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\begin{document}

\title{Left ventricular pseudo-aneurysm complicating a ruptured isolated
congenital diverticulum}



\author[1]{Liang Fang}%
\author[2]{Chao Xue}%
\author[2]{Ying Zhao}%
\author[2]{Zhaoying Wen}%
\author[3]{Michael Henein}%
\affil[1]{Chengde Medical College Affiliated Hospital}%
\affil[2]{Capital Medical University Affiliated Anzhen Hospital}%
\affil[3]{Umea University}%


\vspace{-1em}



  
  \date{May 15, 2024}


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\selectlanguage{english}
\begin{abstract}
We report a case of 41-year-old woman who presented with chest tightness
and shortness of breath. Transthoracic echocardiogram (TTE) showed left
ventricular (LV) pseudo-aneurysm of the inferior wall with preserved LV
systolic function. Coronary angiogram was normal. Surgical repair of the
pseudo-aneurysm with a pericardial patch was performed, and pathological
results confirmed rupture of an isolated congenital LV diverticulum.as
the most likely etiology.%
\end{abstract}%



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\textbf{Introduction}

Congenital left ventricular (LV) diverticulum is a rare cardiac anomaly
which can be complicated with infective endocarditis, embolisms,
arrhythmia and, rarely, rupture\textsuperscript{1-4} . If ruptured, the
outcome is usually poor with high mortality. We report a case of LV
pseudo-aneurysm with ruptured diverticulum and successful surgical
repaired.

A 41-year-old woman presented with a history of chest tightness and
shortness of breath for 6 months. On clinical examination, blood
pressure was 124/86 mmHg. The ECG showed sinus rhythm without evidence
for ischemic changes, and chest X-ray revealed an enlarged heart. A
transthoracic echocardiography (TTE) showed 20 mm discontinuity (with
narrow connection neck) of basal segment of the inferior wall with 88 mm
\selectlanguage{ngerman}× 71 mm echo-free space, suggesting pseudo-aneurysm formation wrapped
within the pericardial (Fig 1A). There was a marked thrombus in the
pseudo-aneurysm with large pericardial effusion (Fig 1B). Color Doppler
demonstrated blood flow across the narrow neck of the pseudo-aneurysm
(Fig 1C). Overall LV systolic function was preserved with no other
combined abnormality.

The patient did not have any history of myocardial infarction, prolonged
fever, chest trauma or any cardiac surgery. Coronary angiogram confirmed
normal coronary arteries (Fig 2A \& 2B), and cardiac magnetic resonance
imaging (MRI) confirmed the presence of the LV pseudo-aneurysm, on late
gadolinium enhancement (LGE) showing LV pseudo-aneurysm with thin wall
corresponding to fibrosis and/or scar (Fig 3A \& 3B).

The patient underwent surgery, during which a large pseudo-aneurysm of
LV inferior wall was confirmed with a thrombus inside. The
pseudo-aneurysm was closely adherent to the pericardium of the
diaphragmatic surface (Fig 4). Surgical repair with a pericardial patch
was performed, and postoperative TTE showed normal LV function with the
patch located at the inferior wall. Pathological investigations showed
the aneurysm size was 70 × 60 × 35 mm. Based on the imaging,
pathological results and medical history we considered that the
ventricular aneurysm was a congenital LV diverticulum, which ruptured
and caused a ventricular pseudo-aneurysm.

\textbf{Discussion}

Congenital LV diverticulum is a rare cardiac anomaly with 0.42\%
prevalence in adults and may range from 5 to 90 mm, with normal LV
systolic function \textsuperscript{5-7} . It is thought to occur early
in embryogenesis, emerging from an outer pouching of the endomyocardium
through a weak LV segment. On cardiac imaging, LV diverticulum appears
like an outer sac arising from the LV wall, the ratio of the connecting
neck compared with the maximum diameter of the diverticulum varies,
always less than 1\textsuperscript{7}. LV diverticulum may be associated
with midline thoracoabdominal defects, other congenital cardiac
malformations, or can be isolated without intracardiac and extracardiac
anomalies\textsuperscript{8,9}. In isolated LV diverticulum, the patient
is usually asymptomatic or may have overlapping symptoms of heart
failure, chest pain, or dyspnea as seen in both aneurysms and
pseudoaneurysms. And complications like embolism, infective
endocarditis, arrhythmia and, rarely, rupture can be the initial
presentation \textsuperscript{2,3,5}.

Cardiac imaging is the most powerful tool for diagnosing LV diverticulum
and for differentiating it from aneurysms especially pseudo-aneurysms,
and delayed enhancement cardiac MRI is the most recently
recommended\textsuperscript{10-12}. LV diverticulum is usually
classified into two types, fibrous and muscular types. Muscular type
contains all three muscle layers with normal contractile function and is
not prone to rupture, and is easier to differentiate from true-aneurysms
which always accompany akinetic or dyskinetic wall complicating
myocardial infarction. The fibrous type diverticulum, with complete
absence of myocardium is usually located at the cardiac base or at the
subvalvular area, making it difficult to differentiate from
pseudo-aneurysms\textsuperscript{2,7,13}. The LV pseudo-aneurysm is
usually caused by cardiac rupture with discontinuity of the ventricular
wall, and is encircled by adherent pericardium or scar tissue, with no
myocardial tissue. The cause of pseudo-aneurysms includes acute
myocardial infarction (AMI), endocarditis, trauma, iatrogenic operation
(cardiac surgery, intervention), purulent pericarditis
\textsuperscript{14-17}. Over 50\% of cases are due to AMI, and the most
common location is the posterior and inferior wall.

Our patient denied any previous cardiac medical history, the coronary
arteries were normal without any evidence for atherosclerotic disease
and all other imaging investigations including; echocardiography, MRI,
left ventriculography and surgery confirmed the presence of the LV
pseudo-aneurysm. There were only few cardiomyocytes in the whole layer
of the ventricular wall on histology. Based on the history and imaging
findings, we believe that the most accurate diagnosis of this case is
the ruptured LV diverticulum causing pseudo-aneurysm which was
complicated with a large thrombus and pericardial effusion. Since the LV
systolic function was preserved and the patient was hemodynamically
stable, the surgical repair with patch was successful.

\textbf{References:}

1. Gowitt GT, Zaki SA. Rupture of a cardiac diverticulum resulting in
sudden death. \emph{Am J Forensic Med Pathol.} 1988;9(2):155-158.

2. Ohlow MA, von Korn H, Lauer B. Characteristics and outcome of
congenital left ventricular aneurysm and diverticulum: Analysis of 809
cases published since 1816. \emph{Int J Cardiol.} 2015;185:34-45.

3. Aquaro GD, Strata E, Di Bella G, et al. Prognostic role of isolated
left ventricular diverticuli detected by cardiovascular magnetic
resonance. \emph{J Cardiovasc Med (Hagerstown).} 2015;16(8):562-567.

4. Westaby S, Katsumata T, Runciman M, Burch M. Ruptured left
ventricular diverticulum in infancy. \emph{Ann Thorac
Surg.}1997;64(4):1181-1182.

5. Ohlow MA, Secknus MA, Geller JC, von Korn H, Lauer B. Prevalence and
outcome of congenital left ventricular aneurysms and diverticula in an
adult population. \emph{Cardiology.} 2009;112(4):287-293.

6. Paz Y, Fridman E, Shakalia FM, Danieli J, Mishaly D. Repair of an
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8. Ojha V, Chandrashekhara SH, Ganga KP, Saxena A, Gulati G. Congenital
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9. Li Q, Qu H, Wang H, Wang D, Li P, Liu T. Ventricular diverticulum: a
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10. Sharma A, Kumar S. Overview of left ventricular outpouchings on
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11. Srichai MB, Hecht EM, Kim DC, Jacobs JE. Ventricular diverticula on
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12. Cianciulli TF, Del Carmen Gonzalez Colaso P, Saccheri MC, et al.
Left ventricular diverticulum, a rare echocardiographic finding: two
adult patients and review of the literature. \emph{Cardiol
J.}2009;16(1):76-81.

13. Marijon E, Ou P, Fermont L, et al. Diagnosis and outcome in
congenital ventricular diverticulum and aneurysm. \emph{J Thorac
Cardiovasc Surg.} 2006;131(2):433-437.

14. Elshaer F, Altwijri F, Alrasheed F, Kharabsheh S, Habeeb W.
Congenital ventricular diverticulum and MI - Diagnostic challenges and
implications. \emph{J Saudi Heart Assoc.} 2015;27(4):277-282.

15. Meng X, Yang YK, Yang KQ, et al. Clinical characteristics and
outcomes of left ventricular pseudoaneurysm: A retrospective study in a
single-center of China. \emph{Medicine (Baltimore).} 2017;96(18):e6793.

16. Sunkara B, Briasoulis A, Afonso L, Ramappa P. Left ventricular
pseudoaneurysm as a fatal complication of purulent
pericarditis.\emph{Heart Lung.} 2015;44(5):448-450.

17. Frances C, Romero A, Grady D. Left ventricular
pseudoaneurysm.\emph{J Am Coll Cardiol.} 1998;32(3):557-561.\selectlanguage{english}
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