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\begin{document}

\title{Ruptured aneurysm of coronary arteriovenous fistula complicated with
pericardial tamponade}



\author[1]{Tetsuro Uchida}%
\author[2]{Yoshinori Kuroda}%
\author[2]{Eiichi Ohba}%
\author[2]{Atsushi Yamashita}%
\author[3]{Shingo Nakai}%
\author[4]{Kimihiro Kobayashi}%
\author[1]{Tomonori Ochiai}%
\author[3]{Mitsuaki Sadahiro}%
\affil[1]{Yamagata University}%
\affil[2]{Yamagata University Faculty of Medicine}%
\affil[3]{Yamagata Daigaku Igakubu}%
\affil[4]{Yamagata University Hospital}%


\vspace{-1em}



  
  \date{May 25, 2024}


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\selectlanguage{english}
\begin{abstract}
A quarter of coronary arteriovenous fistulas may present aneurysmal
dilatation; however, spontaneous rupture of the aneurysm is rare. We
present a rare case of ruptured coronary artery aneurysm (CAA)
associated with coronary fistulas that presented cardiac tamponade.
Subsequent to pericardial drainage, surgical repair of the CAA
associated with coronary fistulas was performed electively. The aneurysm
was located on the left anterior aspect of the pulmonary artery trunk.
It communicated with both the left and right coronary arteries by
anomalous fistulous vessels that drained into the pulmonary artery
trunk. The feeding arteries and fistulous opening were ligated and
closed, respectively, from within the aneurysm. Intraoperative
fluorescence imaging was performed pre- and postoperatively to ensure no
residual coronary fistulas were present. Although the rupture of CAA
associated with coronary fistulas is rare, it should be considered as
one of the potential causes of acute cardiac tamponade.%
\end{abstract}%



\sloppy


\textbf{Introduction}

Coronary arteriovenous fistulas (CAVFs) are well-known, but are
relatively rare cardiac anomalies with an occurrence of 0.002\% of the
general population.\textsuperscript{1}Although approximately a quarter
of all CAVFs may become aneurysmal, its spontaneous rupture with CAVFs
is rare.\textsuperscript{2-6} Rupture of coronary aneurysm can be lethal
because of cardiac tamponade. Herein, we discuss a successful surgical
case of ruptured coronary artery aneurysm (CAA) associated with CAVFs
presenting with cardiac tamponade.

\textbf{Case Report}

A 48-year-old woman was referred to our institution for surgical
management of ruptured CAA associated with CAVFs. Four days before
referral, she had experienced chest discomfort followed by presyncope.
Following medical examination at the neighboring hospital, her condition
was diagnosed as cardiac tamponade due to rupture of the CAA with CAVFs.
Computed tomography (CT) scan revealed an aneurysmal formation measuring
3.0 \selectlanguage{ngerman}× 2.0 cm, which was located at the left anterior aspect of the
pulmonary artery trunk with a large amount of pericardial effusion (Fig.
1a). Pericardial drainage was performed and 100 mL of blood was
aspirated (Fig. 1b). Subsequently, her symptom improved immediately.
Additionally, CT scan revealed that the aneurysm was communicated with
the left and right coronary arteries (RCA) through anomalous fistulous
vessels that drained into the pulmonary artery trunk (Fig. 1c).
Fistulous vessels originating from proximal RCA and left anterior
descending artery (LAD) were confirmed by coronary angiography. With the
diagnosis of ruptured CAA with CAVFs, operation was urgently performed.

