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\begin{document}

\title{GIANT PLACENTAL CHORIOANGIOMA WITH AN UNEVENTFUL CLINICAL COURSE}



\author[1]{F. Limaiem}%
\author[2]{Anissa Ben amor}%
\author[3]{Khalil Saffar}%
\author[2]{Ahmed Halouani}%
\author[1]{saadia bouraoui}%
\affil[1]{Centre Hospitalier Universitaire Mongi Slim}%
\affil[2]{University Hospital Center Mongi Slim}%
\affil[3]{University of Tunis El Manar}%


\vspace{-1em}



  
  \date{May 19, 2024}


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\selectlanguage{english}
\begin{abstract}
A 33-year-old primiparous woman presented with reduced fetal movements
during the gestational age of 33 weeks. Ultrasonography showed a
heterogeneous and hypoechoic placental lesion measuring 8 cm. A week
later, the patient delivered a live healthy male neonate. Histological
examination of the placental mass established the diagnosis of placental
chorioangioma.%
\end{abstract}%



\sloppy


\textbf{INTRODUCTION}

Initially described by Clarke in 1798, placental chorioangiomas are
benign non-trophoblastic, vascular neoplasms of the placenta {[}1{]}.
Their true incidence is unknown since the majority of them are small,
clinically asymptomatic and may be missed during obstetric screening
procedures. Large chorioangiomas (\textgreater{} 5 cm) are rarer and are
often associated with an increased risk of pregnancy complications
{[}2{]}. Herein, we report a peculiar case of a giant placental
chorioangioma without significant feto-maternal complications except for
prematurity.

\textbf{CLINICAL HISTORY}

A 33-year-old primigravida woman with a past medical history of
thyroidectomy, presented to the outpatient gynecology clinic during the
gestational age of 33 weeks, complaining of reduced fetal movements.
Physical examination revealed a uterine fundal height of 29 cm
appropriate for the gestational age. Ultrasonography showed a single
live fetus of 32-33 weeks in breech presentation and an anterior fundal
placenta. A well-defined heterogeneous and hypoechoic space-occupying
lesion measuring 8.3 x 4.9 cm noted in the placenta bulging into
amniotic cavity at the site of umbilical cord insertion was interpreted
as consistent with a chorioangioma (Figure 1A \& 1B). Neither fetal
cardiomegaly nor early signs of hydrops fetalis were noted. Non stress
tests revealed no sign of fetal distress. Spontaneous labor commenced at
34 weeks of gestation and she delivered a live male baby with a breech
presentation via a cesarean delivery with an Apgar score of 9 in the
first minute and 10 in the fifth minute. The newborn weighed 2.7 kg and
did not show any signs of anemia (Hb : 19 g/dl) or distress. The
placenta was sent to the pathology department for histological
examination. Grossly, the delivered placenta measured 16\selectlanguage{ngerman}×15×3 cm and
weighed 835 g. There was a well-defined lobular mass on the fetal
surface measuring 8×4.9 cm and located near the insertion of the
umbilical cord (Figure 1 C). The length of the umbilical cord was 40 cm.
On serial sections, the cut surface of the mass was solid whitish with
few reddish and micro cystic areas (Figure 1 D). The rest of the
placenta, membranes, and umbilical cord were unremarkable. The placenta
was intact and complete, and the outer surface of the mass was congested
with prominent vascular markings. Microscopic examination of the
placental mass showed a well-demarcated lesion composed of numerous
capillary sized vascular channels set in a fibroid matrix. Sections from
the umbilical cord were unremarkable. The histology results, therefore,
confirmed the diagnosis of a chorioangioma of capillary type (Figures
2A, 2B, 2C). Immunohistochemical study showed strong positive
immunostaining of endothelial cells for CD 34 (Figure 2D). The infant
did not have anemia and hypoproteinemia and there was no evidence of
hydrops fetalis at birth. The mother and her baby were discharged on the
fifth day post-delivery after the newborn was found to be normal on
subsequent examinations.

