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\begin{document}

\title{Late detection of communication between juxtaposed atrial appendages}



\author[1]{Shinichi Ishida}%
\author[2]{Masato Mutsuga}%
\author[1]{Takashi Fujita}%
\author[1]{Kei Yagami}%
\affil[1]{Gifu Kenritsu Tajimi Byoin}%
\affil[2]{Nagoya University Graduate School of Medicine Faculty of Medicine}%


\vspace{-1em}



  
  \date{May 24, 2024}


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\selectlanguage{english}
\begin{abstract}
A 40-year-old woman underwent an atrial septal defect closure with
juxtaposition of the atrial appendages without communication between
both appendages 4 years before presentation. She recently experienced
desaturation on exercise, and right-to-left shunt coming from the
residual communication between both appendages was found. The residual
communication was closed from the right to the left atrium. Herein, we
report a rare case of juxtaposition of the atrial appendages with
residual communication between them after an atrial septal defect
closure.%
\end{abstract}%



\sloppy


Title:Late detection of communication between juxtaposed atrial
appendages

Authors:

Shinichi Ishida\textsuperscript{1}, Masato Mutsuga\textsuperscript{2},
Takashi Fujita\textsuperscript{1}, Kei Yagami\textsuperscript{1}

\textsuperscript{1}Department of Cardiac Surgery, Gifu Prefectural
Tajimi Hospital, Tajimi, Gifu, Japan.

\textsuperscript{2}Department of Cardiac Surgery, Nagoya University
Graduate School of Medicine, Nagoya, Aichi, Japan.

Short running title: juxtaposed atrial appendages

Keywords: atrial septal defect, juxtaposition of the atrial appendages,
residual communication

Acknowledgement: There are no sources of funding for this manuscript.

Conflicts of Interest: The authors declare that there are no conflict of
interests.

Corresponding Author: Shinichi Ishida

5-161 Maebata-cho, Tajimi-city, Gifu 507-8522, Japan, Tel:
+81-572-22-5311,

E-mail: shin1dinho@yahoo.co.jp

Abstract:

A 40-year-old woman underwent an atrial septal defect closure with
juxtaposition of the atrial appendages without communication between
both appendages 4 years before presentation. She recently experienced
desaturation on exercise, and right-to-left shunt coming from the
residual communication between both appendages was found. The residual
communication was closed from the right to the left atrium. Herein, we
report a rare case of juxtaposition of the atrial appendages with
residual communication between them after an atrial septal defect
closure.

Introduction:

Juxtaposition of the atrial appendages (JAA) is a rare congenital
cardiac malformation with both atrial appendages located on the same
side of the great arteries. Majority of reports described this disease
to be complicated with other malformations; however, communication
between the appendages is rarely reported, and thereby, its surgical
procedure is not yet established. We present the case of a 40-year-old
woman with left JAA associated with residual communication between them
after an atrial septal defect closure.

Case Report:

A 40-year-old woman presented with dyspnea and desaturation on exertion.
She had a history of atrial septal defect (ASD) patch closure 4 years
ago and chronic pulmonary artery hypertension {[}mean pulmonary artery
pressure (mPAP): 31 mmHg{]}, and JAA was found simultaneously. During
that time, no obvious communication was found between the appendages
before or after the operation. Enhanced computed tomography (eCT) was
performed for detailed examination on this admission and showed slight
communication between the appendages (Fig. 1). Additionally, right heart
catheter angiography showed a right-to-left shunt through the tunnel.
The catheter data showed mPAP of 30 mmHg, pulmonary vascular resistance
of 232 dyn s/cm\textsuperscript{5}, and Qp/Qs of 1.1. Cardiology team
decided to close this shunt due to a risk of paradoxical embolus.

Through a median resternotomy, cardiopulmonary bypass was established.
After right atriotomy, the tunnel ostium was observed in the right
atrium (Fig. 2A). Next, left atriotomy from the right side of the left
atrium was performed, and the ostium at the left atrium was found by
pouring saline from the ostium of the right atrium (Fig. 2B). Both ostia
were closed with polypropylene running suture. The subsequent course was
uneventful. Postoperative eCT showed no residual communication. Dyspnea
on effort gradually improved, and desaturation on exertion was resolved;
thus, further follow-up is required.

