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\begin{document}

\title{An Unusual Presentation of an Intrapericardial Teratoma. A Case Report.}



\author[1]{Arega Leta}%
\author[2]{Anees Razzouk}%
\author[1]{Marx Genovez}%
\affil[1]{Loma Linda University Health}%
\affil[2]{Loma Linda University Medical Center}%


\vspace{-1em}



  
  \date{May 08, 2024}


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\selectlanguage{english}
\begin{abstract}
We're presenting a unique case where a 2 month old female presented with
URI symptoms at a local clinic and went into cardiac arrest after a
nasopharyngeal swab test was done. Subsequent work up revealed she had
an intrapericardial mass and a large pericardial effusion, which may
have precipitated the arrest. The patient recovered after emergent
pericardiocentesis and subsequent pericardial mass excision via
sternotomy and pericardiectomy. The infant was recovering appropriately
at her 4 month post-op follow up.%
\end{abstract}%



\sloppy


\textbf{Introduction:}

Intrapericardial teratomas come from three different germ
layers\textsuperscript{3}. They are very rare, (incidence of
0.05-0.15\%) but very aggressive\textsuperscript{3,4,6}. Their early
detection and interventions are very important for effective management
of the fetus. Most intrapericardial teratomas are attached to the aorta
and tend to compress the right side of the heart
structures\textsuperscript{1,5}. As a result, fetuses can present with
ascites, anasarca, and hydrops fetalis\textsuperscript{2,5,8}. Once
evidence of hydrops is present in a fetus, the survival rate decreases
when compared to a non-hydrops fetus (citation).

Therefore, early detection and good prenatal monitoring is essential.
Prenatal period detection of intrapericardial teratomas is useful as
interventions such as pericardiocentesis, or thoraco/pericardio-amniotic
shunt can be done\textsuperscript{2,5,8}. Tumor excision is the
definitive treatment as its rapid growth could lead to right atrium,
superior vena cava, and inferior vena cava
compression\textsuperscript{7}. This compression leads to ascites,
hydrops fetalis, and/or anasarca. In our unique case a 2 month-old
female did not present with the usual features of intrapericardial
teratomas such as hydrops fetalis, anasarca, or ascites, and instead
presented with cardiac arrest after an upper respiratory infection.

\textbf{Case:}

A 10 week old previously healthy female infant born via spontaneous
vaginal delivery (SVD) at full term gestation, who was doing well until
2 months at which time she developed signs and symptoms of URI followed
by cough but no fever. While in the medical office the patient went limp
after a nasopharyngeal swab test and subsequent cardiopulmonary
resuscitation (CPR) was done with return to spontaneous circulation
(ROSC) after 2 minutes.

She was transferred to a local Emergency Department where she was
intubated with a 3.5 uncuffed tube for concern of severe acidosis and
work of breathing. A bedside ECHO showed a large circumferential
pericardial effusion \emph{(Figure 1)} . The infant was transferred to
Loma Linda University Children's Hospital (LLUCH) for higher level of
care.

At LLUCH a pericardiocentesis was done and a pigtail was sutured into
position and placed on low intermittent suction. A CT Chest with
contrast showed a 4.2 x 3.6 x 3.8 cm right sided pericardial/mediastinal
hypodense mass with internal calcifications. The heart was shifted to
the left due to mass effect. The mass was partially surrounding the
aorta and SVC as well as the right pulmonary artery.

Echocardiogram showed normal biventricular systolic function with and EF
of approximately 67\% with no evidence of flow obstruction. A patent
foramen ovale with a small shunt was also found. It also showed a
multi-cystic mass appearing on top of the right atrium \emph{(Figure
2).}

The mediastinal teratoma was excised via sternotomy and pericardiectomy

\emph{(Figure 3).}

The tumor was found to be compressing the upper portion of the right
atrium. The tumor was removed without the use of cardiopulmonary bypass.
The teratoma seemed to have originated in the aorta because a 2 cm area
of aortic adventitia needed to be shaved off \emph{(Figure 4).}

Pathology report confirmed the mass to be a mature teratoma with tissue
types from intestinal and respiratory epithelium, smooth muscle,
skeletal muscle, cartilage, and glial tissue. There were no malignant or
immature elements seen. The tissue from the aorta showed similar mature
teratoma and the pericardium showed focal adhesions with no tumor
infiltration (Figure 5 \& 6).

The infant initially had problems feeding at her one and two month
follow up visits, but was doing well at her 4 month post-operative
follow up.