\textbf{DISCUSSION}

Also called `' chorangioma placentae'', placental chorioangioma is the
most frequent non-trophoblastic tumor of the placenta which originates
from the placental chorionic mesenchyme {[}3{]}. Placental chorioangioma
accounts for approximately 1\% of pregnancies and is predominant in
Caucasian women {[}4{]}. The majority of researchers consider
chorioangioma to be a malformation rather than a true neoplasm {[}4{]}.
Some environmental factors, namely hypoxic stimulus, high altitude, and
vascular growth factors, are possible associations with this tumor's
occurrence {[}5{]}. Large chorioangiomas can induce several
complications in pregnancy such as polyhydramnios, premature labor,
placenta previa, and premature placental separation, leading to high
perinatal morbidity and mortality {[}6,7{]}. Other complications of
large chorioangiomas include placental functional inadequacy, mechanical
obstruction of the blood flow, and increased vascular surface area. This
induces increased transudation of fluid, resulting in polyhydramnios
{[}8{]}. The pathological complications seen in the neonates include
thrombocytopenia, anemia, stillbirth, edema, non-immune hydrops fetalis,
prematurity, intrauterine growth retardation, or fetal death.
Arteriovenous shunts in large chorioangiomas cause impairment of the
fetal circulation. {[}9,10{]}. In the present case, the pregnancy of
this 33-year-old primiparous woman with a large placental chorioangioma
had prematurity as the only adverse effect. Prematurity was probably the
result of the large volume of the placental chorangioma occupying a
significant amount of intrauterine volume and therefore leading to the
induction of early uterine contractions that culminated in the premature
delivery. On ultrasonography, chorioangiomas are well-circumscribed
hypoechoic masses that often protrude into the amniotic cavity from the
fetal surface near the cord insertion. Calcification and necrosis are
occasionally visible {[}10{]}.

n sonography, the lesion

is well circumscribed and has echogenicity dif-

ferent from that of the rest of the placental tissue.

Large tumors can be of variable shapes, divided

by fibrous septa, and often protrude into the

amniotic cavity from the fetal surface near the

cord insertion

n sonography, the lesion

is well circumscribed and has echogenicity dif-

ferent from that of the rest of the placental tissue.

Large tumors can be of variable shapes, divided

by fibrous septa, and often protrude into the

amniotic cavity from the fetal surface near the

cord insertion

n sonography, the lesion

is well circumscribed and has echogenicity dif-

ferent from that of the rest of the placental tissue.

Large tumors can be of variable shapes, divided

by fibrous septa, and often protrude into the

amniotic cavity from the fetal surface near the

cord insertion

n sonography, the lesion

is well circumscribed and has echogenicity dif-

ferent from that of the rest of the placental tissue.

Large tumors can be of variable shapes, divided

by fibrous septa, and often protrude into the

amniotic cavity from the fetal surface near the

cord insertion In the present case, we used Doppler ultrasound to
monitor fetal wellbeing and circulation; however, despite the presence
of the large chorioangioma, there were no signs of anemia or cardiac
failure. Macroscopically, chorioangiomas are well-circumscribed tumors.
On cut section, they are usually fleshy, congested, and red to tan. We
occasionally note the presence of myxoid, fibroma-like or degenerative
changes. Placental chorioangiomas are often found on the fetal surface
of the placenta with a few cases reported on the maternal surface as
well {[}6{]}.

