Cite this article as: Hsiao H-Y, Lee W-C, Sheu J-J, Fang C-Y. Fistulae ligation and left main artery ligation for a bilateral giant coronary arterial fistulae-related aneur- ysm. Eur J Cardiothorac Surg 2019;55:798–9. Fistulae ligation and left main artery ligation for a bilateral giant coronary arterial fistulae-related aneurysm Hao-Yi Hsiao a , Wei-Chieh Lee a,† , Jiunn-Jye Sheu b and Chih-Yuan Fang a, * a Division of Cardiology, Department of Internal Medicine, Kaohsiung Chang Gung Memorial Hospital, Chang Gung University College of Medicine, Kaohsiung, Taiwan, Republic of China b Department of Cardiothoracic and Vascular Surgery, Kaohsiung Chang Gung Memorial Hospital, Chang Gung University College of Medicine, Kaohsiung, Taiwan, Republic of China * Corresponding author. Division of Cardiology, Department of Internal Medicine, Kaohsiung Chang Gung Memorial Hospital, Chang Gung University College of Medicine, 123 Ta Pei Road, Niao Sung District, Kaohsiung City, Taiwan 83301, Republic of China. Tel: +86-7-7317123, ext: 8300; fax: +86-7-7322402; e-mail: leeweichieh@yahoo.com.tw (C.-Y. Fang). Received 7 June 2018; received in revised form 4 July 2018; accepted 22 July 2018 Abstract Bilateral congenital coronary artery fistulae complicated with a giant coronary artery aneurysm is a very rare condition. A coronary artery aneurysm is a coronary artery dilatation that exceeds the diameter of normal adjacent segments or the diameter of the patient’s largest coronary vessel by 1.5 times. The complications associated with a coronary artery aneurysm include thrombosis, embolization, rupture, vasospasm, congestive heart failure and infectious endocarditis. We report on a 63-year-old woman presenting with severe heart failure related to bilateral coronary artery fistulae. A giant coronary aneurysm was noted in the right coronary artery, and a tortuous coronary ar- tery fistula was noted in the left coronary artery. Symptoms were relieved after surgical intervention for bilateral coronary artery fistulae. Keywords: Coronary arterial fistulae • Giant coronary aneurysm • Biventricular heart failure • Fistulae ligation • Left main artery ligation INTRODUCTION A coronary artery fistula is a rare coronary artery abnormality with an incidence ranging from 0.1% to 0.8% in adults in whom blood is shunted into a cardiac chamber, a great vessel or other structures [1]. A coronary artery fistula is associated with a high risk of cardiac complications, including heart failure, myocardial ischaemia, infective endocarditis, arrhythmias and rupture, espe- cially when combined with a coronary artery aneurysm or dilata- tion. Surgical resection may be necessary for concomitant coronary artery fistulae and a giant coronary aneurysm. Most coronary artery fistulae are incidentally identified, as they often do not cause haemodynamically significant shunts or symptoms, but in some cases where the shunt is huge, heart failure, arrhyth- mia or chest discomfort can occur owing to the coronary steal phenomenon [2]. CASE REPORT A 63-year-old woman, without underlying systemic disease, experienced exertional dyspnoea for many years. In the recent 6 months, she experienced orthopnoea and could not lie flat, and chest tightness during daily activities. Physical examination showed jugular vein engorgement, bilateral lower limb pitting oedema and Grade 3/6 continuous soft murmur over the right upper and left upper sternal borders. Chest radiography showed severe cardiomegaly and pulmonary congestion. Electrocardiography showed atrial fibrillation with rapid ventricu- lar rate. Transthoracic echocardiography showed very poor biventricular performance and turbulent flow in the pulmonary artery (PA). Coronary angiography was performed. A giant aneur- ysm was noted, originating from the proximal segment of the right coronary artery (RCA) and draining into the PA (Fig. 1A). The left coronary artery showed a tortuous fistula with drainage to the PA near the ostium of the left anterior descending artery (LAD) (Fig. 1B). A significant pulmonary-systemic shunt ratio was estimated to be 1.7. Cardiac computed tomography (CT) showed a giant aneurysm and a fistula from the proximal RCA into the PA and a torturous fistula from the proximal LAD into the PA (Fig. 1C and D). Because of biventricular failure, surgical correc- tion of bilateral coronary artery fistulae and the aneurysm was performed, even though operative risk was high. Intraoperatively, a giant aneurysm and tortuous fistulae were noted (Fig. 1E). For the fistula and the aneurysm in the RCA, we ligated and excised both proximal feeding arteries of the giant aneurysm and the dis- tal drainage vessels to the PA. For the tortuous fistula at the left coronary artery, we ligated the left main coronary artery, the proximal segment of the LAD and mid-segment of the left cir- cumflex artery. The left internal mammary artery was †The second author contributed equally as corresponding author. V C The Author(s) 2018. Published by Oxford University Press on behalf of the European Association for Cardio-Thoracic Surgery. All rights reserved. European Journal of Cardio-Thoracic Surgery 55 (2019) 798–799 CASE REPORT doi:10.1093/ejcts/ezy287 Advance Access publication 27 August 2018 Downloaded from https://academic.oup.com/ejcts/article/55/4/798/5084886 by guest on 24 February 2022