dilation, and often tortuosity, of the entire
length of the main coronary artery (Fig. 1). Those that
arise near the origin of the coronary will generally have
normal distal coronary artery anatomy (Fig. 2). In our
practice we subjectively consider that trivial or small fistulas are ones that result in little or no dilation of the
proximal coronary artery from which they arise, and are
themselves no larger at any point than twice the normal
expected proximate coronary artery diameter for the
patient (Fig. 3). These small fistulas are usually, but not
always, clinically silent and are generally detected incidentally. Fistulas that, at any point, are larger than two
times but less than three times the expected proximate
normal coronary artery diameter, or that are associated
with similar ranges of dilation of the proximal associated
coronary artery, are considered to be medium size fistulas. Fistulas with bigger dimensions are considered
large.
Symptoms of coronary artery fistulas tend to vary
with age and the size of the fistula. Heart failure related
to a large volume shunt is most likely to occur in neo-
nates or young infants with a large fistula. The neonatal
symptoms from a large left-to-right shunt are often controllable with typical medical management and an initial
trial of medications is indicated in very young patients.
Those who do not respond to medical management need
intervention in infancy. Intervention for asymptomatic
patients may best be delayed for observation unless the
fistula is large. Approximately half of symptomatic neonates are likely to become asymptomatic with the relative size of the fistula decreasing over time [12,13].
Spontaneous closure has been seen in some patients over
time, but these cases appear to be in patients with small
or medium size fistulas [8]. The most common symptoms in adults are angina, arrhythmias, or shortness of
breath. These symptoms are most likely to occur in adult
patients rather than children, but signs of coronary ischemia have been seen in some small children and should
be sought. Heart failure related to myocardial ischemia
is more likely to be seen in adults with concomitant coronary artery disease. Intervention is indicated in any
patient with likely myocardial ischemia.
Endocarditis is a risk for any type of arterio-venous
fistula and has been reported in 3% of patients (both
pediatric and adult) with a medium or large coronary artery fistula [9]. Endocarditis appears to be rare, but not
unheard of, before teenage years. The risk of endocarditis in patients with clinically unapparent fistulas detected
incidentally by echocardiography or angiography in
modern times appears to be much less. Intervention
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Coronary Artery Fistulas
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Fig. 3. (A) Left coronary arteriogram demonstrating a small,
tortuous fistula from the proximal LAD to the main pulmonary
artery. There are multiple openings into the MPA (arrowheads).
This is a small fistula that was associated with a murmur in a
17-year-old. If the fistula had not been fairly easily amenable to
catheter closure, the patient would have simply been followed
for the unlikely development of any later complications. (B) Multiple microcoils have been deployed to occlude the fistula. The
most proximal are immediately adjacent to the first fistulous
branch into the MPA and this branch was completely occluded
on a later aortogram.
should be offered to any patient with a coronary artery
fistula and a history of endocarditis or, at an appropriate
age, if the patient has a large or medium sized fistula
that is clinically detectable. Standard endocarditis prophylaxis precautions should probably be observed by
any patient with any type of coronary artery fistula. We
have not seen any reports of endocarditis in patients after
successful closure of coronary artery fistulas. We also
are unaware of any cases of endocarditis in patients with
a \silent," clinically undetectable, small coronary artery
fistula. Details about the clinical findings in patients with
endocarditis is lacking in most reports however. Therefore, it is not clear that an invasive procedure is warranted in this subgroup with the sole justification of
endocarditis prevention, especially if the anatomy is difficult for catheter closure. We do conservatively recommend routine endocarditis prophylaxis for the presence of any small fistulas, even if they are not clinically
apparent.
