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Three vessel coronary cameral fistulae associated with new onset atrial fibrillation and angina pectoris.
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PMID:  24716096     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Coronary cameral fistulas are abnormal communications between a coronary artery and a heart chamber or a great vessel which are reported in less than 0.1% of patients undergoing diagnostic coronary angiography. All three major coronary arteries are even less frequently involved in fistula formation as it is the case in our patient. A 68-year-old woman was admitted to cardiology clinic with complaints of exertional dyspnea and angina for two years and a new onset palpitation. Standard 12-lead electrocardiogram revealed atrial fibrillation (AF) with a ventricular rate of 114 beat/minute and accompanying T wave abnormalities and minimal ST-depression on lateral derivations. Transthoracic echocardiographic examination was normal except for diastolic dysfunction, minimally mitral regurgitation, and mild to moderate enlargement of the left atrium. Sinus rhythm was achieved by medical cardioversion with amiodarone infusion. Coronary angiography revealed diffuse and multiple coronary-left ventricle fistulas originating from the distal segments of both left and right coronary arterial systems without any stenosis in epicardial coronary arteries. The patient's symptoms resolved almost completely with medical therapy. High volume shunts via coronary artery to left ventricular microfistulas may lead to increased volume overload and subsequent increase in end-diastolic pressure of the left ventricle and may cause left atrial enlargement.
Authors:
Murat Yuksel; Abdulkadir Yildiz; Mustafa Oylumlu; Nihat Polat; Halit Acet; Necdet Ozaydogdu
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Publication Detail:
Type:  Journal Article     Date:  2014-02-19
Journal Detail:
Title:  Case reports in vascular medicine     Volume:  2014     ISSN:  2090-6986     ISO Abbreviation:  Case Rep Vasc Med     Publication Date:  2014  
Date Detail:
Created Date:  2014-04-09     Completed Date:  2014-04-09     Revised Date:  2014-04-11    
Medline Journal Info:
Nlm Unique ID:  101585357     Medline TA:  Case Rep Vasc Med     Country:  United States    
Other Details:
Languages:  eng     Pagination:  475325     Citation Subset:  -    
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Full Text
Journal Information
Journal ID (nlm-ta): Case Rep Vasc Med
Journal ID (iso-abbrev): Case Rep Vasc Med
Journal ID (publisher-id): CRIVAM
ISSN: 2090-6986
ISSN: 2090-6994
Publisher: Hindawi Publishing Corporation
Article Information
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Copyright © 2014 Murat Yuksel et al.
open-access:
Received Day: 10 Month: 11 Year: 2013
Accepted Day: 12 Month: 12 Year: 2013
Print publication date: Year: 2014
Electronic publication date: Day: 19 Month: 2 Year: 2014
Volume: 2014E-location ID: 475325
PubMed Id: 24716096
ID: 3971846
DOI: 10.1155/2014/475325

Three Vessel Coronary Cameral Fistulae Associated with New Onset Atrial Fibrillation and Angina Pectoris
Murat Yuksel12*
Abdulkadir Yildiz1
Mustafa Oylumlu1 http://orcid.org/0000-0003-2434-6690
Nihat Polat1
Halit Acet1
Necdet Ozaydogdu1
1Department of Cardiology, Faculty of Medicine, Dicle University, Diyarbakir, Turkey
2Dicle Üniversitesi, Kalp Hastanesi 1.Kat, Kardiyoloji Kliniği, Sur, 21280 Diyarbakir, Turkey
Correspondence: *Murat Yuksel: yukselmurat@yahoo.com
[other] Academic Editors: A. El-Menyar, P. Georgiadou, and H. Nakajima

1. Introduction

Coronary cameral fistulas (CCFs) are abnormal communications between a coronary artery and a heart chamber which are reported in less than 0.1% of patients undergoing diagnostic coronary angiography [1]. The patients are usually asymptomatic so these fistulas are detected incidentally during coronary angiography most of the time. However, if fistulas are widespread, they may cause exertional angina due to coronary steal phenomenon concomitant with left ventricular volume overload and left atrial enlargement. All three major coronary arteries are even less frequently involved in fistula formation.


2. Case Report

A 68-year-old woman was admitted to cardiology clinic with complaints of exertional dyspnea and angina for two years and a new onset palpitation. Physical examination was unremarkable except for irregular beats. Standard 12-lead electrocardiogram (ECG) revealed atrial fibrillation (AF) with a ventricular rate of 114 beat/minute and accompanying T wave abnormalities and minimal ST-depression on lateral derivations (Figure 1). The patient underwent transthoracic echocardiography (TTE) for the investigation of dyspnea and AF etiology. TTE examination was normal except for diastolic dysfunction and mild to moderate enlargement of left atrium (46 mm). Left atrial thrombus was ruled out with transesophageal echocardiography (TEE) and sinus rhythm was achieved by medical cardioversion with amiodarone infusion. Afterwards coronary angiography was performed which revealed diffuse and multiple coronary cameral fistulae originating from the distal segments of both left and right coronary arterial systems without any stenosis in epicardial coronary arteries (Video 1 and Video 2 in the Supplementary Material available online at http://dx.doi.org/10.1155/2014/475325) and significant amount of contrast media passing into left ventricular cavity through diffuse coronary cameral fistulae at each beat (Figure 2). Surgical or percutaneous closure of multiple and diffuse microfistulae is difficult technically. Also treatment of these fistulae is controversial [2]. A cardiovascular surgeon evaluated the patient and medical treatment was advised. Symptoms of our patient resolved almost completely under beta blocker therapy (50 mg of metoprolol daily). She was asymptomatic at the third month of followup.


