International Journal of Cardiology 177 (2014) e120–e124
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Letter to the Editor
Left circumflex coronary–pulmonary artery fistula and transmediastinal participation of bronchial arteries best shown by CT☆ Farhood Saremi a,⁎, Ajay Patel b, David Shavelle c, James R. Licht b, Joseph Kang a a b c
Department of Radiology, University of Southern California, United States Department of Medicine, University of California Irvine, United States Department of Medicine, University of Southern California, United States
a r t i c l e
i n f o
Article history: Received 3 September 2014 Accepted 20 September 2014 Available online 28 September 2014 Keywords: Coronary fistula Bronchial artery Coronary CT angiography Vascular malformations
Fistulous communication of the coronary arteries with the pulmonary arteries in adults is a common type of coronary artery fistula [1–3]. In most reported cases, the fistula usually arise from the proximal left and/or right coronary arteries via the anterior conal branches that connect to the anterior wall of the main pulmonary artery. These anteriorly located abnormal communications are usually asymptomatic and are incidentally found during transcatheter or CT angiography (CTA) of the coronary arteries in 0.2–0.3% of studies [1,2]. Another type of fistula between the coronary and pulmonary arteries occurs posteriorly at the base of the heart, mainly within the transverse sinus [3]. In this rare type, fistulous communications exist with the branch pulmonary arteries, specifically the right pulmonary artery (RPA), via small atrial branches arising directly from the left circumflex (LCx) or the left sinuatrial (SA) node artery. Participation of one or more of the bronchial arteries in the arterial supply of these complicated fistulas is very common and can be easily missed in routine catheter angiographies. Although reports exist describing the collateral communication of the bronchial arteries with stenotic coronary arteries
☆ We attest that the article is the Authors' original work, has not received prior publication and is not under consideration for publication elsewhere. We adhere to the statement of ethical publishing as appears in the International of Cardiology. ⁎ Corresponding author at: Department of Radiology, University of Southern California, USC University Hospital, 1500 San Pablo St., Los Angeles CA 90033, United States. Tel.: +1 323 442 8541. E-mail address:
[email protected] (F. Saremi).
http://dx.doi.org/10.1016/j.ijcard.2014.09.078 0167-5273/Published by Elsevier Ireland Ltd.
[4,5], to our knowledge no clear description of the posteriorly located coronary fistulas with the RPA and indirect participation of the bronchial arteries has been documented. In this review three cases of posteriorly located coronary fistulas, via the LCx artery branches, to the RPA will be reported using CTA and coronary catheterization angiography. The extracardiac arterial supply of the bronchial arteries in these complex fistulas will also be shown by CT angiography. Case 1 (Fig. 1): A 61 year old man presents to his primary cardiologist office with history of recent non-ST elevation myocardial infarction. Five days prior to presentation the patient had an episode of chest pain and dyspnea with excessive exertion. He presented to a local physician and was noted to have elevated troponin and negative EKG stress test. Past medical history was significant for chronic atypical chest pain, ankylosing spondylitis, hypertension, hypercholesterolemia, pre-diabetes and a family history of coronary artery disease. He subsequently underwent coronary angiogram and was found to have a large coronary artery fistula involving the LCx. He was also noted to have a significant mid left anterior descending (LAD) artery narrowing, estimated to be 80%. He was found to have a normal echocardiogram with normal left ventricular function. The right heart catheterization revealed pulmonary blood flow (Qp) to systemic blood flow (Qs) ratio (Qp/Qs) of 1.38. He subsequently underwent percutaneous coronary intervention of his mid LAD artery with two drug eluting stents. He then underwent a cardiac CTA which revealed communications of collateral bronchial/mediastinal arteries with an enlarged atrial branch (left SA node artery) of the LCx to the RPA and the left atrium (LA) on the anterior wall. The patient was scheduled for outpatient follow-up. Case 2 (Fig. 2): A 60 year old female with past medical history for an atrial septal defect (ASD) and coronary artery fistula connecting to the pulmonary artery and LA. At a multidisciplinary conference three years earlier, the decision was made to close the ASD percutaneously if the patient developed a decreased exercise tolerance. The patient was currently asymptomatic and was therefore treated conservatively. An exercise treadmill stress test showed good exercise capacity. The most recent echocardiography showed a secundum type ASD which measured 1.6 cm at its maximum diameter with a left to right shunting and Qp/Qs of 1.8. The right ventricle was mildly dilated with normal systolic function. The right ventricular systolic pressure was estimated at 30–35 mm Hg. The left ventricle was normal in structure and
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Fig. 1. Case 1: A. and B. Coronal CTA images show a tangle of enlarged vessels around the superior and inferior walls of the left atrium (LA) arising from the left circumflex (LCx) artery (A) and bronchial artery (A and C). The origin of the bronchial artery from the descending aorta (DA) is shown in B. C. Axial CT at the level of the right pulmonary artery (RPA) shows fistulous communication (short arrows) to the anterior wall of the RPA. D. Volume rendered view of the cardiac base show extensive collateral vessels around the LA. E. Anterior view of the left coronary angiogram shows atrial arteries arising from the LCx. F. Left anterior descending (LAO) view shows contrast pool in an aneurysm (green arrows) before connecting (red arrow) to the RPA.