Age has a significant bearing on the decision to intervene in a particular patient at a particular time. There
are numerous reports of spontaneous regression or even
complete resolution of coronary artery fistulas in childhood. The size and developmental maturity of the patient
may have some effect on the ease and safety of either
surgical or catheter interventional procedures to close a
fistula. Symptoms and complications of coronary artery
fistulas are less common in children, but become more
significant in adults [3,9]. Elective intervention for medium or large asymptomatic fistulas in childhood
appears reasonable to prevent late complications. The
exact age in childhood to intervene may depend on some
of the other factors such as the anatomy of the fistula
and whether the fistula appears to be growing or shrinking over time relative to the child’s size. Small, asymptomatic fistulas that are clinically silent may best be
observed over time since the risks of complications or
symptoms appear to be low and spontaneous closure is a
possibility [14].
Approximately 10–30% of patients with a coronary
artery fistula also have another congenital cardiovascular
anomaly [3,8]. The most commonly seen defects include
variations of Tetralogy of Fallot, patent ductus arteriosus, and atrial septal defect. If a pediatric patient is
undergoing a surgical or catheter intervention for the
additional defect, elective closure of even a small coronary artery fistula at the same procedure is justified if the
fistula can be easily accessed. Similarly, intervention for
even a small fistula may be justified in an adult who is
undergoing a surgical or catheter procedure for an
acquired cardiovascular problem such as coronary artery
or valvular disease.
SURGICAL CLOSURE OF CORONARY
ARTERY FISTULAS
The first reported successful attempt at surgical closure of a coronary artery fistula was by Fell et al. in
1958 [15]. Coronary artery fistulas may be closed by
external placation on the beating heart or by intracardiac
closure using cardiopulmonary bypass with or without
cardiac arrest. In recent series, bypass was utilized in
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50% of cases [3,16]. Atriotomy or ventriculotomy may
be necessary to attempt to visualize the openings of the
fistula. Mortality related to surgical closure of isolated
coronary artery fistulas should be rare (<1%) [17]. There
have been occasional instances of periprocedural ST
changes, myocardial infarctions, arrhythmias, and
strokes. Recurrence or incomplete closure may occur in
10% [3,18]. Most recurrent or residual shunts have
been small and repeat surgical procedures have rarely
been reported [19]. Surgical approaches for coronary artery fistula closure can be readily utilized in patients of
all ages and sizes, and surgical closure is the preferred
approach in patients who are undergoing operative repair
of other cardiovascular problems [20]. The surgical
approach can also allow for reduction in the size of very
large aneurysmal dilations of either the fistula or the
proximal coronary artery [21].
CATHETER DEVICE CLOSURE OF CORONARY
ARTERY FISTULAS
Transcatheter occlusion of a coronary artery fistula
was first reported in 1983 [22]. Since then there have
been multiple reports of series including up to 33
patients [18,23–28]. Results have generally been comparable with reported surgical results with an expected
mortality rate of <1%. There have been rare instances of
periprocedural ST changes, myocardial infarctions, or
arrhythmias. Occluder device embolizations have
occurred in a number of reports, but the devices have
been removed successfully in the catheterization lab in
nearly all cases. Many different types of occluding devices have been utilized including detachable balloons,
Gianturco and other types of coils, Rashkind and
Amplatzer PDA and ASD devices, Grifka Bags, and
Amplatzer Muscular VSD Device, and Vascular Plugs.
Improved devices and delivery systems have made catheter closure applicable to over 90% of patients, including
infants and some neonates, in centers with extensive experience [29–31]. Catheter techniques may be difficult
or impossible in a small percentage of patients due to
extreme vessel tortuosity and inability to deliver a catheter far enough distally, presence of multiple drainage
sites, presence of normal coronary branches too close to
the drainage site to allow selective occlusion, or very
small size of the patient (neonate requiring treatment of
large fistula). Small residual or recurrent leaks have been
seen in 10% of patients treated by catheter techniques
[18,30]. Catheter interventions for residual small shunts
after either surgical or catheter interventions are readily
repeatable if necessary. Because of a much shorter recovery time and avoidance of a scar, transcatheter closure is considered the procedure of choice in applicable
patients.
