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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[3]{Reda  Abuelatta}%
\author[4]{Ayman Naguib}%
\affil[1]{Al-Azhar University Faculty of Medicine}%
\affil[2]{King Fahd Military Medical Complex}%
\affil[3]{Madinah Cardiac Center}%
\affil[4]{Zagazig University}%


\vspace{-1em}



  
  \date{June 17, 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 using an intracoronary injection of
SonoVue contrast agent, whilst performing transthoracic
echocardiography. Method and results: A referred 46-year-old male, with
a history of exertional dyspnea for 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 drainage site , so CAG
(coronary angiography) was repeated with the 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) and passing through the fistula to LV. Conclusion:
Contrast-Enhanced Echocardiography by direct intracoronary injection of
SonoVue contrast agent, is safe and can aid in the delineation of
fistula drainage.%
\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), Among the
treatment options, an elective surgical closure 1, 3,6,7) or
percutaneous closure in the cardiac catheterization laboratory. To
enable successful and safe closure, it is imperative to define the
anatomy of the fistula. (15)

A large fistula can occasionally appear as a mass lesion on
echocardiography, the course of the fistula can be difficult to confirm
by echocardiography. Here, we report a new diagnostic approach to
identifying CAF, using an intracoronary injection of SonoVue contrast
agent, whilst performing transthoracic echocardiography.

\textbf{CASE REPORT}

A 46-year-old male was admitted to the 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 drainage
site. 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 Regurgitation) (Figure 1), 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)
(Figure 2).

CT angiography was performed, but the results non informative about the
drainage site.

Therefore, a further coronary angiogram was performed (figure 3) 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 flow in the left ventricle just below the PML
passing through fistula to LV (figures 4) and at the systolic phase with
MR to LA and through PFO to RA and RV.

\textbf{Treatment:} After heart team discussion and Given symptoms of
heart failure attributed to a hemodynamically significant shunt, the
decision was made to close it. The antegrade approach was used with
percutaneous closure with coils (figure 5). Immediately after closure,
the patient's MR improved (figure 6).

\textbf{Discussion}

A CAF is defined as an abnormal communication between one of the
coronary arteries and a cardiac chamber o or another blood vessel. 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 the 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. 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 may not 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 the
visualization of both the myocardial tissue and the contrast agent
simultaneously.

Contrast agents (including SonoVue) have been reported previously as
safe for direct coronary artery injection. 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 has 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 by intracoronary injection of SonoVue
contrast agent is safe and can aid in the delineation of fistula
drainage.

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 fistula 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 artery 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 of 2018.

15. Al-Qahtani A, Zakaria A, Hijazi ZM. Current Interventional
Management Strategies for Coronary Arteriovenous Fistulae. Structural
Heart Disease 2018;4(5):212-221. DOI:~10.12945/j.jshd.2018.043.17

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