\textbf{Discussion:}

Our 10 week old female infant was a unique case as she was asymptomatic
until she was 9 weeks old. It is unclear as to what specifically caused
her to have cardiac arrest. We also suspect that when the baby limped
and cried with sudden increase of intrathoracic pressure the cystic
compoment of the mass might have ruptured to aggravate the pericardial
effusion and precipitated the tamponade, resulting in cardiac arrest. It
is equally possible that the large pericardial effusion found in bedside
echocardiogram may have caused electric conduction disruptions in her
heart leading to cardiac arrest. Review of the literature showed no case
reports detailing an otherwise healthy infant presenting with cardiac
arrest as a result of a cardiac mass. This case highlights the
importance of close prenatal screening and to have a high suspicion for
intrapericardial masses as a possible cause of cardiac arrest. Early
identification allows for interventions such as pericardiocentesis, or
thoraco/pericardio-amniotic shunt to be done in-utero allowing for
prolongation of pregnancy\textsuperscript{2,5,8}. On echocardiogram a
patent foramen ovale with a small shunt was present, which may have
prevented symptoms from presenting earlier.

\textbf{Conclusion:}

The case of our patient emphasizes the importance of routine meticulous
prenatal ECHO screening. This may help identify pathologies such as
intrapericardial masses early enough for an intervention. There is ample
evidence that early identification of these conditions allow for
effective interventions at the prenatal and post-natal
period\textsuperscript{2,5,8}. Our case highlights the importance for
early identification as it is likely that our patient had evidence of a
pericardial mass in-utero as most pericardial teratomas are present as
early as 21 weeks gestation\textsuperscript{5,8}. Furthermore, untreated
or undiagnosed intrapericardial masses could present with sudden cardiac
arrest as in our case, which could have a devastating outcome if not
treated immediately.

\textbf{Notes on patient consent:}

Subject's parents were contacted via phone and asked to provide consent
for usage of their child's case to be reported. Subject's parents
provided consent for usage of the subject's case.

\textbf{Works Cited Page:}

\begin{enumerate}
\tightlist
\item
  Arvind, A. Successful management of a neonate with antenatally
  detected mature intrapericardial teratoma. (2019)
\item
  Banfiled, F. Intrapericardial Teratoma: A new and treatable cause of
  hydrops fetalis. (12/1980)
\item
  Heerema-McKenney A, Harrison MR, Bratton B, Farrell J, Zaloudek C.
  Congenital teratoma: a clinicopathologic study of 22 fetal and
  neonatal tumors. Am J Surg Pathol 2005;29:29-38.
\item
  Laquay, N. Intrapericardial teratoma in newborn babies (12/2019)
\item
  Rychik, J. Fetal intrapericardial teratoma: natural history and
  management including successful in utero surgery. (12/2016)
\item
  Solomon N. Adherent Intrapericardial Teratoma: A Case Report
\item
  Wall J, Albanese CT. Pediatric Surgery. In: Doherty GM.
  eds.\emph{CURRENT Diagnosis \& Treatment: Surgery, 14e} New York, NY:
  McGraw-Hill; 2014.
  http://accessmedicine.mhmedical.com/content.aspx?bookid=1202\&sectionid=71529153.
  Accessed May 14, 2020.
\item
  Yuan, S. Fetal intrapericardial teratomas. (2/2019)
\end{enumerate}

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

\end{center}
\end{figure}

Figure 1. Large pericardial effusion evident in bedside ECHO.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image2/image2}

\end{center}
\end{figure}

Figure 2. A multi-cystic mass was seen compressing the right atrium
during ECHO evaluation.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image3/image3}

\end{center}
\end{figure}

Figure 3. Closure of pericardium during anterior pericardiectomy with
pericardial window to the left pleural space.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image4/image4}

\end{center}
\end{figure}

Figure 4. A 4.2 x 3.6 x 3.8 X 5 cm multicystic, combined with soft and
hard tissue mass adherent to the adventitia of the ascending aorta was
excised at the level of the aortic root up to the distal ascending aorta
along a 2 cm long segment.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image5/image5}

\end{center}
\end{figure}

Figure 5. Pathology resection showing external surface of showing a
mature teratoma with black ink on roughened margins.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image6/image6}

\end{center}
\end{figure}

Figure 6. Pathology resection showing cut surface of the tumor, showing
multiple cystic spaces.

\selectlanguage{english}
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\end{document}

