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Multiple Fistula Emptying into the Left Ventricle through the Entire Left Ventricular Wall.
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MedLine Citation:
PMID:  22787530     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Coronary artery fistulae are usually identified during invasive coronary angiographies. However, in this case, we made the early detection of coronary artery fistulae during non-invasive transthoracic echocardiography, by demonstrating diastolic multiple abnormal color Doppler flows on the entire left ventricular walls including left ventricular free wall, interventricular septum and apex, which were mimicking firecracker on the whole left ventricle. Fistulous communication from the coronary artery to the left ventricle is rare. Moreover, a case of multiple coronary fistulae emptying into the left ventricle through the entire left ventricular walls including left ventricular free wall, interventricular septum and apex is uncommon. We report a case of a 31-year-old woman who was diagnosed with multiple fistula communicating with entire left ventricular wall.
Authors:
Dong-Yeub Lee; Seon Hee Park; Myeong Hwan Bae; Jang Hun Lee; Dong Heon Yang; Hun-Sik Park; Yongkeun Cho; Shung-Chull Chae; Jae-Eun Jun
Publication Detail:
Type:  Journal Article     Date:  2012-06-25
Journal Detail:
Title:  Journal of cardiovascular ultrasound     Volume:  20     ISSN:  2005-9655     ISO Abbreviation:  J Cardiovasc Ultrasound     Publication Date:  2012 Jun 
Date Detail:
Created Date:  2012-07-12     Completed Date:  2012-10-02     Revised Date:  2013-05-30    
Medline Journal Info:
Nlm Unique ID:  101477138     Medline TA:  J Cardiovasc Ultrasound     Country:  Korea (South)    
Other Details:
Languages:  eng     Pagination:  108-11     Citation Subset:  -    
Affiliation:
Division of Cardiology, Department of Internal Medicine, Kyungpook National University Hospital, Daegu, Korea.
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Journal Information
Journal ID (nlm-ta): J Cardiovasc Ultrasound
Journal ID (iso-abbrev): J Cardiovasc Ultrasound
Journal ID (publisher-id): JCU
ISSN: 1975-4612
ISSN: 2005-9655
Publisher: Korean Society of Echocardiography
Article Information
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Copyright © 2012 Korean Society of Echocardiography
open-access:
Received Day: 15 Month: 3 Year: 2012
Revision Received Day: 16 Month: 4 Year: 2012
Accepted Day: 15 Month: 5 Year: 2012
Print publication date: Month: 6 Year: 2012
Electronic publication date: Day: 25 Month: 6 Year: 2012
Volume: 20 Issue: 2
First Page: 108 Last Page: 111
ID: 3391627
PubMed Id: 22787530
DOI: 10.4250/jcu.2012.20.2.108

Multiple Fistula Emptying into the Left Ventricle through the Entire Left Ventricular Wall
Dong-Yeub Lee, MDA1
Seon Hee Park, MDA1
Myeong Hwan Bae, MDA1
Jang Hun Lee, MDA1
Dong Heon Yang, MDA1
Hun-Sik Park, MDA1
Yongkeun Cho, MDA1
Shung-Chull Chae, MDA1
Jae-Eun Jun, MDA1
Division of Cardiology, Department of Internal Medicine, Kyungpook National University Hospital, Daegu, Korea.
Correspondence: Address for Correspondence: Dong Heon Yang, Division of Cardiology, Department of Internal Medicine, Kyungpook National University Hospital, 130 Dongdeok-ro, Jung-gu, Daegu 700-721, Korea. Tel: +82-53-200-5524, Fax: +82-53-426-2046, ddhyang@knu.ac.kr

INTRODUCTION

Coronary artery fistulae are direct connections from one or more coronary arteries to cardiac chambers or a large vessel. Congenital anomalies are more common than acquired,1), 2) being found in up to 0.2% of routine angiocardiography.3) Most reported cases are about coronary artery fistulae to right cardiac structure,4) but the fistula between coronary artery and left ventricle is considerably rare.4-6) Moreover, a case of multiple fistula emptied into the left ventricle through the entire left ventricular walls including left ventricular free wall, interventricular septum and apex is uncommon. The following we presented is such a case, with impressive echocardiac images which demonstrating diastolic massive abnormal color Doppler flows on the entire left ventricular walls which were mimicking firecracker on the whole left ventricle.