The majority of placental chorioangiomas are small measuring less than 5
cm in diameter and are located entirely in the placental parenchyma;
that is why we can be easily miss them if the placenta is not
systematically sliced after delivery {[}11{]}. In the present case, the
tumor measured 8.3 x 4.9 cm and was located on the fetal surface near
the umbilical cord insertion with focal cystic change. The definitive
diagnosis of placental chorioangioma relies on histopathological
examination. Histologically, chorioangioma is a benign tumor
proliferation of vessels accompanying connective tissue and an abundant
stromal component. The proportion of vascular and stromal components are
variable, depending upon the dominant histologic type. The vascular
component may comprise either capillaries or sinusoids. Based on
histology, the tumor can be classified as capillary, cavernous,
endotheliomatous, fibrosing, and fibromatous. Such differentiation is
not of clinical concern, since the prognosis depends mainly upon the
size of the tumor. In the current case, the tumor was of the capillary
type {[}10-11{]}. There is a rare variant of chorioangioma: ``atypical
cellular chorioangioma'' characterized by high mitotic activity,
cellularity and abnormal nuclei with proliferating malignant trophoblast
interspersed among proliferating capillaries with large areas of
necrosis {[}12{]}. The immunohistochemical study shows a positive
immunostaining of tumor cells for CD31, CD 34, factor VIII, GLUT 1, and
cytokeratin 18. The differential diagnosis of placental chorioangioma
includes chorangiosis and chorangiomatosis. Prognostic factors that
influence the outcome of placental chorioangioma are vascularity and
location of the tumor. Increased vascularity leads to greater shunting
of blood to the physiologic dead space of the tumor, weaning the fetus
of a significant amount of nutrient and air exchange and perhaps
manifesting as intrauterine growth restriction and fetal demise.
Likewise, location is also critical in deciding the pregnancy outcome,
as, in certain reported cases, chorioangiomas as small as 2 cm located
at the outer edge of the placenta have been associated with
complications {[}8{]}.

Management of chorioangioma depends on its degree of severity. For mild
to moderate types, the treatment is conservative as it was the case for
our patient. For severe cases, there are several management modalities
including in-utero endoscopic devascularization and prenatal treatment
by ultrasound guided interstitial laser therapy. In case of fetal
anemia, in-utero blood transfusion can be done with good outcome. In
case of polyhydramnios, maternal indomethacin therapy or amniocentesis
is useful in management. If complications appear late in pregnancy
delivery is the option of management. {[}13{]}.

In summary, placental chorioangioma is associated with an increased risk
of complications influencing the outcome of pregnancy. Despite the
existence of uncomplicated cases, close monitoring and follow up of the
patients by serial ultrasound, surveillance by Doppler, and fetal
echocardiography is highly recommended for these patients. Careful
examination of the placenta, timely antenatal diagnosis, and early
intervention are of paramount importance, to prevent associated
morbidity and mortality to both the fetus and the mother. In the current
case, except for prematurity, there were no clinically significant
complications due to the giant placental chorioangioma measuring 8 cm in
diameter.

\textbf{CONFLICT OF INTEREST STATEMENT}

None declared.

\textbf{AUTHORS' CONTRIBUTIONS:}

\textbf{Dr} \textbf{Faten LIMAIEM} prepared, organized, wrote, and
edited all aspects of the manuscript. She performed the gross and
microscopic pathologic evaluation of the pathology specimen. She
prepared all of the histology figures in the manuscript. She read,
edited, and approved the final version of the manuscript.

All authors should have made substantial contributions to the
manuscript.

\textbf{Dr Khalil SAFFAR} and \textbf{ANISSA BEN AMOR} participated in

The conception and design of the study,

The acquisition of data, analysis and interpretation of the data,

\textbf{Pr Saadia Bouraoui and Pr Amel Triki} participated in

The drafting of the article and revising it critically for important
intellectual content,

All authors participated in the

Final approval of the manuscript before its submission

\textbf{ETHICAL APPROVAL}

All procedures performed were in accordance with the ethical standards.
The examination was made in accordance with the approved principles.

\textbf{ACKNOWLEDGEMENT STATEMENT}

I would like to offer my special thanks to~Mrs~Saïda~Jerbi~who prepared
the microscopic slides. Published with written consent of the patient.

\textbf{DATA AVAILABILITY}

In accordance with the DFG Guidelines on the Handling of Research Data,
we will make all data available upon request.

\textbf{REFERENCES}

{[}1{]} Dao AH, Rogers W, Wong SW. Chorangiomas of the placenta: Report
of two cases with Ultrasound study in one. Obstet Gynecol.
1981;57(6):46-9.

