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\begin{document}

\title{Innovative method to diagnose Coronary Cameral fistula by Contrast
Echocardiography}



\author[1]{Moustafa Eldeib}%
\author[2]{Fatema Qaddoura}%
\author[2]{Marawan Sadek}%
\author[2]{Donya Alhassan}%
\author[3]{Reda  Abuelatta}%
\affil[1]{Al-Azhar University Faculty of Medicine}%
\affil[2]{King Fahd Military Medical Complex}%
\affil[3]{Madinah Cardiac Center}%


\vspace{-1em}



  
  \date{May 08, 2024}


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\selectlanguage{english}
\begin{abstract}
Introduction: Coronary artery fistula (CAF) is a rare cardiac anomaly
that typically presents as a continuous murmur in an otherwise
asymptomatic patient. Occasionally, it can result in congestive heart
failure or bacterial endocarditis. Objective: To better delineate the
course of coronary artery fistula for the first time, using the SonoVue
contrast agent during transthoracic echocardiography. Method and
results: A referred 46-year-old male with a known history of dyspnea on
exertion for a period of almost three months was admitted to the
hospital with progressive dyspnea, and assessed under suspicion of CAF.
CAF, was seen with a coronary angiogram, but the exact entry point in
the left ventricle or left atrial wall could not be determined. CT
angiography also failed to establish the exact point of entrance to LV
(left ventricle), so CAG (coronary angiography) was repeated with
SonoVue contrast agent injected into LM (Left main) while using a
Siemens echocardiography machine. Multiple views were obtained during
the injection, and revealed unusual flow in the left ventricle just
below the PML (posterior mitral leaflet) postero-lateral and passing
through the fistula to LV. Conclusion: Contrast Enhanced
Echocardiography is thought to be more sensitive and in affording an
accurate delineation of CAF drainage into cardiac chambers, and can be
utilized in a direct precise and safe way.%
\end{abstract}%



\sloppy


\textbf{Introduction}

Congenital CAF is an uncommon cardiac anomaly that usually presents with
a continuous murmur in otherwise asymptomatic patients (1).
Occasionally, it leads to congestive heart failure or bacterial
endocarditis (1-3). If fistulas remain undetected or untreated in
childhood, they reportedly (1, 3) become symptomatic more frequently in
adulthood due to chronic volume overload and myocardial ischemia.
Although spontaneous closure can occur sporadically (4, 5), elective
surgical closure is the preferred mode of treatment (1, 3,6,7).

A large fistula can occasionally appear as a mass lesion on
echocardiography, but cannot easily be confirmed. Here, we report a new
diagnostic approach to identifying CAF, using a contrast agent injected
into the left coronary artery and transthoracic echocardiography. The
case described related to a 46-year-old male suffering from dyspnea with
turbulent flow in the left ventricle.

\textbf{CASE REPORT}

A 46-year-old male was admitted to hospital with progressive dyspnea. He
reported suffering from dyspnea upon exertion for 3 months and had been
referred from another hospital for further review due to suspicion of
coronary fistula, which was present on a coronary angiogram. It had
previously not been possible to determine the exact entrance point in
the left ventricle or the left atrial wall. No abnormal findings had
been observed using an initial chest posterior-anterior view and
electrocardiogram.

\textbf{Investigations:} Transthoracic and trans-esophageal
echocardiography revealed severe LV systolic dysfunction, moderate MR
(Mitral Regurge), and with agitated saline small PFO (patent foramen
ovale) was observed with TEE (Trans-Esophageal Echocardiography) between
the LA (Left Atrium) and the RA (Right Atrium).

CT angiography was performed, delineating the course of the fistula, but
did not provide detailed information about the site of drainage into the
cardiac chamber (figure 4).

Therefore, a further coronary angiogram was performed (figure 5) using
the SonoVue contrast agent (injection of 1.0 ml SonoVue diluted with 9.0
ml normal saline, and injected into LM), at the same time as using a
Siemens echocardiography machine. Multiple views were obtained with the
injection, and found unusual flow in the left ventricle just below the
PML postero-lateral and passing through fistula to LV (figures 1 and 3 )
and at the systolic phase with MR to LA and through PFO to RA and RV
(figure 2).

\textbf{Treatment:} After consultation with the heart team, the decision
was made to close it. The antegrade approach was used with percutaneous
closure with coils. Immediately after closure, the patient's MR
improved.

