We present a case of multiple hydatidosis in an 8-year-old boy that resulted from a ruptured hydatid cyst of the spleen and spread into the peritoneal cavity. We also present a new approach for managing these difficult and high-risk cases

Deftereos, Savvas

Kirmanidis, Michail A

Limas, Christos

Soultanidis, Chrisostomos

Tsigalou, Christina

English

PubMed

Christos Limas

Chrisostomos Soultanidis

Michail A Kirmanidis

Christina Tsigalou

Savvas Deftereos

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Management of abdomen hydatidosis after rupture of a hydatid splenic cyst: a case report

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Case report
Open Access
Management of abdomen hydatidosis after rupture of a hydatid
splenic cyst: a case report
Christos Limas1*, Chrisostomos Soultanidis1, Michail A Kirmanidis1,
Christina Tsigalou2 and Savvas Deftereos3
Addresses: 1Paediatric Surgery Department, University General Hospital of Alexandroupolis, Dragana, 68100 Alexandroupolis, Greece
2Microbiology Laboratory, University General Hospital of Alexandroupolis, Dragana, 68100 Alexandroupolis, Greece
3Radiology Department, University General Hospital of Alexandroupolis, Dragana, 68100 Alexandroupolis, Greece
Email: CL* - christoslimas@yahoo.com; CS - msoultan@in.gr; MAK - mihaliskirmanidis@yahoo.gr; CT - xtsigalou@yahoo.gr;
SD - sdefter@med.duth.gr
*Corresponding author
Received: 25 June 2009 Accepted: 19 August 2009 Published: 2 September 2009
Cases Journal 2009, 2:8416 doi: 10.4076/1757-1626-2-8416
This article is available from: http://casesjournal.com/casesjournal/article/view/8416
© 2009 Limas et al.; licensee Cases Network Ltd.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0),
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Abstract
We present a case of multiple hydatidosis in an 8-year-old boy that resulted from a ruptured hydatid
cyst of the spleen and spread into the peritoneal cavity. We also present a new approach for
managing these difficult and high-risk cases.
Introduction
Hydatidosis is a parasitic disease caused by Echinococcus
granulosus in humans andmanyothermammals. Thedisease
is characterized by a worldwide distribution but is endemic
mainly around the Mediterranean Sea. There has been
recently considerable progress in the diagnosis andmanage-
ment of hydatid disease. Although the radical therapy
remains the surgical excision of the cyst, very good results
have been reported by the administration of chemotherapy
with anthelminthics (mainly albendazole). Preoperative
administration of chemotherapy aims to sterilize the cysts,
to soften them, and to reduce intracystic pressure enabling
the surgeon to remove the endocyst more easily, whereas
postoperatively is used to reduce the rate of recurrence.
Case presentation
An 8-year-old boy, Greek, presented in the emergency
department with abdominal pain, abdominal distension,
diarrheas and fever. A physical examination revealed
multiple palpable painless abdominal masses that were
mainly confined to the lower abdomen and around the
omphalic region. He had a history of acute abdominal pain
and anaphylactic reaction with urticaria rash 2 months
previously, but this had regressed withoutmedical therapy.
An ultrasound and computed tomography examination
revealed a cyst containing a “floating membrane” in the
spleen and numerous unilocular cysts in the peritoneal
cavity, measuring from a fewmillimeters to 3 cm (Figures 1
and 2) Floating membrane in the splenic cyst andmultiple
cysts in the peritoneal cavity). Laboratory data were within
normal limits except for the eosinophil count (12%) and
antibodies against Echinococcus granulosus (1/4096). The
diagnosis of multiple hydatid disease was established based
on imaging findings and positivity for anti-echinococcal
antibodies.
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We applied chemotherapy (albendazole, 10 mg/Kgr/day)
for 6 months, since the spread of the disease into the
peritoneal cavity and the multiplicity of lesions were
strong contraindications for a radical surgical approach. At
around 3 months the cysts in the peritoneal cavity had
disappeared and the splenic cyst had reduced in size.
However the echinococcal antibody titers, (which were
measured monthly), did not fall noticeably throughout
the 6-month chemotherapy period.
Our fundamental concerns on exploration were to avoid
spreading the disease and to preserve the spleen. A wide
trocar was used to open the splenic cyst and suck the
contents out. The result was negative since the contents