3. Discussion

Coronary cameral fistulas (CCFs) are rare anomalies connecting coronary arteries to cardiac chambers or great vessels, which are detected rarely during routine angiographic evaluation. Frequency of congenital coronary fistulas is reported in 0.08% of 11350 adult patients undergoing diagnostic coronary angiography in Turkey [3] which is compatible with the literature (approximately 0.1%) [1]. The major sites of origin are the right coronary artery (55%), the left coronary artery system (35%), and both coronary arteries (5%). The main termination sites are the right ventricle (40%), right atrium (26%), and pulmonary arteries (17%). Less frequently they may drain into the superior vena cava or coronary sinus and least often into the left atrium or left ventricle [1, 4] as it is the case in our patient.

Although asymptomatic in the vast majority, CCFs may cause chronic myocardial ischemia and angina, congestive heart failure, myocardial infarction, pulmonary hypertension, rhythm disturbances, subacute bacterial endocarditis, thromboembolism, rarely rupture of aneurysmal segment, and sudden death [5]. Small fistulae usually do not cause any hemodynamic compromise. However, the larger and multiple fistulae may cause “coronary steal phenomenon” leading to myocardial ischemia [6]. The outcome with these connections depends upon the termination site. If termination is to the systemic venous side, it means a left-to-right shunt, and there will be left-sided volume overload when the termination is into left-sided cardiac structures. The volume of the shunt varies with the size of the fistula and the difference between systemic resistance and resistance in the terminating vessel/chamber. Blood flow moves from the coronary arteries to the lower pressure chambers/vessels. The best way to manage coronary cameral fistulae is not well known due to rarity of the condition [2]. Most patients are treated conservatively because symptoms relieve significantly with medical therapy especially beta blockers but not nitrates which may increase “coronary steal phenomenon” and worsen anginal symptoms in patients with CCFs [7].

To date there is only one case of CCF reported in association with paroxysmal AF [8]. In the present case without systemic hypertension, left atrial enlargement leading to AF may be due to increased volume overload passing through the coronary-cameral fistulae and subsequent increase in end-diastolic pressure of the left ventricle.


Supplementary Material

Video 1. Antero-posterior oblique projection of left coronary angiogram depicting the CCFs opacifying the left ventricular cavity mimicking a left ventriculography.

Video 2. Left oblique projection of right coronary angiogram demonstrating contrast media leaking into left ventricle from the distal segment of right coronary artery through CCFs.


Click here for additional data file (475325.f1.zip)


Conflict of Interests

The authors declare that there is no conflict of interests regarding the publication of this paper.


References
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2. Carrel T,Tkebuchava T,Jenni R,Arbenz U,Turina M. Congenital coronary fistulas in children and adults: diagnosis, surgical technique and resultsCardiologyYear: 19968743253302-s2.0-00299790478793168
3. Serçelik A,Mavi A,Ayalp R,Pestamalci T,Gümüsburun E,Batiraliev T. Congenital coronary artery fistulas in Turkish patients undergoing diagnostic cardiac angiographyInternational Journal of Clinical PracticeYear: 20035742802832-s2.0-003854160912800458
4. Ho HH,Cheung CW,Jim MH,Lam L. Images in cardiology: coronary-cameral fistulaHeartYear: 20059112p. 15402-s2.0-33644813012
5. Brooks CH,Bates PD. Coronary artery-left ventricular fistula with angina pectorisThe American Heart JournalYear: 198310624044062-s2.0-0020955986
6. Iadanza A,Del Pasqua A,Fineschi M,Pierli C. Three-vessel left-ventricular microfistulization syndrome: a rare case of anginaInternational Journal of CardiologyYear: 20049611091112-s2.0-294258870815203269
7. Heper G,Kose S. Increased myocardial ischemia during nitrate therapy: caused by multiple coronary artery-left ventricle fistulae?Texas Heart Institute JournalYear: 200532150522-s2.0-1464442390315902822
8. Isik T,Kurt M,Kaya A,Tanboga IH. Multiple septal coronary-cameral fistulae associated with paroxysmal atrial fibrillationTürk Kardiyoloji Derneği arşiviYear: 2012402p. 200

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