Atypical chest pain due to multiple coronary arteries fistulas occluded with percutaneous interlock coils: A case report

Abstract

Coronary artery fistulas, although rare, should be included in the differential diagnosis of atypical chest pain, generally unveiled by cardiac catheterization or multidetector computed tomography. Such anatomical findings in conjunction with detectable ischemia and severe symptoms should prompt their closure. Transcatheter closure of fistulas is an attractive alternative to surgery, especially with the novel devices such as the interlock fibered detachable coils, which can be safely and effectively performed in a variety of circumstances, including the coronary arteries with tortuous anatomies. We present a case of atypical chest pain and large burden of ischemia in the stress scintigraphy, due to multiple coronary fistulas to the bronchial arteries successfully occluded with percutaneous interlock coils.

<Learning objective: This report describes the feasibility and safety of multiple tortuous coronary-bronchial fistulas treated with the novel interlock fibered detachable coils, in a patient with prior thromboembolism. This is the first case report to use this device in this situation and shows that, in symptomatic patients with documented ischemia, such novel devices may help in treating coronary fistulas, even in tortuous anatomy.>

Introduction

Chest pain is a common complaint among middle-aged women and is often atypical and rarely associated with coronary artery disease. Differential diagnoses include various diseases and more infrequently coronary artery fistulas [1], [2], [3]. Coronary artery fistulas although rare, are generally disclosed by cardiac catheterization or multidetector computed tomography (MDCT) in the evaluation of chest pain. Such anatomical findings in conjunction with detectable ischemia and severe symptoms should prompt their closure [1], [4], [5], [6]. Transcatheter closure of fistulas is an attractive alternative to surgery, especially with novel devices. We describe a case of multiple tortuous fistulas treated with the novel interlock fibered detachable coils in a patient with prior thromboembolism.

Case report

A 46-year-old woman with anxiety disorders and no risk factor for coronary artery disease or use of any medication, presented with palpitations and atypical chest pain in the past 3 months. No abnormalities were revealed in the physical examination, chest X-ray, and electrocardiogram. In addition, an echocardiogram showed a preserved left ventricular ejection fraction, with mild mitral valve regurgitation and mild pulmonary hypertension. Due to persistence of the symptoms a stress 99mTc-sestamibi scintigraphy was performed under dipyridamole, and multiple perfusion defects were detected (anterolateral and inferolateral walls) leading to ischemia in >10% of the left ventricle (Fig. 1). Despite optimal medical therapy with beta-blockers and nitrates, she persisted with symptoms. Therefore, a cardiac catheterization was undertaken, revealing no coronary artery disease, but multiple coronary artery fistulas from the circumflex - and the left anterior descending arteries (LAD) to the right pulmonary circulation (Fig. 2; Video S1). Additional computed tomography (CT) scan revealed the thrombotic interruption of the proximal right pulmonary artery, with multiple systemic fistulas to the pulmonary circulation, in addition to the coronary fistulas, with reduced volume of the right lung (Fig. 3). The exact timing of the thrombosis of the proximal right pulmonary artery is unknown. The patient persisted with symptoms even after the association of calcium-channel blockers so that percutaneous closure of the fistulas was scheduled. Uneventful percutaneous closure of the three coronary artery fistulas was successfully performed, using the Fathom steerable guidewire for the placement of a Renegade microcatheter (Boston Scientific, Natick, MA, USA), followed by the position of multiple fibered interlock-18 coils (Boston Scientific) (Fig. 1; Video S2). The circumflex artery fistula and the septal one arising from the LAD were both closed with a 2D shape interlock coil of 3 mm × 6 cm, and 4 mm × 15 cm, respectively. The left obtuse marginal was closed with a diamond shape interlock coil of 2/3 × 2.3 cm. The patient was discharged two days following the procedure asymptomatic and at 12-month follow-up remained asymptomatic with a normal stress 99mTc-sestamibi scintigraphy (Fig. 1) and a control MDCT showing the proper closure of the coronary fistulas (Fig. 3).

Fig. 1.

