<title>1. INTRODUCTION</title> <p>Hydatid
disease is a parasitic infection caused by Echinococcus, which is endemic in
farming areas. It primarily involves the liver and presents as the gastrointestinal
manifestations such as abdominal pain, hepatomegaly, anorexia, vomiting, and
jaundice. The primary involvement of the pelvic cavity is a very rare entity and
patients usually present with pressure symptoms affecting the adjacent organs [##REF##11931192##1##–##REF##9853499##5##].
Herein, we report this interesting case that presented differently and also
discuss the significance of the awareness of physicians about this rare but
well-documented disease.</p>
<title>3. DISCUSSION</title> <p>Hydatid disease is a parasitic disease
caused by the larval stage of the tapeworm <italic>Echinococcus granulosus</italic>. While
liver and lung are the most commonly affected areas in adults, hydatid
cysts may develop in almost any part of body [##REF##11931192##1##, ##REF##2585340##2##, ##REF##9563934##4##]. Primary hydatidosis of
the pelvic cavity is very rare but well documented in endemic areas such as the
Mediterranean countries, South America, the Middle East, and Australia
[##REF##11931192##1##, ##REF##12530482##3##]. Our patient is
the first reported case of primary hydatidosis of the pelvic cavity from Iran
and underlines the difficulties in the diagnosis of cases with striking
resemblance to malignant disease of the reproductive tract. To our knowledge,
the primary involvement of the pelvic cavity usually presents with pressure
symptoms affecting the adjacent organs [##REF##9853499##5##] and this manifestation has been
reported exceedingly rare in the English literature so far [##REF##12530482##3##]. Diagnosis of
the hydatid cyst is mainly on the basis of serologic tests and/or ultrasonography
and CT scan. However, surgical exploration may be necessary for definitive
diagnosis [##REF##9853499##5##–##REF##11045332##8##]. Previous history of hydatid cysts or exposure to dog and farm
animals should raise the suspicion of this diagnosis. Following
the surgical diagnosis in our patient, we
inquired about such a history in our patient but revealed no identifiable
exposure. Specific serological methods using specific antigens,
especially native AgB, have been recommended for proper diagnosis because the serological
tests using crude antigens are sensitive, but their specificity is not
satisfactory. In one study, the enzyme-linked immunosorbent assay (ELISA)
system is much more specific in detecting antihydatid cyst antibody than countercurrent
immunoelectrophresis (CCIEP), while CCIEP is more sensitive in detecting
antihydatid cyst antibody [##REF##17767016##9##]. Interestingly,
we carried out the CCIEP assay immediately
after the confirmatory result of the pathology that was positive for hydatid
cyst therefore serologic tests in the patients with suspicious diagnosis may
render the diagnosis of the hydatid cyst. Ultrasound is an important
imaging modality for hydatid disease and may clearly demonstrate the floating
membranes, and daughter cysts characteristically seen in purely cystic lesions.
The ultrasonographic findings range from purely cystic lesions to a completely
solid appearance [##REF##18362529##10##]. In another study, CCIEP could detect only 62.0% of
cases, whereas the pathology and ultrasound results were positive for 96.3% of
cases. This study emphasized the usefulness of ultrasound and suggested that CCIEP
may be useful for diagnosing cystic <italic>Echinococcus</italic> only in doubtful cases
[##REF##15332731##11##]. CT and magnetic resonance
imaging (MRI) play a key role in recognizing the complications such as rupture
and infection of cysts associated with hydatid disease. We believe that CT scan—because of its
capability for better evaluation of the cystic masses, and better demonstration
of their extension in the pelvic cavity as well as excellent depiction of the
visceral organs involvement—is superior to
the ultrasonographic examination. The scan in suspected hydatid disease should
include the whole abdomen from liver to pelvis and a chest radiograph should
also be obtained. Skin tests, complement
fixation, blood eosinophil count, and indirect hemagglutination tests can also
be used for diagnostic purpose however their tendency toward the false-positve
results limits their validity. The gold standard test for diagnosis of
hydatidosis is microscopic examination that shows the laminated membrane and scolices
[##REF##11931192##1##–##REF##9563934##4##, ##REF##7913060##6##–##REF##17767016##9##]. Surgical removal is the optimal treatment. Complete removal of the
parasitic cysts and fluid is the major advantage of the surgery. Single cysts
are easy to excise but due to the risk of adhesions, excision of the multiple
cysts may be difficult and even impossible [##REF##11931192##1##–##REF##7913060##6##]. We agree with this comment which
emphasizes that in the younger women, even in those with multiple cysts, every
effort should be made to preserve reproductive organs [##REF##2585340##2##]. As previously stated
in this case, extensive involvement and hard adhesion of the adnexa to the
adjacent cysts of the broad ligaments forced us to perform left-sided salpingoophorectomy.
Another important problem is dealing with intraoperative spillage of the cyst
contents that contain protoscolices and can disseminate in tissue and grow new
cysts. Furthermore, acute anaphylactic reaction may ensue secondary to the
spilled cyst fluid therefore spillage should be avoided by all means [##REF##2585340##2##, ##REF##9563934##4##–##REF##11045332##8##].