The patient underwent a median sternotomy. Surgical exposure revealed
mild fibrous adhesion mainly around the pulmonary artery and a
saccular-type mass measuring 3.0 cm in diameter at the left anterior
surface of the pulmonary artery (Fig. 2a). Prior to repair of the CAVFs,
intraoperative near-infrared fluorescence imaging of CAVFs was performed
using photodynamic eye (Hamamatsu Photonics, Hamamatsu,
Japan).\textsuperscript{7} Approximately 15 seconds after intravenous
injection of indocyanine green, abnormal fistulous vessels were
visualized at the anterior surface of the pulmonary artery (Fig. 3a,
3b). Cardiopulmonary bypass was established between the ascending aorta
and bicaval cannulation. Following aortic cross clamping, cardiac arrest
was achieved by antegrade cardioplegia. First, the feeding arteries
originating from the proximal RCA were ligated at their origin. Second,
the pulmonary artery was incised longitudinally and the fistulous
opening that communicated between the pulmonary artery and aneurysm was
closed with a pledgeted suture (Fig. 2b). The CAA was opened and the
drainage sites from the feeding arteries were identified by antegrade
infusion of cardioplegia (Fig. 2c). These drainage sites were closed
from within the aneurysm followed by aneurysmorrhaphy. Weaning from the
cardiopulmonary bypass was uneventful. After completion of surgical
repair, no residual CAVFs was confirmed from repeated fluorescence
imaging (Fig. 3c). The patient's postoperative recovery was uneventful
and she was discharged 7 days after surgery. Postoperative CT did not
reveal any evidence of residual fistulas. Recurrent CAVFs and CAA were
not observed during follow-up. Written informed consent was obtained
from the patient for the publication of this report.

\textbf{Discussion}

Aneurysmal change of CAVFs is common and 19-26\% of CAVFs are
complicated with saccular type CAA\textsuperscript{8}; however
spontaneous rupture of aneurysm is rare. Treatment of asymptomatic
patients with CAA associated with CAVFs is still debated given its
rarity. CAAs exceeding 30 mm in diameter are considered to be at the
risk of rupture\textsuperscript{9}; however, there are reports of
rupture in cases of aneurysms measuring 10 mm and 15
mm.\textsuperscript{10,11} Therefore, surgical intervention should be
considered for coronary aneurysm of any size with CAVFs to prevent
rupture of the aneurysm.

Cardiac tamponade with hemodynamic deterioration is often caused by
acute aortic dissection, free wall rupture due to acute myocardial
infarction, or a traumatic thoracic aortic injury. Some reports,
including our report, have described ruptured CAA with CAVFs resulting
in cardiac tamponade.\textsuperscript{11-13} In fact, there has been a
report of emergency surgery without a definitive
diagnosis.\textsuperscript{12} Therefore, rupture of CAA with CAVFs
should be considered as one of the potential causes of acute cardiac
tamponade.

Although prognosis after successful surgical closure of CAVFs is
excellent even in ruptured cases, long-term follow-up is essential,
because residual CAVFs and recurrences are reported with considerable
frequency.\textsuperscript{14} Residual and late recurrence of CAVFs are
considered to result from incomplete surgical
procedure.\textsuperscript{14} To avoid these complications, it is
important to ensure the absence of residual CAVFs. Fluorescence imaging
can furnish sharp images with notable spatial and temporal resolution.
Intraoperative fluorescence imaging is a safe, non-invasive, and useful
diagnostic tool for the identification of residual
CAVFs.\textsuperscript{7} The resulting images enable easy detection of
residual CAVFs in real time.



\textbf{Author Contributions:}

TU has a major contribution in writing the manuscript.

\textbf{References}

1. Dodge-Khatami A, Mavroudis C, Backer CL. Congenital heart surgery
nomenclature and database project: anomalies of the coronary arteries.
Ann Thorac Surg 2000;69:S270-297.

2. Said SA, el Gamal MI, van der Werf T. Coronary arteriovenous
fistulas: collective review and management of six new cases--changing
etiology, presentation, and treatment strategy. Clin Cardiol
1997;20:748-752.

3. Sugiyama K, Suzuki S, Kamiya K, Koizumi N, Ogino H. Ruptured coronary
artery aneurysm with pulmonary artery fistulae. J Card Surg
2017;32:799-800.

4. Uzuka T, Nakamura M, Nakajima T, Watanabe N, Fukazawa Y. Surgery of
giant right coronary artery aneurysm complicated with coronary artery
fistula to left ventricle. J Card Surg 2018;33:95-96.

5. Kumagai M, Takatoku K, Kawamoto A, Shinoda E, Nishizawa J. Giant
coronary artery aneurysm with a coronary artery fistula to the pulmonary
artery. J Card Surg 2018;33:131-132.