(A) Stress 99mTc-sestamibi scintigraphy was performed under dipyridamole, and multiple perfusion defects were detected (anterolateral and inferolateral walls) leading to ischemia in >10% of the left ventricle. (B) After closure of the fistulas a repeated stress 99mTc-sestamibi scintigraphy showed no residual inducible ischemia under stress.

Fig. 2.

(A) Coronary angiography pre-procedure showing the multiple fistulas connecting the circumflex artery (white arrows) and the left anterior descending artery (blue arrow) with the bronchial arteries. (B) Even in tortuous anatomy the Renegade catheter and interlock fibered coils are able to reach; (C and D) Uneventful closure of the fistulas was performed using multiple interlock fibered coils for the circumflex artery (white arrows) and the left anterior descending artery (blue arrow).

Fig. 3.

(A) Computed tomography (CT) scan revealed the thrombotic interruption of the proximal right pulmonary artery (white arrow). (B) CT revealing multiple systemic fistulas to the right pulmonary circulation from the bronchial (blue arrow) and the phrenic circulation (white arrow). (C) Phrenic fistulas to the right lung (white arrows). (D) Control CT showing the coil placement with the proper closure of the coronary fistula (blue arrows).

Discussion

Chronic thromboembolic pulmonary hypertension (CTEPH) is a major cause of progressive pulmonary hypertension and right heart failure. Yet, systemic and coronary artery collaterals to the pulmonary circulation may help maintain pulmonary parenchymal viability, especially among patients with more significant pulmonary artery obstruction, in whom the prevalence of coronary fistulas is ∼11% [2]. This is similar to our case that presented proximal occlusion of the right pulmonary artery, with multiple systemic fistulas to the right pulmonary circulation, in addition to the coronary-bronchial fistulas. Of note, coronary-bronchial fistula is a rare finding (0.08%–0.6%) [3], [7], most commonly related to bronchiectasis [6], but other causes may also include pulmonary thromboembolism, such as in our case, but also Takayasu arteritis, pulmonary artery tumor, chronic bronchitis, tetralogy of Fallot with pulmonary atresia, hypoplasia of the pulmonary artery, supravalvular aortic stenosis, and pulmonary tuberculosis. Most of such cases are asymptomatic and unveiled as incidental finding from MDCT or angiography. Still, dyspnea, angina, arrhythmias may ensue, especially when this disorder induces coronary steal syndrome [2], [6], [8]. In such circumstances, surgical or percutaneous management should be evaluated, both with good results in prior small case series or case reports [2], [4], [5], [6], [7].

More recently, the interlocking detachable coils have been posed as an attractive alternative to the conventional coils in the treatment of a variety of pathologies, especially in congenital and peripheral interventions [1], [9], [10]. They are retrievable and can be introduced using 3Fr microcatheters coaxially used through a 5Fr guiding catheter, which provides a controlled method of delivery, in a variety of sizes, even in vessels with tortuous anatomies. Positioning and stability can be continually assessed throughout the deployment, until the coil is optimally placed. Furthermore, these coils contain polyethylene terephthalate fibers that promote thrombogenicity, permitting faster and more effective closure of the fistulas. The experience with such devices in the treatment of coronary artery fistulas is very limited in the literature so that we report herein a successful closure of multiple coronary artery fistulas in a highly symptomatic patient [9]. Despite the closure of the fistula, pulmonary parenchymal viability is maintained by multiple systemic fistulas as demonstrated in our case by CT. The present case highlights that this novel device may help in the less invasive treatment of such difficult anatomies and these results should be further evaluated in larger series, comparing the different devices.

In conclusion, coronary artery fistulas although rare, should be included in the differential diagnosis of atypical chest pain, generally unveiled by cardiac catheterization and/or MDCT. Such anatomical findings in conjunction with detectable ischemia and severe symptoms should prompt closure of the fistulas. Transcatheter closure of fistulas with the interlock fibered detachable coils can be safely and effectively performed in a variety of circumstances, including the coronary arteries.

Conflict of interest

The authors declare that there is no conflict of interest.

Funding sources

The authors declare that there are no funding sources.

Appendix A. Supplementary data

The following are Supplementary data to this article: