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\begin{document}

\title{Recurrent atrial septal defect following closure with CardioCel: A case
report}



\author[1]{Nischay Shah}%
\author[2]{Haytham Mously}%
\author[2]{Yakov Elgudin}%
\author[3]{Sadeer Al-Kindi}%
\affil[1]{University Hospitals of Cleveland}%
\affil[2]{University Hospitals Case Medical Center}%
\affil[3]{University Hospitals Harrington Heart \& Vascular Institute}%


\vspace{-1em}



  
  \date{May 22, 2024}


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\selectlanguage{english}
\begin{abstract}
An atrial septal defect is a commonly seen congenital heart defect of
which secundum atrial septal defect is the most common. In a patient
with evidence of right ventricular failure or significant shunt,
operative repair is the treatment of choice to achieve hemodynamic
stability. The use of a pericardial patch is required for the closure of
large atrial septal defects. Case presentation: Here we present a case
of a 50-year-old male who underwent surgical closure of secundum atrial
septal defect who subsequently found to have part of the pericardial
patch used for repair degraded in less than a month. Conclusion: There
is a paucity of long-term outcomes data following the use of the
CardioCel for septal defects, with no reports of such degradation within
a month to the best of our knowledge. Further study is required to
identify the incidence and implications of such findings.%
\end{abstract}%



\sloppy


Recurrent atrial septal defect following closure with CardioCel: A case
report

\textbf{Nischay Shah DO\textsuperscript{a} ; Haytham Mously MD,
MPH\textsuperscript{a}; Yakalov Elgudin MD, PhD\textsuperscript{b} ;
Sadeer Al-Kindi MD\textsuperscript{a};}

\textsuperscript{a}Harrington Heart and Vascular Institute and Case
Western Reserve University, Cleveland, Ohio

\textsuperscript{b}Department of Cardiothoracic Surgery, University
Hospitals Cleveland Medical Center, Ohio

Short title: Recurrent ASD with CardioCel

Correspondence

Nischay N. Shah DO,

Department of Internal Medicine,

University Hospitals Cleveland Medical Center,

11100 Euclid Ave, Cleveland,

OH 44106, USA.

Phone: 631-745-2345

Email: Nischay.Shah@UHhospitals.org

The authors have no sources of funding to disclose.

\textbf{Abstract}

An atrial septal defect is a commonly seen congenital heart defect of
which secundum atrial septal defect is the most common. In a patient
with evidence of right ventricular failure or significant shunt,
operative repair is the treatment of choice to achieve hemodynamic
stability. The use of a pericardial patch is required for the closure of
large atrial septal defects. Case presentation: Here we present a case
of a 50-year-old male who underwent surgical closure of secundum atrial
septal defect who subsequently found to have part of the pericardial
patch used for repair degraded in less than a month. Conclusion: There
is a paucity of long-term outcomes data following the use of the
CardioCel for septal defects, with no reports of such degradation within
a month to the best of our knowledge. Further study is required to
identify the incidence and implications of such findings.

\textbf{Background}

In adult congenital heart defects, atrial septal defect (ASD) accounts
for more than one-fourth of all cases, out of which secundum ASD
accounts for almost 75\% of cases. Often Secundum ASD is diagnosed when
a patient is an adult as its detection is difficult due to delayed
symptomatic presentation. Currently, there is a class IIa recommendation
that any asymptomatic patient with an isolated Secundum ASD with
evidence of right atrium (RA) enlargement, right ventricle (RV)
enlargement, and net left-to-right shunt with physiological sequelae but
without cyanosis is a candidate for transcatheter or surgical closure.
However, when ASD closure is difficult as indicated by morphological
features of large size([?]30 mm), wide rim([?]15 mm) deficiency, and
multiple defects surgical closure is preferred compared to transcatheter
closure. The surgical approach to Secundum ASD repair is a safe and
effective operation with little to no morbidity and mortality.

Many congenital heart defects including ASD require a pericardium patch
repair. Recently, CardioCel\selectlanguage{ngerman}® a tissue engineered ADAPT\selectlanguage{english} bovine
pericardial patch known to have a good outcome in terms of preventing
calcification and inflammation reactions has been commonly used in
cardiovascular surgery since 2014. Here we present a novel case of a
patient who underwent Secundum ASD repair with CardioCel patch that
failed within a with presumed degeneration of the patch.

\textbf{Case Presentation}

A 50-year-old male with no past medical history presented with shortness
of breath in an outpatient clinic. He underwent transthoracic
echocardiography (TTE) and was found to have left ventricle ejection
fraction (LVEF) of 65\%, moderate dilation of the RA, moderate to severe
dilation of the RV, and possible ASD with positive bubble study.
Subsequently, a month later he underwent left heart catheterization
(LHC) and right heart catheterization (RHC) and was found to have
nonobstructive coronary artery disease and significant left-to-right
shunt at the atrial level. Immediately after the procedure, he underwent
a transesophageal echocardiogram (TEE) which showed LVEF 60\%, Secundum
ASD measuring 36x24 millimeters in diameter with deficient retroaortic
rim, and moderate enlargement of RA and RV. A month later patient was
found to be an atrial flutter. A repeat TTE was performed before
cardioversion and showed a large secundum ASD 35x25 millimeter with a
left to right shunting.