The treatment of the recurrence and complications is very difficult and definition
of the best option needs multidisciplinary approach therefore medical therapy
should be applied postoperatively in the patients with multiple cysts and
multiple initial locations. The clinical manifestation of our case such as
severe weight loss, as well as the large multicystic mass of the pelvic cavity 
erroneously
points us to the diagnosis of the ovarian malignancies. The current case
underlines the possibility of the striking resemblance between the clinical and
radiological manifestation of the hydatid cyst and malignant disease of the
reproductive organs which may make the correct preoperative diagnosis very
difficult. The tumor markers were not checked in our case however lack of the other evidences of the metastasis to the visceral
organs in the chest and abdominal CT scan and normal result of the bone scan
was the useful guidelines against the diagnosis of the ovarian malignancy.
Although normal results of the tumor markers such as CEA and CA-125 may be
found in the patients with genitourinary malignancies especially ovarian
cancer, we note that these markers should be
evaluated in the patients with the suspicious diagnosis of the genitourinary
malignancies. In conclusion, primary hydatidosis of the pelvic cavity should
always be considered in the differential diagnosis of any tumor-like growing
mass even in the absence of accompanying involvement of liver or other visceral
organs. We believe that in the cases like our patient, the most important
factor in diagnosis of hydatid disease of the pelvic cavity is the high index
of suspicion about its possibility which can provide the accurate diagnosis and
prevent the erroneous treatment. The
other important consideration is the
accidental rupture of hydatid cyst during surgery which may be life threatening,
therefore preoperative diagnosis of this
rare lesion is very important. The patient's history as well as the
serologic tests may yield important clues about the diagnosis, furthermore the
radiologist's familiarity with the imaging findings of the disease is very important
for earlier diagnosis and an appropriate treatment.</p>
<p>Recommended by Daniel Landers</p> <p>We report and discuss a case of primary hydatidosis of the pelvic cavity in a woman who presented with severe weight loss and abdominal pain. This unusual presentation was initially considered as a tumor process until surgical exploration and microscopic studies confirmed the diagnosis. The gynecologists should be aware of possibility of primary hydatid cyst of the pelvic cavity and should be considered in the differential diagnosis of cystic pelvic masses, especially in areas where the disease is endemic.</p>
<title>2. CASE REPORT</title> <p>A 34-year-old woman was admitted to our
center with complaints of lower abdominal pain, anorexia, constipation, and
significant weight loss (more than 10 Kg during 4 months). There was no history
of fever, nausea, or vomiting. Habitual and occupational history was
uneventful. She had 3 normal full-term deliveries. As the patient had severe
constipation and other gastrointestinal symptoms, colonoscopy had been carried
out in another center with normal findings. 
On pelvic examination a nontender, uniformly cystic
mass approximately 28-week size was palpated. Speculum examination was normal.
Laboratory investigations including blood sugar, blood urea nitrogen,
creatinine, liver enzymes, and urinalysis were normal and stool occult blood
was negative. Pregnancy test was negative. Complete blood count was normal
except mild anemia (Hb: 7 gr/dL). Chest radiograph was also normal. On transabdominal
ultrasound, multiple anechoic masses were demonstrated in the pelvic and
uterine cavities. For further delineation of the cystic masses and disease
extension in the pelvic cavity, computed tomography (CT) scan was performed, that
revealing multiple cystic space-occupying lesions in the pelvic cavity,
involving the uterus, broad ligament, and adnexa (##FIG##0##Figure 1##). The patient
underwent an exploratory laparotomy under general anesthesia for further
diagnosis of the cystic mass, and ruling out of the genitourinary malignancy. Preoperative laboratory evaluation including HIV antibody,
VDRL test, HBS antigen, and HCV antibody assay were normal. Additionally, whole
body bone scan, chest and abdominal CT scan were also performed that revealed
unremarkable results. On the 10th day of her admission after obtaining the
informed consent, the patient underwent surgical exploration. At the operation,
numerous ovarian and paraovarian cystic masses densely adhered to the uterus,
to the pelvic side wall, and to the fallopian tubes were demonstrated. One of
the cysts was removed completely and sent to the laboratory and this surgical
specimen was diagnosed as the hydatid cyst. Due to extensive involvement of the
uterus, adnexa, and broad ligament, complete removal of the cysts and preservation
of the all reproductive organs was impossible; furthermore complete removal of
the cysts due to the high risk of rupture could be life threatening, so after
obtaining the signed consent we decided to perform removal of the cysts of the
right adnexa and uterus with preservation of them and left side salpingoophorectomy. The rest of the abdomen and pelvis were free of pathology. The abdomen was then carefully irrigated with isotonic saline. Microscopic
examination disclosed the scolices of <italic>Echinococcus granulosis</italic> with
adjacent laminated membrane and confirmed the diagnosis (##FIG##1##Figure 2##). The patient
recovered uneventfully and was discharged on the 11th postoperative day. Albendazole
(800 mg per day) as adjuvant therapy was administered for 4 months
postoperatively. Blood count and liver transaminases were checked during the
course of therapy which showed normal results. At 6-month follow-up the patient
was doing well with no detectable abnormality on follow-up ultrasound.</p>
<fig id="fig1" position="float"><label>Figure 1</label><caption><p>Axial
pelvic CT scan demonstrates multiple cystic masses (arrow) in the pelvic cavity
and also the uterus (arrowhead).</p></caption></fig> <fig id="fig2" position="float"><label>Figure 2</label><caption><p>Microphotograph of the lesion reveals laminated membrane (large arrow) and the scolices (small arrow).</p></caption></fig>
<graphic xlink:href="IDOG2008-782621.001"/> <graphic xlink:href="IDOG2008-782621.002"/>
[]
Infect Dis Obstet Gynecol. 2008 Aug 28; 2008:782621