function. There was no significant valve dysfunction. There was an abnormal turbulant Doppler pattern suggestive of an atrioventricular malformation interposed between the LA, superior vena cava (SVC), and aorta. Serial echocardiographic studies over the last three years did not show significant changes. CT of the heart showed the ASD and mild right ventricular enlargement. A wide tangle of vessels suggestive of a vascular malformation was present in the middle mediastinum extending above the LA and behind the aortic root, filling the entire transverse sinus. Feeding arteries in the order of 2–3 mm appeared to be, a large tortuous branch arising from the proximal LCx that traveled in the transverse sinus toward the SVC, mildly enlarged bronchial arteries, and a prominent right SA node artery. Fistulous communications were seen with right superior pulmonary vein near confluence with the LA, at posterior and superior left atrial walls, and the RPA. No significant atherosclerotic narrowing was present in any of the coronary arteries. The lungs appeared clear. Cardiac catheterization found the arteriovenous fistula with connections from the LCx and right coronary artery to the RPA and the LA. Case 3 (Fig. 3): A 58 year old female with history of mild pulmonary hypertension and a nonspecific chest pain. The stress echocardiography after dobutamine infusion was normal and there was no stress induced chest pain. Coronary CTA showed two atrial branches arising from the proximal LCx with one passing along the right posterior wall of the LA and the second one along the left lateral wall of the LA. Both arteries entered the transverse sinus to connect to the anterolateral wall of the RPA. Two additional enlarged feeding vessels were seen arising from the inferior wall of the aortic arch (bronchial arteries) to join
the atrial branches within the transverse sinus. The RPA was mildly enlarged. The posteriorly located (retrotruncal) coronary–pulmonary artery fistula has certain anatomical differences with those occurring anterior to the main pulmonary artery (pretruncal). Common findings in both types include arterial supply by the conotruncal and atrial branches of the coronary arteries [3]. Extracardiac vascular supply including bronchial arterial supply is more common in posteriorly located fistulas but can be seen in anterior lesion in 10% of cases [2] (Fig. 4). The drainage in the anterior type usually occurs into the main pulmonary artery and in the posterior type into the RPA. Aneurysmal formation at the entrance of the vascular malformation to the pulmonary artery is common [2] and seen in all of the presented cases. For the posterior type, it is very common to see additional drainage routes into the LA and the confluence of the pulmonary veins. These differences may have important clinical implication when treatment with transcatheter embolization is considered [6]. In adults, these vascular anomalies are usually supplied by atrial branches of the left coronary system directly arising from the LCx artery and branches of the left SA node artery, Kugel's interatrial anastomotic pathway, or right superior septal artery [3]. Although symptomatic congenital fistulas are reported in children and especially in ones with congenital heart malformation [7], most of these fistula in adults usually have little hemodynamic importance and are simply discovered during angiographic studies. Pulmonary hypertension, coronary obstruction and chronic lung disease may contribute to the development or formation of these malformations by recruiting preexisting mediastinal and atrial vessels [3].
