ISSN: 2233-601X (Print) ISSN: 2093-6516 (Online)

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Received: August 29, 2016, Revised: October 12, 2016, Accepted: October 17, 2016, Published online: June 5, 2017

Corresponding author: Hwan Wook Kim, Department of Thoracic and Cardiovascular Surgery, Seoul St. Mary’s Hospital, College of Medicine,

The Catholic University of Korea, 222 Banpo-daero, Seocho-gu, Seoul 06591, Korea

(Tel) 82-2-2258-2858 (Fax) 82-2-594-8644 (E-mail) kimhwanwook@gmail.com

© The Korean Society for Thoracic and Cardiovascular Surgery. 2017. All right reserved.

This is an open access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/

licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly

cited.

Surgical Management of a Coronary-Bronchial Artery Fistula Combined with Myocardial Ischemia Revealed

by 13

N-Ammonia Positron Emission Tomography

Hang Jun Choi, M.D., Hwan Wook Kim, M.D., Ph.D.,

Do Yeon Kim, M.D., Kuk Bin Choi, M.D., Keon Hyon Jo, M.D., Ph.D.

Department of Thoracic and Cardiovascular Surgery, Seoul St. Mary’s Hospital,

College of Medicine, The Catholic University of Korea

A 71-year-old male with known bronchiectasis and atrial fibrillation was admitted to Seoul St. Mary’s

Hospital with recurrent transient ischemic attack. Radiofrequency ablation was performed to resolve the pa-

tient’s atrial fibrillation, but failed. However, a fistula between the left circumflex artery and the bilateral

bronchial arteries was found on computed tomography. Fistula ligation and a left-side maze operation were

planned due to his recurrent symptom of dizziness, and these procedures were successfully performed. After

the operation, the fistula was completely divided and no recurrence of atrial fibrillation took place. A coro-

nary-bronchial artery fistula is a rare anomaly, and can be safely treated by surgical repair.

Key words: 1. Coronary artery disease

2. Fistula

3. Bronchial arteries

Case report

A 71-year-old male with known atrial fibrillation,

diagnosed 2 years previously, visited Seoul St. Mary’s

Hospital with symptoms of dysarthria and motor

weakness. The patient had a history of lobectomy for

treating uncontrolled bronchiectasis in his 30s. The

symptoms were eliminated soon after hospitalization

without any intervention, and the patient was dis-

charged with the diagnosis of a transient ischemic

attack. However, the symptom of dysarthria took

place again. Based on the possibility of thromboemb-

olism of the cerebral arteries, which can be caused

by atrial fibrillation, radiofrequency catheter ablation

was performed to treat his atrial fibrillation, but un-

fortunately his cardiac rhythm did not convert to si-

nus rhythm. However, in a multidetector computed

tomography (MDCT) scan that was taken to verify

whether coronary artery disease was present before

the ablation procedure, an abnormal communication

between the left circumflex artery (LCX) and the

bronchial artery was discovered incidentally (Fig.

1A). The coronary arteries themselves were revealed

to be intact. With the suspicion of a relationship be-

tween those symptoms and the coronary-bronchial

artery fistula, which could induce myocardial ische-

mia, a gated 13

N-ammonia positron emission tomog-

raphy (PET)–computed tomography scan with an ad-

enosine stress test was performed, and showed glob-

al left ventricular ischemia in the resting state.

Korean J Thorac Cardiovasc Surg 2017;50:220-223 □ CASE REPORT □

https://doi.org/10.5090/kjtcs.2017.50.3.220

Coronary-Bronchial Artery Fistula

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Fig. 1. Preoperative reconstructed image of chest computed tomography (A) and coronary angiography (B, C) show an abnormal commu-

nication from proximal LCX to bilateral bronchial arteries (arrow). LAD, left anterior descending artery; LCX, left circumflex artery, LM,

left main coronary artery.

Fig. 2. 13

N-ammonia positron emission tomography myocardial perfusion imaging demonstrating a global defect of the left ventricle (A, B)

and the decrease of CFR (C). HLA, horizontal long axis; VLA, vertical long axis; CFR, coronary flow reserve.

Cardiac contractility likewise did not sufficiently in-

crease after adenosine administration (Fig. 2A, B).

The flow rates at rest were 0.78 mL/g/min for the

left anterior descending artery (LAD), 0.78 mL/g/min

for the LCX branch, and 0.66 mL/g/min for the right

coronary artery (RCA). The flow rates in the ad-

enosine stress were 1.06 mL/g/min for the LAD, 0.98

mL/g/min for the LCX, and 0.84 mL/g/min for the

RCA. The coronary flow reserve was 1.37 mL/g/min,

1.24 mL/g/min, and 1.27 mL/g/min, respectively,

and the average flow reserve was 1.31 mL/g/min,

which indicated a generalized decrease of the coro-

nary flow reserve, suggesting the possibility of dif-

fuse microvascular disease (Fig. 2C). The flow re-

serve in the LCX territory was lower than that of the

other areas, but the decrease in flow was not espe-

Hang Jun Choi, et al

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Fig. 3. Postoperative coronary angiography shows the successful

occlusion of the coronary-bronchial artery fistula using surgical

clips (arrow). LCX, left circumflex artery.

cially significant. Transthoracic echocardiography re-

vealed only biatrial enlargement without a wall mo-

tion abnormality, and no coronary artery disease was

found on a coronary angiogram (CAG). A large fistula

from the LCX to both bronchial arteries was present

on the CAG (Fig. 1B, C), as previously shown on the

MDCT scan.

