ANALYTICAL AND QUANTITATIVE CYTOPATHOLOGY AND HISTOPATHOLOGY

1. 34
Analytical and Quantitative Cytopathology and Histopathology®
0884-6812/21/4301-0034/$18.00/0 © Science Printers and Publishers, Inc.
Analytical and Quantitative Cytopathology and Histopathology®
BACKGROUND: Hepatic hydatid disease is a parasit­
ic zoonosis caused by Echinococcus granulosus. The
liver is the most frequently parasitized organ in humans.
E. granulosus typically forms a small, fibrous, edged
cyst when there is any surrounding host reaction. Clas­
sically, there is a large parental cyst with a large number
of peripheral daughter cysts. Satellite daughter cysts are
common. E. granulosus has two forms: pastoral and
sylvatic.
CASE: A 36-year-old woman was hospitalized upon
complaint of nonspecific, continuous, moderate-to-severe
epigastric pain of 1 week’s duration. There was no fever
or vomiting. Only serum aspartate transaminase (420
U/L), alanine aminotransferase (180 U/L) (normal up
to 50 U/L), and erythrocyte sedimentation rate (65 mm/
hour) were increased in her laboratory findings. She had
a hydatid cyst in the right lobe of the liver and refused
all treatment protocols. Her magnetic resonance imaging
and magnetic resonance cholangiopancreatography data
showed a ruptured liver hydatid cyst associated with
closed perforation in the antrum region of the stomach.
CONCLUSION: Typically, locations of hepatic hydatid
cyst can be seen into the biliary tree, peritoneal space,
and blood stream, but extension outside of the liver is
rarely seen in the literature. (Anal Quant Cytopathol
Histpathol 2021;43:34–39)
Keywords: Echinococcus granulosus, hepatic hy­
datid cyst, liver, gastrointestinal organs, parasitic
zoonosis, perforating cyst, trauma.
Human cyst hydatid disease usually occurs with
invasion of Echinococcus granulosus and, less fre­
quently, Echinococcus multilocularis.1 Although re-
ported in various countries, the disease is particu-
larly endemic for the Mediterranean region, Africa,
South America, the Middle East, Australia, and
New Zealand.2-6 Hydatid illness has been known
since the time of Hippocrates, who described it
as “a liver full of water.” The anatomy of hydatid
cyst structure is well known; however, findings of
hydatid disease complications and unusual anato-
mic locations have been less frequently described.
The hydatid cyst has 3 layers: an external pericyst
consisting of modified host cells forming an inten-
sive and fibrous protective area, a middle lami-
nar membrane which is cellular and permits the
passage of nutrients, and an inner germinal layer
Spontaneous Rupture of a Hepatic Hydatid Cyst
Perforating into the Gastric Antrum Diagnosed
with Magnetic Resonance Imaging
A Case Report and Review of the Literature
Özgür Ertuğrul, M.D., Mehmet Cudi Tuncer, Ph.D., and Ercan Gedik, M.D.
From the Departments of Anatomy and of General Surgery, Faculty of Medicine, University of Dicle, Diyarbakır; and the Department of
Radiology, Memorial Hospital, Diyarbakır, Turkey.
Özgür Ertuğrul is Physician, Department of Radiology, Memorial Hospital (ORCID ID: 0000-0002-7178-2164).
Mehmet Cudi Tuncer is Professor, Department of Anatomy, Faculty of Medicine, University of Dicle (ORCID ID: 0000-0001-7317-5467).
Ercan Gedik is Professor, Department of General Surgery, Faculty of Medicine, University of Dicle (ORCID ID: 0000-0002-5812-6998).
Address correspondence to: Mehmet Cudi Tuncer, Ph.D., Department of Anatomy, Faculty of Medicine, University of Dicle, 21280
Diyarbakır, Turkey (drcudi@hotmail.com).
Financial Disclosure: The authors have no connection to any companies or products mentioned in this article.

2. Volume 43, Number 1/February 2021 35
Hepatic Hydatid Cyst Perforating into the Gastric Antrum
where the scolices (parasitic larval stage) and the
laminar membrane are produced. The middle lami-
nated membrane and the germinal layer form the
true wall of the cyst, usually referred to as the
endocyst, while the acellular laminated membrane
is occasionally referred to as the ectocyst.2,3,7 Cys-
tic fluid is formed of sodium chloride, proteins,
glucose, ions, lipids, and polysaccharides. The anti-
genic fluid may also contain scolices and hooklets.