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Fig. 2. Case 2. Color coded volume rendered CTA images in superior (top row) and anterior (middle row) cardiac orientations show extensive vascular malformation along superior aspect of the left atrium (LA, in yellow). Enlarged tortuous atrial branches arising from the left circumflex artery (LCx) are shown in purple. This vascular anomaly extends to the right through the transverse sinus and connects to the right superior pulmonary vein (RSPV) and the inferior aspect of the right pulmonary artery (RPA). Note aneurysmal dilatation at the fistulous connection sites (red arrows). Also shown are enlarged right sinuatrial node arteries (R-SANA) in red and left SANa (L-SANa) in green, both participating in the arterial supply to the vascular anomaly. Anterior body view images of coronary catheterization (lower row) show the findings described above. MPA = main pulmonary artery.
Enlargement of the bronchial arteries is common in patients with chronic inflammation of the tracheobronchial tree, COPD, chronic thromboembolism, long standing pulmonary hypertension and pulmonary artery stenosis/atresia [3,8]. Two of our patients had pulmonary hypertension; one secondary to an ASD and the second one was idiopathic. One of our cases has chronic interstitial and bronchial wall thickening related to the ankylosing spondylitis. Bronchial artery enlargement may also contribute to the coronary vascular supply when the flow is compromised [5]. Bronchial anastomosis with the coronary arteries occurs through a pericardial reflection
from superior and posterior mediastinum. Connections occur commonly with the left atrial branches of the coronary arteries in the posterior mediastinum. These minute mediastinal anastomotic network serve as collateral between the coronary and pulmonary arterial branches and may enlarge in patients with coronary artery stenosis (bronchial steal) or pulmonary diseases (coronary steal) such as chronic bronchiectasis (cystic fibrosis, tuberculosis) and pulmonary artery stenosis/ atresia (TOF) [3,9]. Coronary chest pain can develop when coronary steal occurs in the presence of coronary artery stenosis as seen in our case #1.
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Fig. 3. Case 3. Right lateral (upper row) and posterior (lower row) color coded volume rendered CT images of the aorta (red), pulmonary artery (blue) and left atrium (pink). Coronal and axial CT images at the level of the right pulmonary artery (RPA) also are shown. Anomalous vessels are color coded. Two atrial branches (#1 and #2) arising from the left circumflex artery (LCx) are shown in green. One (#2) travels along the right posterior wall of the left atrium (LA) and the second (#1) along the left lateral wall of the LA, both entering the transverse sinus to connect, with other vessels, to the anterolateral wall (yellow arrows) of the RPA. Other feeding vessels arising from the inferior wall of the aortic arch (bronchial arteries) are colored in blue (#4) and pink (#3). These two bronchial arteries gave off branches, in transverse sinus, to connect with the RPA. The RPA is mildly enlarged. AA = ascending aorta, Ao = aorta.
Fig. 4. 60 year old male. An example of the anteriorly located coronary to pulmonary artery fistula shown with color coded volume rendered CTA. Aneurysmal dilatation of the vessels before entering the main pulmonary artery is shown in purple. Note major arterial supply by the left anterior conal artery (#1) from the left anterior descending (LAD) artery. Smaller arterial supply is provided by the bronchial arteries (#2) and the right anterior conal artery (#3). AA = ascending aorta, MPA = main pulmonary artery, LCx = left circumflex artery, LAO = left anterior oblique, RCA = right coronary artery, RV = right ventricle.
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Disclosures 1) All authors have read and approved the manuscript. 2) No potential conflict of interest for authors. References [1] Kim MS, Jung JI, Chun HJ. Coronary to pulmonary artery fistula: morphologic features at multidetector CT. Int J Cardiovasc Imaging 2010;26(Suppl. 2):273–80. [2] Zhang LJ, Zhou CS, Wang Y, et al. Prevalence and types of coronary to pulmonary artery fistula in a Chinese population at dual-source CT coronary angiography. Acta Radiol Nov 26 2013;55(9):1031–9. [3] Matsunaga N, Hayashi K, Sakamoto I, Ogawa Y, Matsuoka Y, Imamura T, et al. Coronaryto-pulmonary artery shunts via the bronchial artery: analysis of cineangiographic studies. Radiology 1993;186:877–82. [4] Bjork L. Angiographic demonstration of extracardial anastomoses to the coronary arteries. Radiology 1966;87:274–7.
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