CASE

A 31-year-old woman was referred from local clinic for further investigation of cardiac murmur. Past health status was good. Physical examination revealed a blood pressure of 107/43 mmHg, and regular pulse of 86 beats/min. There was grade III/VI diastolic murmur from base to apex of heart. Electrocardiography demonstrated biphasic T wave in lead V1-3 and borderline prolongation of QT interval (Fig. l). Transthoracic echocardiography demonstrated a normal functioning left ventricle with hypertrophy and trabeculation of left ventricular free wall and interventricular septum (Fig. 2). Imaging with color flow Doppler demonstrated multiple sparkling colors Doppler flow through left ventricular free wall and interventricular septum during diastole (using a 4-6 MHz-frequency transducer and high Nyquist limit: 74), and these multiple sparkling color flows demonstrated multiple fistula formation from the epicardial surface into the entire left ventricular cavity through the hypertrophied segment of myocardium during diastole (Fig. 2). These communications have plexiform pattern forming arteriosinusoidal vessels along interventricular septum. It also showed suspicious non-compacting myocardium at apical infero-posterior wall of left ventricle. We observed two separated layers of myocardium with a thin, compacted, epicardial layer and a thick, non-compacted endocardial zone. The ratio of non-compacted myocardium to compact myocardium at the end of systole was 2.9 : 1 with mean non-compacted myocardium of 1.3 cm and compacted myocardium thickness mean of 0.4 cm. For detailed anatomical information, multidetector computed tomography coronary angiography is carried out. The computerized tomographic (CT) angiography also identified multiple fistula from the left coronary artery towards the entire left ventricular walls including left ventricular free wall, interventricular septum and apex. Coronary artery had markedly tortuous and dilated morphology from left main coronary artery to left anterior descending artery. There was no coronary anomaly in other cardiac structures (Fig. 3).


DISCUSSION

Coronary artery fistulae are rare and are most often diagnosed by angiography incidentally. In our case, because of the prominent firecracker on the whole left ventricle, we could make the early detection of coronary fistulae by noninvasive transthoracic echocardiography without invasive coronary angiography. Imaging with color flow Doppler, using a 4-7 MHz-frequency transducer and high Nyquist limit revealed unusual multiple high velocity color-flows from the epicardial surface into the left ventricular cavity during diastole. It seemed that those multiple diastolic mosaic flows reflect the presence of fistula between epicardial coronary system and left ventricular cavity. Left ventricular walls including left ventricular free wall and interventricular septum had sinusoidal appearance and mesh like structure which were plexus of multiple small vessels emptying exclusively into the left ventricle through the whole left ventricular wall, and also she had marked trabeculations and deep sinusoidal recesses involving the apex, apico-lateral and apico-inferior walls. This exam revealed two separated layers of myocardium with the ratio of non-compacted to compacted myocardium was greater than 2, measured at end systole in the parasternal short-axis view. The definition and diagnostic criteria of non-compaction is not yet fully established, but non-compaction is generally characterized by continuity between the ventricular cavity and the intertrabecular recesses, which are filled with blood from the ventricle and which have no communication with the epicardial coronary system. However, in the presented case, which had multiple direct communications with the epicardial coronary system through the entire left ventricular wall. In that point, we concluded this case is more compatible with multiple fistula than non-compaction. Although the etiology of non-compaction has not been fully investigated, our case is thought to result from an arrest in the regression of embryonic sinusoids during the embryonic period. The Arrest of myocardial compaction that occurs in the early stages of fetal development result in the persistence of intertrabecular spaces and incomplete formation of capillaries. In this case, we believed that this process have a role in the formation of multiple fistula with the ventricular cavity and epicardial coronary circulation. But there was no conclusive proof of the etiology of this case. The main limitation of our case was that we could not perform angiography to confirm coronary artery fistula. But CT angiographic finding was also compatible with multiple fistula draining into the left ventricle. In the presented case, the left ventricular functions were preserved, although the fistulae were very extensive. In a patient with coronary artery fistulae, myocardial ischemia and diastolic volume overload of the left ventricle can be caused by left-to-left shunt: however, the clinical and hemodynamic consequences are incompletely understood. In the presented case, the patient had no symptom and refused further evaluations including coronary angiography and catheterization. With regard to this special case, due to the fact of patient's refusal and absence of angina, it was decided clinical follow-up of the patient. She remained asymptomatic and no complicate events were recorded in the next 5-year follow-up. In conclusion, this case report represents multiple fistula emptied into the left ventricle through the entire left ventricular walls including left ventricular free wall, interventricular septum and apex, confirmed by transthoracic echocardiography and CT angiography.


References
1. Parra-Bravo JR,Beirana-Palencia LG. [Right coronary artery fistula opening into right ventricle. Echocardiographic findings and interventional management. Report of a case]Arch Cardiol MexYear: 20037320521114635481
2. Chen ML,Lo HS,Su HY,Chao IM. Coronary artery fistula: assessment with multidetector computed tomography and stress myocardial single photon emission computed tomographyClin Nucl MedYear: 200934969819352262
3. Abelin AP,Sarmento-Leite R,Quadros AS,Gottschall CAM. Coronary fistulaRev Bras Cardiol InvasYear: 200816242243
4. Hobbs RE,Millit HD,Raghavan PV,Moodie DS,Sheldon WC. Coronary artery fistulae: a 10-year reviewCleve Clin QYear: 1982491911977168909
5. Levin DC,Fellows KE,Abrams HL. Hemodynamically significant primary anomalies of the coronary arteries. Angiographic aspectsCirculationYear: 1978582534348342
6. Sherwood MC,Rockenmacher S,Colan SD,Geva T. Prognostic significance of clinically silent coronary artery fistulasAm J CardiolYear: 19998340741110072233

Article Categories:
  • Case Report

Keywords: Multiple fistula, Left ventricle, Firecracker.

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