When the vesicles are broken off in the cyst, scolices
come in the cyst fluid and form a hydatid cyst.3
The clinical course of the disease varies from
an asymptomatic liver lesion to an invasive mass.
Invasion of biliary and vascular structures of the
liver may result in biliary obstruction, portal hy­
pertension, esophageal variceal bleeding, or Budd-
Chiari syndrome. In humans 50–75% of hydatid
cysts occur in the liver, 15% are seen in the lungs,
and 5–10% are distributed throughout the arterial
circulation.8 The right lobe of the liver is the most
common location of the liver. Visual findings in
hepatic hydatid illness are due to the evolution of
cyst growth. The lungs are the second most com-
mon location of hematogenous spread in adults
and are almost certainly the most common loca-
tion in children (15–25% of cases).4,9 Collapsible
organs such as the lung or brain facilitate cyst
growth and are suggested as a reason for the high
prevalence of childhood illness. Extension of a
hepatic hydatid cyst is rarely seen outside of the
liver.10-16 Complications in hepatic hydatid cysts
count in cyst rupture and secondary bacterial infec-
tion.8 The cyst may be split up spontaneously
after a injury or as a result of increased intracystic
pressure. Superficial cysts, large cysts, and high-
pressure live cysts tend to enter the body cavities,
especially the pleural and peritoneal cavity, or are
discharged into the bile or gastrointestinal tract.
The primary diagnostic processes are ultrasonog-
raphy (US), computed tomography (CT), and mag-
netic resonance imaging (MRI). The findings of
dramatic and acute abdominal symptoms such as
reflection and sensitivity are usually present. This
complication should take place especially in the
differential diagnosis of acute abdominal involve-
ment in endemic regions. In patients with peri-
toneal penetration, specific management has not
been adequately assessed and no clear methods
are present. The primary treatment process for
uncomplicated cases also applies to complicated
cases such as peritoneal perforation. Hydatid cyst
rupture requires urgent surgical intervention.17,29
We describe a rare case related to a ruptured hepat-
ic hydatid cyst perforating into the gastric antrum,
diagnosed with the help of MRI.
Case Report
A 36-year-old woman living in the southeast of
Turkey presented to our outpatient internal medi-
cine clinic complaining of a continuous moderate-
to-severe epigastric pain of 1 week’s duration. She
had been recently diagnosed with right hepatic
lobe, which included a hydatid cyst, but she re-
fused all the treatment options. On physical exam-
ination diffuse tenderness and pain were noted at
the epigastric and right upper quadrant regions
of the abdomen. The patient had no fever or vom-
iting. Laboratory findings revealed an increase in
serum aspartate transaminase (420 U/L) and ala-
nine aminotransferase (180 U/L) (normal up to 50
U/L) levels and elevated erythrocyte sedimenta-
tion rate (65 mm/hour). All the other laboratory
examinations were normal. MRI and magnetic reso-
nance cholangiopancreatography (MRCP) showed
a thick-walled cyst with a detached laminated
membrane in the liver, perigastric fluid, gastric
antrum wall thickening, and capsular perforation
in the posterior wall of the liver. According to the
2001 World Health Organization classification of
liver hydatid cysts, this case was type 2 (Table I).
This type includes multiple septated cysts which
give a multivesicular, rosette, or honey-packed
appearance in a unilocular maternal cyst. This stage
is the active phase of the cyst. These findings were
confirmed by our results in MRI (Figure 1). Because
cyst rupture is direct in this case, the cyst perforated
both the liver and the antrum region of the stomach
(Figures 2–3). Through the gastrohepatic ligament,
it is seen that the cyst progresses downward and
extends into the stomach (Figure 4). In radiologi-
cal findings, axial T2-weighted MRI and MRCP
revealed free air at the posterior gastric pyloric
wall, gastric antral luminal high-density content,
and enlargement (Figures 2–3). MRCP revealed a
detached laminated membrane in the hydatid cyst
of the liver, free perigastric fluid, and lobulated
thick-walled cysts at the antral luminal mucosa
(Figure 3). The case was interpreted as a ruptured
hydatid cyst of the liver associated with closed per-
foration in the antrum region of the stomach.