Discussion:

JAA is a rare congenital cardiac malformation named by Dixon in
1954.\textsuperscript{1} It is defined as the presence of both
appendages on the same side of the right or left great
arteries.\textsuperscript{1,2} Left JAA is more frequent than right JAA
and is sometimes classified as complete or partial according to its
location.\textsuperscript{3} The present case is of left JAA, and a
diminutive right auricle also exists at the right side of the heart.
Therefore, this is also a partial form of JAA. JAA is a known
complication of different congenital cardiac diseases with dextrocardia,
great artery transposition, tricuspid valve atresia or stenosis, right
ventricle hypoplasia, and ASD.\textsuperscript{4} The communication
between two appendages was a rare anomaly, shown in a few
reports.\textsuperscript{5} In this case, tunnel-like forms with
communication between two appendages existed just behind the atria,
which were not detected during ASD closure. Thus, in the current
operation, both sides of the ostium at the right and left atria were
closed. Injecting saline from the right atrial appendage is useful in
determining the left-sided ostium.

Detecting the communication between appendages using the conventional
transthoracic echocardiography or computed tomography (CT) is difficult
in ASD because the major shunt flow is derived from ASD. Late occurrence
of residual communication between the appendages with the juxtaposition
of atrial appendages after ASD closure is a rare phenomenon. Therefore,
the presence or absence of the communication should be confirmed with
enhanced CT or angiography when JAA occurs.

Conclusion:

Late communication between the appendages could occur in JAA after ASD
closure; hence, the possibility of communication between appendages in
patients with JAA should be considered, and closing it from both sides
is beneficial.

Author contributions:

S.I., M.M., T.F. and K.Y. designed and performed the experiments,
analyzed data and interpreted it. S.I. and K.Y. Drafted article. S.I.,
M.M., F.T. and K.Y. revised it critically. S.I., M.M., F.T. and K.Y.
approved of the article, collected data and supported technical and
logistical.

References:

\begin{enumerate}
\tightlist
\item
  Dixon AS. Juxtaposition of the atrial appendages: Two cases of an
  unusual congenital cardiac deformity. Br Heart J. 1954;16:153-64.
\item
  Lai WW, Ravishankar C, Gross RP, Kamenir SA, Lopez L, Nguyen KH et al.
  Juxtaposition of the atrial appendages: a clinical series of 22
  patients. Pediatr Cardiol. 2001;22:121-7.
\item
  Frescura C, Thiene G. Juxtaposition of the atrial appendages.
  Cardiovasc Pathol. 2012;21:169-79.
\item
  Anjos RT, Ho SY, Anderson RH. Surgical implications of juxtaposition
  of the atrial appendages. A review of forty-nine autopsied hearts. J
  Thorac Cardiovasc Surg. 1990;99:897-904.
\item
  Kara\selectlanguage{ngerman}çelik M, Karagöz U, Doyurgan O, Özdemir R, Öztürk P, Sariosmano\selectlanguage{polish}ğ\selectlanguage{english}lu
  ON. Right juxtaposition and a tunnel between the atrial appendages in
  a patient with atrial septal defect and pulmonary valve stenosis.
  World J Pediatr Congenit Heart Surg. 2015;6:105-7.
\end{enumerate}

Figure 1: Enhanced computed tomography showing a left juxtaposition of
the atrial appendages. (A) The right atrial appendage is not observed at
the rightful site and a little right auricle exists on the right side of
the heart (yellow arrow). Left atrial appendage is in normal position
(red arrow). The tunnel exists between the appendages (red asterisk).
(B) The sagittal plane shows that the hole of the actual communication
between appendages is 10 mm (yellow arrow). LAA, left atrial appendage

Figure 2: Intraoperative images obtained from the surgeon's perspective.
(A) The ostium of the tunnel (yellow arrow) was found in the right
atrium. (B) The ostium in the left atrium (blue arrow) was found during
the right lateral left atriotomy.\selectlanguage{english}
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\includegraphics[width=0.70\columnwidth]{figures/Figure-1/Figure-1}

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\includegraphics[width=0.70\columnwidth]{figures/Figure-2/Figure-2}

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