The basic technique of transcatheter closure is to
advance a delivery catheter to the most distal portion of
the fistula (past any branches that may feed normal myocardium) and place the occluding device in that location.
In smaller fistulas, it may not be difficult to deliver small
devices like coils directly through a small arterial catheter (Fig. 3). Controlled release coils or delivery techniques may widen the spectrum of applicable patients
[32]. Small diameter coils can be delivered through 3F
microcatheters coaxially placed through a coronary
guide catheter [27]. In some large, high flow fistulas, we
have found that it may be advantageous to first pass a
small catheter and/or guidewire completely through the
fistula. The arterial guidewire can then be snared and
exteriorized on the venous side to provide a guidewire
rail. This arterio-venous guidewire rail can be used to
position even a large delivery system through the vein in
preference to the retrograde arterial approach. An
adequate size closure device such as an Amplatzer PDA
device or Amplatzer Vascular Plug can be precisely
deployed using this technique. If the fistula itself is large
and lengthy with its origin proximally from a main coronary artery, we generally will try to fill the fistula with
coils or also place a proximal occluding device. We feel
that this may reduce the risk of clot propagation into
normal structures or later dilation of the distally
occluded and possibly thin-walled fistula (Fig. 2). A covered stent may be a good device to close some coronary
artery fistulas [33]. The origin of the fistula from the normal coronary artery is excluded by placement of a covered coronary artery stent in the main coronary vessel.
This is an especially useful technique in adult patients
with coexisting coronary artery disease.
LONG TERM OUTCOMES AFTER TREATMENT
OF CORONARY ARTERY FISTULAS
There is limited longitudinal information about the
long term outlook for patients with coronary artery fistulas following surgical or transcatheter treatment. Many
papers indicate that most patients remain clinically
asymptomatic for a number of years after a closure procedure, but there are concerning findings from papers in
which follow-up has been more detailed. Small residual
leaks after either type of procedure appear to be present
in 10% of patients, but there is tremendous variability
between series. As expected higher rates are seen if follow-up evaluations have included detailed echocardiographic or angiographic study in all the patients [3].
Other residua and sequelae of the fistulas and closure
procedures include persistent dilation of the coronary artery, late stenosis in tortuous coronary arteries, arrhythmias, and late occlusion of dilated and tortuous coronary
arteries with or without myocardial infarction.
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Coronary Artery Fistulas
In one of the most rigorous follow-up studies to date,
Cheung et al. performed coronary arteriograms on 21 of
41 asymptomatic, mostly adult, patients after surgical
fistula closure and found 20 of the 21 studies to show a
persisting significant abnormality [3]. There was persisting proximal coronary dilation in 16/21. In six of these
16, the distal coronary artery was normal, but in 10, the
distal vessel was threadlike or completely occluded.
The remaining four patients had a small residual fistula
with the exit at the original or a previously unseen exit
location. McMahon et al. found persisting coronary artery dilation in all four of their pediatric patients 4 years
after catheter closure of large fistulas performed at 1.5–
3 years of age [26]. Hiraishi et al. found an absolute or
relative decrease in coronary artery size in three children after surgical fistula closure. IVUS and angiography however showed areas of stenosis and intimal
thickening in two of three who demonstrated an absolute reduction in coronary artery size. Inspite of normal
exercise ECGs, two out of their three patients had
abnormalities on thallium stress testing. At our institution, three of four patients who recently underwent follow-up evaluations 4–41 years after surgical fistula closure were found to have thrombosis of the entire parent
coronary artery. Thus, even though most patients are
asymptomatic after coronary artery fistula closure, they
should not be dismissed from follow-up. It may be prudent to follow them with appropriate tests to evaluate
myocardial perfusion. Long term anticoagulation with
at least an antiplatelet agent appears to be advisable and
we currently recommend warfarin for patients with
more than moderate persisting aneurysmal dilation of
the coronary arteries.