{[}2{]} Saxena A, Faujdar M, Kaur A, Gupta S. Large Placental
Chorangioma with an Unusual Uneventful Course. Journal of Gynecologic
Surgery. 2017; 33 (1): 30-33.

{[}3{]}. Kirkpatrick A, Podberesky DJ, Gray AE, McDermott JH. Placental
chorangioma. Radiographics. 2007;27:1187.

{[}4{]}. Esen UI, Orife SU, Pollard K. Placental chorangioma: A case
report and literature review. Br J Clin Pract. 1997; 51:181.

{[}5{]}. Guschmann M, Henrich W, Entezami M, Dudenhausen JW.
Chorioangioma---new insights into a well-known problem: I. Results of a
clinical and morphological study of 136 cases. J Perinat Med.
2003;31:163.

{[}6{]}. Khanam R, Ghosh PK, Jahan F, Rahman S. Chorangioma. J Med Case
Reports. 2013;14:185-6.

{[}7{]}. Nanda SS, Dash S, Huria A. Placental chorioangioma and preterm
labour: A case Report. Sch J Appl Med Sci. 2013;15:400-2.

{[}8{]}. Kataria N, Singh A, Bedi PK. Giant Placental Chorangioma: A
Rare Case Report. J Clin Diagn Res. 2016;10:ED 03-4.

{[}9{]}. Sabhikhi AK, Chaudhary MC, Singh D, Raja LN. Chorangioma of the
placenta with Hydrops Fetalis. Indian Paediatr. 1996;33:520-1.

{[}10{]}. Zalel Y, Weisz B, Gamzu R, Schiff E, Shalmon B, Achiron R.
Chorioangiomas of the placenta: Sonographic and Doppler flow
characteristics. J Ultrasound Med. 2002;21:909-13.

{[}11{]} Duro EA, Moussou I. Placental chorioangioma as the cause of
non-immunologic hydrops fetalis: A case report. Iran J Pediatr.
2011;21:113.

{[}12{]}. Majlessi HF, Wagner KM, Brooks JJ. Atypical cellular
chorangioma of the placenta. Int J Gynecol Pathol. 1983;1:403.

{[}13{]}. Das R, Subedi N, Gurung G. Chorioangioma of Placenta. J Nepal
Med Assoc. 2017; 56: 472-4.

\textbf{FIGURE LEGENDS:}

\emph{Multipanel figure 1}\textbf{:}

\textbf{Figure 1 A :} Ultrasonography showing a well-defined
heterogeneous and hypoechoic space-occupying placental lesion measuring
8.3 x 4.9 cm and bulging into the amniotic cavity at the site of
umbilical cord insertion. \textbf{Figure 1 B:} Color Doppler imaging
showing a heterogeneous echogenic large lobular mass measuring
approximately 8x4.9 cm and increased blood flow within the
tumor.\textbf{Figures 1 C + 1 D:} Macroscopic view of the placental
chorioangioma. On cut section, there is a well-defined lobular mass on
the fetal surface measuring 8 ×4.9 cm and located near the insertion of
the umbilical cord. On serial sections, the cut surface of the mass was
solid whitish with few reddish and micro cystic areas. The rest of the
placenta, membranes, and umbilical cord were unremarkable.

\emph{Multipanel figure 2}\textbf{:}

\textbf{Figure 2 A} Microscopic examination of the placental mass
showing a well-demarcated lesion composed of numerous capillary-sized
vascular channels within a fibrous matrix. \textbf{(} Hematoxylin and
eosin, magnification × 40\textbf{), Figure 2 B:} Variable sized
capillaries within a fibrous matrix \textbf{(} Hematoxylin and eosin,
magnification × 100\textbf{).} \textbf{Figure 2 C:} Capillaries are
lined by endothelial cells devoid of atypia (Hematoxylin and eosin,
magnification × 400).

\textbf{Figure 2 D:} Immunohistochemical study showing strong positive
immunostaining of the endothelial cells with the CD34 antibody,
(Immunohistochemistry, magnification × 400).\selectlanguage{english}
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\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/Multipanel-Figure-2-JPEG/Multipanel-Figure-2-JPEG}

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