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

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) [email protected]

© 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/

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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. R adiofrequency 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

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

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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.

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

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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

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

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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