SUMMARY OF RECOMMENDATIONS
There appears to be good consensus that all symptomatic patients should undergo closure of medium or
large coronary artery fistulas. In our opinion, trivial to
small (not clinically discernable), asymptomatic fistulas
do not require a specific procedure for closure simply
because they are detected incidentally. However, closure
of even small fistulas may be recommended (1) if the
patient is considered to be at high risk for later endocarditis, or (2) if longitudinal follow-up is not feasible, or
(3) if the patient is undergoing an invasive procedure for
some other cardiac problem. The decision of whether,
and when, to submit a patient with a clinically detectable, but asymptomatic, moderate or large fistula can be
difficult. The risk of endocarditis in patients detected
clinically in earlier studies is not insignificant (3%). The
incidence of symptoms related to clinically apparent
fistulas increases significantly with advancing age
(especially after age 20). Surgical and catheter closure
115
procedures are relatively safe and catheter procedures
leave no residual scar and are well tolerated. In children,
we generally recommend elective closure of any coronary artery fistula which remains clinically apparent past
3–5 years of age, even if the patient is asymptomatic.
The risk of closure may be less, and the risk of late
giant aneurysm higher, in coronary artery fistulas that
arise near the coronary artery origin (Fig. 2). Fistulas
that arise proximally in a coronary artery lead to dilation
of only a short segment of the proximal true coronary
artery. These types of fistulas in our experience may be
the most likely to become grossly dilated and may be at
risk for rupture. We would close these types of fistulas at
any age.
We are most uncertain about medium or large fistulas
that arise very distally in the coronary system and are already associated with long segment coronary artery dilation at the time of detection. Longitudinal studies are
lacking, but it seems reasonable that the longer abnormally high flow persists in a coronary artery with a distal
fistula, the more likely for the coronary to become
increasingly dilated and tortuous. We generally recommend closing such fistulas early if discovered in children. Recent studies indicate that late stenosis due to
intimal hyperplasia may still leave these children at risk
for later myocardial ischemia, but we hope that relatively early intervention may lessen the secondary
changes. In older patients, if the proximal coronary has
become grossly enlarged, closure of distal fistulas has
been associated with a very high incidence of late thrombosis of the permanently dilated and tortuous proximal
coronary artery. If the older patient is asymptomatic, the
best course of action in this scenario is unclear to us.
Closure of the fistula results in relative stasis in the giant
coronary artery, and this is likely the etiology of late
thrombosis. These types of fistulas can be difficult to
close by catheter techniques. Since late thrombosis is
likely and may be no better than late development of the
coronary \steal" syndrome, nonintervention may have a
similar outlook to surgical closure, without the thoracotomy. It is not known whether innovative approaches
such as surgical reduction of the coronary artery would
be beneficial or detrimental. A conclusive trial of intervention versus novel therapy or expectant medical management for these patients with the worst coronary artery
fistulas will be nearly impossible because of the relative
rarity of this specific anatomy.
Coronary artery fistulas are rare but fascinating anomalies. The long term outlook of specific types of coronary
artery fistulas is sketchy at best. These fistulas can be
closed, but our understanding of the long term outcomes
of patients after fistula closure is woefully lacking. We
feel that this lack of information warrants following
patients with coronary artery fistulas, whether closed or
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Latson
not, indefinitely. Routine noninvasive, or even invasive,
studies of the anatomy and function of the coronary system in these patients should be the standard of care.
ACKNOWLEDGMENTS
We gratefully acknowledge the contribution of the
excellent cardiac CT image from Harold Litt, MD-PhD,
Assistant Professor of Radiology and Medicine Chief,
Cardiovascular Imaging Section, Department of Radiology, University of Pennsylvania Medical Center; and the
hard work and dedication of the physicians and staff of
the Center for Pediatric and Congenital Heart Diseases
at the Cleveland Clinic.
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Catheterization and Cardiovascular Interventions DOI 10.1002/ccd.
Published on behalf of The Society for Cardiovascular Angiography and Interventions (SCAI).