Discussion
Intrahepatic hydatid cyst rupture can occur spon-
taneously or following a trauma. Increased risk of
C
ASE
R
EPORT

3. 36 Analytical and Quantitative Cytopathology and Histopathology®
Ertuğrul et al
rupture has been reported with increased size of
the cyst and intracystic pressure.10,15,18 Although
rupture is associated with minor injury, the natu-
ral history of hepatic hydatid cysts suggests rup-
ture as a complication in 50–90% of cases.19,20 The
cyst content into the host’s bloodstream may pro-
duce anaphylaxis owing to the antigenic nature of
the cystic fluid.19,21 Patients with ruptured hyda­
tid cyst of the liver may rarely be asymptomatic.
Echinococcal cysts of the liver can cause compli­
cations in about 40% of cases. The clinical signs
and symptoms of hydatid cyst rupture are not
always severe, but, in free perforation, hydatid
fluid can cause chemical peritonitis. In addition,
peritoneal manifestations and symptoms may deve-
lop earlier and may be more severe.4,17,19 Trans­
mission of hydatid illness with biliary tree has
been described in up to 90% of hepatic cysts.22-24
Rupture into the biliary tree (5–45%) is reported
to be the most common complication of hepatic
hydatid cyst, followed by suppuration of the cyst
and communication to the pleural, pericardial, and
peritoneal cavity.10,24-28 However, a few unusual
presentations of hepatic hydatid cyst reported in
the literature are concomitant rupture into the
pleural cavity and biliary tree,18,28,30 rupture into
the duodenum,23,31,32 spontaneous intraperitoneal
rupture,33 and anaphylactic shock due to spon
taneous rupture into the peritoneal cavity.19,21,34
Other places of hydatid cysts are very changeable.
The lungs are the second most common place of
hematogenous spread in adults and are probably
the most common place in children (15–25% of
cases).4,9 Collapsible organs such as the lung or
brain facilitate cyst growth and are suggested as a
reason for the high prevalence of childhood ill-
ness. It has been reported that the central nervous
system is affected in 1% of hydatid cyst cases and
Table I WHO Classification According to the Ultrasonographic Appearance of the Hydatid Cyst
The 2001 World Health Organization (WHO) classification of hepatic hydatid cysts
CL Unilocular anechoic cystic lesion without any internal echoes and septations
CE1 Uniformly anechoic cyst with fine echoes settled in it representing hydatid sand
CE2 Cyst with multiple septations giving it a multivesicular, rosette, or honeycomb appearance, within a unilocular mother cyst
CE3a Cyst containing a floating membrane due to detachment of the endocyst
CE3b Cyst with a predominantly solid content due to membranes and few peripheral daughter cysts
CE4 Mixed hypo- and hyperechoic contents with absent daughter cysts; these contents give the appearance of a ball of wool (ball of
wool sign), indicating the degenerative nature of the cyst
CE5 Arch-shaped, thick, partially or completely calcified wall
Figure 1 Axial T2-weighted MR image obtained at the same
level shows multiple vesicles within the mother cyst and shows
the detached membrane of a hydatid cyst at the liver (red
asterisk).
Figure 2 Axial T2-weighted MRI showing perigastric fluid and
lobulated thick wall cysts at the antral mucosa (red asterisk). It is
clearly seen that cyst rupture is direct on MRI. The pathway of
the hepatic hydatid cyst is seen to be the hepatogastric ligament
(a part of the lesser omentum).