6. Uchida T, Hamasaki A, Kuroda Y, Ohba E, Yamashita A, Sadahiro M.
Novel surgical technique for coronary fistulas with proximal origin. Ann
Thorac Surg 2017;104:e261-e263.

7. Uchida T, Hamasaki A, Kuroda Y, Yamashita A, Sadahiro M. Surgical
management of proximal coronary arteriovenous fistulas using
intraoperative fluorescence imaging. J Card Surg 2018;33:836-839.

8. Urrutia-S CO, Falaschi G, Ott DA, Cooley DA. Surgical management of
56 patients with congenital coronary artery fistulas. Ann Thorac Surg
1983;35:300-307.

9. Takeuchi N, Takeda M, Nishibori Y, Maruyama T. A case report of
coronary arteriovenous fistulas with an unruptured coronary artery
aneurysm successfully treated by surgery. Case Rep Cardiol
2012;314685:1-6.

10. Sakao T, Tsunooka N, Nakagawa H, Kajiwara S. Ruptured saccular
aneurysm of a coronary artery to pulmonary artery fistula associated
with cardiac tamponade. Kyoubu Geka 2000;53:684-686.

11. Sakamoto S, Kanemitsu S, Shimpo H, Kurita T. Tiny ruptured coronary
aneurysm with coronary-pulmonary arterial fistula. Gen Thorac Cardiovasc
Surg 2019;67:1084-1086.

12. Kawano M, Wada T, Anai H, Shuto T, Miyamoto S. A case of ruptured
aneurysm of coronary-pulmonary artery fistula diagnosed after emergency
thoracotomy. Surg Case Rep 2018;4:24.

13. Shomura Y, Mizumoto T, Fujinaga K, Sawada Y, Ito H, Teranishi S.
Cardiac tamponade due to rupture of a giant coronary artery aneurysm
with a coronary arteruivenous fistula: a case report. Surg Case Rep
2019;5:40.

14. Cheung DL, Au WK, Cheung HH, Chiu CS, Lee WT. Coronary artery
fistulas: long-term results of surgical correction. Ann Thorac Surg
2001;71:190-195.



\textbf{Figure Legends}

\textbf{Figure 1:} CT images of CAA associated with coronary fistulas.

a) Coronary artery aneurysm measuring 3.0 × 2.0 cm located at the
left-anterior aspect of the pulmonary artery trunk (white arrow). b)
Pericardial effusion is noted. c) Three-dimensional CT image showing CAA
associated with coronary fistulas (white arrowheads).

\emph{Ao} , ascending aorta; \emph{An} , aneurysm; \emph{PA} , pulmonary
artery; \emph{RA} , right atrium; \emph{RV} , right ventricle

\textbf{Figure 2:} Intraoperative photographs.

a) CAA is located at lateral surface of the pulmonary trunk (dotted
area). b) Fistulous opening which communicates the pulmonary artery and
coronary aneurysm is observed (black arrow). c) From within the
aneurysm, drainage site of feeding arteries is closed (white arrow).

\emph{Ao} , ascending aorta; \emph{An} , aneurysm; \emph{PA} , pulmonary
artery; \emph{RA} , right atrium; \emph{RV} , right ventricle

\textbf{Figure 3:} Intraoperative fluorescence imaging.

\textbf{a, b)} Preoperative images showing anomalous fistulous vessels
in front of the pulmonary artery (white arrows).
\textbf{c)}Postoperative image showing ligated feeding arteries arising
from the proximal right coronary artery (black arrow). Anomalous
fistulous vessels and CAA are not visualized.

\emph{Ao} , ascending aorta; \emph{An} , aneurysm; \emph{PA} , pulmonary
artery; \emph{RA} , right atrium; \emph{RV} , right ventricle

\textbf{Hosted file}

\verb`Figure.pptx`  available at \url{https://authorea.com/users/312766/articles/443360-ruptured-aneurysm-of-coronary-arteriovenous-fistula-complicated-with-pericardial-tamponade}

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