In order to resolve the patient’s symptoms, closure

of the coronary-bronchial artery fistula was scheduled.

Since pharmacologic and endovascular management

of the patient’s atrial fibrillation had failed before,

both the surgical ligation of the fistula and a maze

operation via median sternotomy were planned. After

median sternotomy followed by pericardiotomy, a

tortuous fistulous tract with a diameter of approx-

imately 1.5 mm was found on the roof of the left

atrium. After cardiopulmonary bypass with cardioplegia,

dissection and clipping of the fistulous tract were

performed. A left-side maze operation was also con-

ducted with a cryocatheter, and internal obliteration

of the left atrial appendage was performed with a

4-0 prolene suture. Electrocardiography after surgery

showed a regular sinus rhythm without recurrence of

atrial fibrillation, and a follow-up CAG revealed the

successful occlusion of the coronary-bronchial artery

fistula (Fig. 3). The patient was discharged with an

uneventful postoperative recovery, and no symptoms

occurred within the 10-month follow-up period.

Discussion

Coronary-bronchial artery fistula, an abnormal

communication between the coronary arteries and

the unilateral or bilateral bronchial arteries, is a rare

anomaly that is present in only 0.5% of patients who

undergo coronary angiography [1]. According to a re-

view article from the Netherlands, only 31 such fistu-

las were reported in the period from 2008 to 2013

[2]. Due to the advanced radiologic techniques that

are currently used, such as MDCT, diagnosing coro-

nary-bronchial artery fistula has become less compli-

cated [3,4]. However, the etiology of coronary-bron-

chial artery fistula is uncertain. Said et al. [2] sug-

gested the possibility that they involve the reopening

of preexisting, nonfunctional congenital communica-

tions between the bronchial arteries and the coro-

nary arteries. They proposed that 2 factors regulate

the reopening and growth of arterial communica-

tions: disequilibrium of the pressure gradient be-

tween the 2 arteries and obstruction of the coronary

arteries. Additionally, several case series have implied

the possibility of a relationship between coronary-

bronchial artery fistula and known bronchiectasis

[2,5]. Similar to the patients who have previously

been described, our patient also suffered from bron-

chiectasis. The progression of bronchiectasis leads to

hypertrophy of the bronchial arteries, and this

change in the bronchial arteries may influence the

presence of communication between the coronary ar-

teries and the bronchial arteries. Further studies are

needed to understand the etiology of coronary-bron-

chial artery fistula and its relationship with bron-

chiectasis.

The clinical presentation of patients with coro-

nary-bronchial artery fistula depends on the degree

of the left-to-right shunt and the concomitant disease

process in the patients. Said et al. [2] reported that

chest pain was the most frequent symptom (63%) in

their review, and Lee et al. [4] suggested an associa-

tion between the coronary steal phenomenon and

chest pain in coronary-bronchial artery fistula patients.

Hemoptysis (26%) and dyspnea (19%) are also fre-

quent, and otherwise asymptomatic disease occurred

in only 5 of 27 subjects (19%) [2]. The patient in

our report complained of recurrent dizziness and

syncope, but not of chest pain or hemoptysis. How-

ever, we cannot infer that the patient’s symptoms

Coronary-Bronchial Artery Fistula

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were due to the fistula, because they could also have

arisen from underlying atrial fibrillation.

Although coronary angiography has emerged as the

preferred diagnostic modality for coronary-bronchial

artery fistula, the invasiveness of this procedure is a

major obstacle. Noninvasive contrast-enhanced MDCT

is as useful as CAG for diagnosing coronary-bronchial

artery fistula and identifying the course of a fistulous

tract [4,6]. According to the reviews by Lee et al. [4]

and Said et al. [2], coronary-bronchial artery fistula

originated in the circumflex artery in 75% (6 of 8)

and 61% (19 of 27) of cases, respectively. Transtho-

racic or transesophageal echocardiography is also

helpful in detecting coexisting cardiac anomalies and

assessing the cardiac function of the patient. PET us-

ing 13

N-ammonia, which was performed in this case,

is also useful for the assessment of cardiac function.

Quantification of absolute coronary flow and meas-

urement of the coronary flow reserve by 13

N-ammo-

nia PET have the advantage of identifying the dis-

eased vessel, and these techniques can also be val-

uable in assessing coronary-bronchial artery fistula [7].

Since the appropriate treatment modality for coro-

nary-bronchial artery fistula has not been established,

the fistula should be treated if a patient shows

symptoms, in order to prevent lethal complications

such as myocardial infarction, infective endocarditis,

and aneurysmal rupture. Percutaneous transcatheter

embolization may be the treatment of choice in most

patients without concomitant cardiac disease, where-

as surgical ligation is also effective in selected pa-

tients [2]. Patients with coronary-bronchial artery fis-

tula and concomitant cardiac disease need more con-

sideration for selecting the best treatment modality.

In our case, the patient experienced atrial fibrillation

as well as coronary-bronchial artery fistula. To man-

age both of these diseases at once, surgical ablation

and fistula revision under median sternotomy fol-

lowed by cardiopulmonary bypass was performed,

and the coronary-bronchial artery fistula was suc-

cessfully repaired.

Conflict of interest

No potential conflict of interest relevant to this ar-

ticle was reported.

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