In January 2021, the patient underwent evaluation for potential
percutaneous closure with the device but the anatomy of ASD was such
that there was no superior rim to land the device, and the patient was
thought to be not a candidate for percutaneous closure. Afterwards, the
patient was offered options for ASD closure through right thoracotomy as
well as median sternotomy and he chose median sternotomy approach. In
February of 2021, the patient underwent ASD repair. Intraoperatively,
the interatrial septum was absent. There were no clefts in the mitral
leaflet or tricuspid septal defect, but ASD extended to the aortic root.
The large ASD was repaired with a CardioCel patch fixed in place with a
running Prolene suture. Post repair TEE showed no residual shunt with
saline contrast.

A month later in March 2021, the patient presented to the emergency
department with heart palpitation. The patient was found to be in atrial
flutter with a rapid ventricular response with HR of 135 and hypotension
with blood pressure 70/40 mmHg. The patient was stabilized but remained
in atrial flutter. TEE was done before cardioversion which showed LVEF
55\%, Secundum ASD(see figure 1) measuring 12 x 14 millimeters with
evidence of residual bidirectional shunting (figure 2, 3 and 4). It was
thought that patient had degeneration of surgical bioprosthetic ASD
repair patch. The patient was discharged with close outpatient follow-up
and a plan to reevaluate in six months for a possibility of
transcatheter closure of a residual ASD.

\textbf{Discussion and Conclusion}

Both the transcatheter or surgical approach are reasonable for Secundum
ASD repair but surgical closure is preferred if defect size is large,
there is significant proximity to adjacent structures, history of failed
percutaneous closure, inadequate atria to device size, or an associated
pathology requiring cardiac surgery. Surgically there are many
approaches including sternotomy, thoracotomy, video-assisted-assisted
thoracoscopic surgery, and robotics. Median sternotomy is thought to be
the gold standard approach and, in this procedure, the sternum is
usually divided in its entirety and cannulation is via the ascending
aorta and both cava. After the surgery, complications are seldom but
arrhythmia, cardiac tamponade, or an infection can develop.

To our knowledge, we describe the first case in the published literature
of secundum ASD closure with the bovine patch that degraded within 2
months with the redevelopment of the bidirectional shunt. In the
literature, data on bioprosthetic patch degeneration are scarce;
however, degradation or failure of the aortic bioprosthetic valve is
well established. One of the most common causes of bioprosthetic valve
degeneration is calcification. Other potential culprits include suture
dehiscence, collagen fiber disruption, tissue disruption due to
mechanical stress and shear forces, and inflammatory or immune response.
Given the acute failure of the patch in our case, we suspect either
dehiscence or host immune response caused degeneration of the ASD repair
patch.

Restoration of cardiac septation in the vast majority of cases requires
a patch material for reconstruction.\textsuperscript{1} CardioCel patch
is one such bovine scaffold of great interest due to greater mechanical
stability and decreased cost. For instance, in one comparative study,
all the bioscaffolds commonly used in cardiac surgery were compared
including bovine pericardial scaffolds, cross-linked with 0.6\%
glutaraldehyde such as XenoLogiX\selectlanguage{english}, PeriGuard(r), dye-mediated
photo-oxidized PhotoFix\selectlanguage{english} and a non-crosslinked porcine scaffold
CorMatrix(r), and CardioCel(r) (decellularized, cross-linked with 0.05\%
monomeric glutaraldehyde, detoxified). CardioCel demonstrated greater
cross-linked stability and comparable tensile strength at 12 weeks
follow-up.

Previous data on CardioCel patch use in adults are sparse. In one study
by Tomisic et al., they looked at 30 patients with a median age of 57
years who underwent mitral valve leaflet repair with a CardioCel patch.
At a mean follow-up of 1.7 years, there were 3 deaths and 2 of them had
infective endocarditis as a complication. In the pediatric population,
the Cardiocel patch has been well studied. A study by Bell et al looked
at 501 CardioCel implanted patches with a median follow up of 31 months
which showed that a total of 14 implants require intervention and
freedom from reintervention was close to 96\% at 3 and 5 years. Multiple
other studies looked at Cardiocel use in congenital heart disease with
short term to long term follow-ups all found positive results with
Cardiocel and no cases on patch degradation or reabsorption ever noted.

Studies on the CardioCel patch have not shown any cases of early
degeneration. We can infer the etiology of degeneration from studies on
bioprosthetic aortic valve degeneration. Currently known common culprits
in bioprosthetic material degeneration are calcification, dehiscence,
microstructure alteration, and host inflammatory or immune response. The
dearth of published cases of degradation of patch closure may be
indicative of the rarity of this complication and its need for further
study.

List of abbreviations

ASD = atrial septal defect

RA = right atrium

RV -- right ventricle

TTE = transthoracic echocardiography

LVEF = left ventricle ejection fraction

LHC = left heart catheterization

RHC = right heart catheterization

TEE = transesopahgeal echocardiography

CONFLICT OF INTERESTS

The authors declare that there is no conflict of interest.

ETHICS STATEMENT

All research meets the ethical guidelines, including adherence to the
legal requirements of the United States.

References\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

\end{center}
\end{figure}

Figure 1: 3D image showing ASD secundum\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image2/image2}

\end{center}
\end{figure}

Figure 2: Subcostal view with agitated saline showing left-to-right
shunt\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image3/image3}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image4/image4}

\end{center}
\end{figure}

Figure 3: Four chamber view of TEE left-to-right shunt across the
secundum ASD\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image5/image5}

\end{center}
\end{figure}\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image6/image6}

\end{center}
\end{figure}

Figure 4: Color flow doppler in TEE images showing left-to-right shunt

\selectlanguage{english}
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