\textbf{Discussion}

A CAF affects communication between one of the coronary arteries and a
cardiac chamber or vein. CAFs are present in 0.2\% of patients
undergoing diagnostic cardiac catheterization. (8)

The right and left coronary arteries are involved in 55 and 35\% of
cases, respectively, with involvement of both vessels in just 5\% of
cases. (2) The arteries serve as a shunt between the coronary system and
the cardiac chamber into which they drain. The most common drainage
sites are at the right ventricle ventricular fistulae and are
exceedingly rare with the incidence being reported as 1.2\% of all
coronary artery fistulae. (9) Large shunts may present with pulmonary
edema, pulmonary hypertension, infective endocarditis, rupture, or
thrombosis of the fistula, and associated arterial aneurysm or
myocardial ischemia distal to the fistula (`myocardial steal
phenomenon'.) (9)

There are several possible reasons why CAF cannot be visualized directly
using transthoracic echocardiography. The CAF may be distally positioned
in the coronary circulation, and could be of smaller caliber. This can
make them much more difficult to detect, particularly during ventricular
systole. However, it is possible to visualize them with contrast when a
higher MI (Mechanical Index) is used. This supports visualization of
both the myocardial tissue and the contrast agent simultaneously.

The safety of contrast agents (including SonoVue) has been reported
previously. It has been used safely during septal ablation in patients
with hypertrophic obstructive cardiomyopathy (14). These agents have a
good safety profile for use in both cardiac and abdominal ultrasound
applications. The incidence of severe adverse reactions to ultrasound
contrast agents is no greater, and may be lower than that reported for
the contrast agents commonly used in other cardiac imaging tests. (8)

Previously, CAF have been diagnosed with aortography, (10) coronary
angiography, (11) and coronary CT. (13)~Although there has been a case
of color Doppler assessment of a CAF, (12) we cannot readily confirm
them with echocardiography. In this study, we employed a new and simple
diagnostic approach for CAF using a contrast agent. This assisted with
the diagnosis, because it clarifies typical turbulence flow, as it
appears in the left ventricle. The use of intraprocedural contrast
echocardiography results in an improved procedure and thereby patient
outcomes, by shortening the procedure and fluoroscopy time. Contrast
echocardiography at the time of aortography proves extremely valuable in
both opacifying the coronary arteries and determining which chambers the
fistula connects to.

The value of localizing the site of drainage of the fistula is high to
the interventional cardiologist, when attempting to close it.

\textbf{Conclusion}

Contrast enhanced echocardiography has been used to diagnose CAF in
adults, and is thought to be highly sensitive and provide precise and
accurate delineation of the CAF drainage into cardiac chambers safely.

This research did not receive any specific grant from any funding agency
in the public, commercial or not-for-profit sector.

There is no conflict of interest.

The co-author confirms my findings, and gave me permission and
responsibility to report on the patient.

\textbf{References:}

1. Neufeld HN, Schneeweis A, eds. Congenital coronary artery fistula.
In: Coronary Artery Disease in Infants and Children. Philadelphia: Lea
\& Febiger, 1983:31-58.

2. Start TJ, Bowman FO, Hordof AJ. Congestive heart failure in a newborn
secondary to coronary artery-left ventricular fistula. Am J Cardiol
1986; 58:366-7.

3. Liberthson RR, Sagar K, Berkoben JP, Weintraub RM, Levine FH.
Congenital coronary arteriovenous fistula. Circulation 1979;59:849-53.

4. Hackett D, Hallidic-Smith KA. Spontaneous closure of coronary artery
fistula. Br Heart J 1984;52:477-9.

5. Mahoney LT, Schieken RM, Lauer RM. Spontaneous closure of a coronary
artery ftstula in childhood. Pediatr Cardiol 1982;2:31 l-2.

6. Lowe JE, Newland Oldham H, Sabiston DC. Surgical management of
congenital coronary artery fistulas. Ann Surg 1981;194:373-80.

7. Kirklin JW, Barratt-Boyes BG. Congenital anomalies of the coronary
arteries. In: Kirklin JW, Barratt-Boyes BG, eds: Cardiac Surgery. New
York: John Wiley \& Sons, 1986:945-55.

8. Bulut S, Al Hashimi HMM, Gehlmann HR. Multiple Coronary Artery
Fistulae or Thebesian Veins? Heart 2007;93:1267.

9. Armsby LR, Keane JF, Sherwood MC, Forbess JM, Perry SB, Lock JE.
Management of coronary artey fistulae: patient selection and results of
transcatheter closure. J Am Coll Cardiol 2002;39;1026--32.

10. Kim YH, Lee SH, Kang CH, et al. A case report of coronary
arteriovenous fistula. Korean Circ J. 1982;12:189--192.

11. Lee SJ, Her SH, Jin SW, et al. A case of bilateral coronary to
pulmonary artery fistulas associated with severe aortic regurgitation.
Korean Circ J. 2008;38:331--334.

12. Choi SH, Seo HS, Oh SJ, et al. A case of multiple coronary
artery-left ventricular microfistulae demonstrated by transthoracic
Doppler echocardiography. Korean Circ J. 2003;33:338--342.

13.~Zenooz NA, Habibi R, Mammen L, Finn JP, Gilkeson RC. Coronary artery
fistulas: CT findings.~Radiographics.~2009;29:781--789.~{[}PubMed{]}.

14. Ehab Cherif, Sami Ghazal. Alcohol Septal Ablation after Suboptimal
Surgical septal Myomectomy.Case Journal 2018.

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