4. Volume 43, Number 1/February 2021 37
Hepatic Hydatid Cyst Perforating into the Gastric Antrum
is usually diagnosed in childhood.2,9 In the lungs,
multiple cysts occur in 30% of cases, 20% are bilate-
ral, and 60% are in the lower lobes.4,30 Calcification
in pulmonary cysts is seen rarely (0.7% of cases),20,30
which may be seen in pericardial, pleural, and me­
diastinal cysts.7,21 Renal involvement occurs in 3%
of cases2,9,21 and usually remains asymptomatic
for many years. The widespread indications and
symptoms are side mass, pain, and dysuria.4,9 Cysts
are often solitary and located in the cortex, and
they can reach 10 cm before any clinical symptoms
are seen.9 The prevalence of splenic involvement
in hydatid cyst illness ranges from 0.9–8%.22,35 The
spleen is the third most common organ of hyda-
tid illness involvement after the liver and lungs.22
Splenic hydatid illness is mainly produced by
sys­
temic or intraperitoneal spread from a rup-
tured liver cyst.21,35 The most common clinical
manifes­
tations and symptoms are abdominal pain,
splenomegaly, and fever.35 The density of bone
involve­
ment in hydatic disease is 0.5–4%.36 It is
most commonly seen in the spine and pelvis, fol-
lowed by the femur, tibia, humerus, skull, and
ribs.4,36,37 Hydatid illness may include almost any
anatomic region due to hematogenous dissemina­
tion. Un­
conventional locations include orbit, gas­
tric wall, retrocrural space, the heart, pericardium,
mediastinum, subcutaneous space, muscle, and
adrenal glands.4,38-45
Three different types of cyst rupture have been
described in the literature: contained, communi-
cating, and direct.2,19,27,46 Internal ruptures occur
when endocyst ruptures occur but the pericist re­
mains intact. The endocyst compartment appears
in cross-sectional view as floating membranes
within the cyst. Contained rupture may be rela-
ted to degeneration, trauma, or response to ther­
apy. Concomitant rupture refers to passage of the
cystic contents into the biliary rootlets that have
been associated into the pericyst.19,27 Direct rup-
ture occurs when both the pericyst and endocyst
rupture, allowing free spillage of hydatid content
into the peritoneal cavity, pleural cavity, abdomi-
nal wall, hollow viscera, and other gastrointestinal
organs.23 The type of cyst rupture in this case was
direct. Anatomically, the cyst perforated both the
liver and the antrum region of the stomach (Fig-
ures 2–3). Mehta et al47 reported direct communi­
cation of a hepatic hydatid cyst with the gastric
lumen causing a reactionary cicatrization of the
antrum and pylorus, causing, in turn, gastric out-
let obstruction similar to our findings. They also
noted that their case is the first one in the English
literature to report direct communication of hep-
atic hydatid cyst with the gastric lumen and caus­
ing a reactionary cicatrization of the antrum and
pylorus, causing gastric outlet obstruction. Al-
though the incidence of hepatic hydatid cyst with
perforation to stomach (gastrointestinal organs,
duodenum, etc.) is rare, very few studies have
reported this direct perforation to the stomach or
other gastrointestinal organs so far in the litera­
ture.11-16 Thomas et al11 reported that tomography
Figure 3 Coronal MRCP shows a hepatic hydatid cyst
originating in the posterior segment of the right hepatic lobe
growing through the hepatogastric ligament into the gastric
antrum of the stomach. Seen are the gallbladder (white asterisk),
hydatid cyst of liver (red arrow), and hydatid cyst of the gastric
antrum (black asterisk).
Figure 4 The pathway of the hepatic hydatid cyst at the
posterior view, shown anatomically. Figure taken from Visible
Body, Digestive System (iPad app, Apple Inc.), and readjusted to
show the pathway of the hepatic hydatid cyst.

5. 38 Analytical and Quantitative Cytopathology and Histopathology®
Ertuğrul et al
scan revealed hydatid cysts communicating with
the stomach and duodenum. In addition, Rueda
Elias et al13 also observed a hepatic hydatid cyst
with perforation to the stomach. The hydatid cyst
can be used to move natural pathways provided
by the liver capsule, ligaments, and peritoneum be-
yond the liver boundaries.13 The two most com-
mon paths of exophytic growth are the nonperi-
toneal area of the liver and the gastrohepatic liga-
ment. Direct rupture is more common in lesions
on the side of the liver, where there may be less
protection for a missing pericardium and a cyst
attached to a small host tissue to provide support.7
In concomitant rupture and direct ruptures, the
cyst is empty and smaller and less spherical.7,19,48
MRI, US, and CT may demonstrate a cyst wall
defect and passage of the cystic contents through
the defect, particularly in direct communication.
CT may display the same findings as seen on
US.7,19 MRI may demonstrate interruption in the
low-signal-intensity rim of the cyst wall as well as
extrusion of contents through the defect.23 Hep-
atic hydatid cysts may have a low signal intensity
ratio on T2-weighted MRI. If present, daughter
cysts are seen as cystic structures attached to the
germinal layer that are hypointense to intracystic
fluid on T1-weighted images and hyperintense on
T2-weighted images.49
In conclusion, we discussed in detail the loca-
tion of a hydatid cyst and the associated clinical
findings, especially in the liver and other organs.
Based on its incidence, a hepatic hydatid cyst
perforating into the stomach region is a matter of
grave concern. Hence, we demonstrated, via MRI,
a ruptured hepatic hydatid cyst perforating into
the liver and the gastric antrum of the stomach.
This atypical location of a hepatic hydatid cyst may
be helpful in making an accurate diagnosis and
planning treatment.
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