appeared to be gelatinous rather than watery. The splenic
cyst cleared and drained externally. The exploration of the
abdominal cavity revealed a myriad of cysts, measuring
from a few millimetres to 3 cm located only in the great
omentum (Figure 3) numerous cysts in the great omen-
tum). The surgical handlingswas very easy and therewas no
possibility of spillage since the cysts were thick, tough,
difficult to cut even with scissors, and containing only a
yellowish gelatin like substance. The greater omentum was
entirely removed, including all the cysts. The postoperative
course was uneventful, and the child was discharged on the
7th postoperative day. Six months later the child was well,
with a slow decrease in titers and no sign of remaining
disease.
Discussion
Hydatid cysts compose of three layers: the pericyst, the
laminated membrane and the endocyst (germinal layer).
The cyst fluid is a transudate of serum that contains
proteins and is antigenic [1]. The liver is the most
commonly infected organ in the human body (55-70%
of cases), followed by the lungs and spleen (20-35% of
cases combined) [2]. Splenic hydatid cysts usually coexist
with liver hydatid cysts, although in some cases the spleen
is the primary location. The cysts typically remain silent
for years, with their size and location determining the
manifesting symptoms. The first symptoms are often
caused by a complication, mainly rupture of the cyst due
to trauma or increased intracystic pressure, which causes
fever, urticaria, eosinophilia and anaphylactic shock, as
well as spreading of the disease [3].
Immunodiagnostic tests lack specificity, but they can be
helpful in the diagnosis since their sensitivity varies from
Figure 1. CT scan demonstrating floating membrane in the
splenic cyst and multiple cysts in the peritoneal cavity.
Figure 2. CT scan demonstrating floating membrane in the
splenic cyst and multiple cysts in the peritoneal cavity.
Figure 3. Numerous cysts in the great omentum.
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Cases Journal 2009, 2:8416 http://casesjournal.com/casesjournal/article/view/8416
60% to 90%. Results of various serologic tests for echino-
coccus should always be interpreted in conjunction with the
patient's medical history, clinical presentation and other
findings. The reaction can be present even years after the
surgical removal of the cyst, since blood titers decrease only
slowly [1]. A serologic survey is critical in the follow-up of
operatedpatients since apositive test during this period is not
diagnostic of recurrence, whereas a rising titter is. Although
serological tests are useful, abdominal ultrasonography
and computed tomography are necessary to establish the
diagnosis.
Surgical excision is still the only curative treatment, but
good results have been reported with albendazole admini-
stration. Medical treatment is indicated in cysts inacces-
sible for surgical removal or as a complementary therapy
to prevent recurrence [4]. Preoperative administration of
anthelminthics sterilizes and softens the cysts, and reduces
intracystic pressure, facilitating the removal of the
endocyst and reducing the rate of recurrence [5]. This
preoperative administration is reported to last from days
to 2 months [6,7]. In our opinion a longer duration
of preoperative anthelminthics administration, -up to
6 months-, will completely degenerate the cysts, facilitate
surgical handlings, minimize the risk of spillage and make
the exploration safer.
Consent
Written informed consent was obtained from the patient’s
parents for publication of this case report and accompany-
ing images. A copy of the written consent is available for
review by the Editor-in-Chief of this journal.
Competing interests
The authors declare that they have no competing interests.
Authors’ contributions
CL performed the exploration and wrote the paper, CS
took part to the exploration, and helped to the writing of
the paper, MAK took part to the exploration and reviewed
the literature, SD performed the radiological exami-
nations, CT performed the hematological examination
and reviewed the literature, All authors read and approved
the final manuscript.
References
1. Col C, Col M, Lafci H: Unusual localizations of hydatid disease.
Acta Med Austriaca 2003, 30:61-64.
2. Hamamci EO, Besim H, Korkmaz A: Unusual locations of hydatid
disease and surgical approach. ANZ J Surg 2004, 74:356-360.
3. Hayrullah D, Tugrul T, Enver R, Ali Dogan B, Okay N: Acute
intraperitoneal rupture of hydatid cysts. World J Surg 2006,
30:1879-1883.
4. Tarcoveanu E, Dimofte G, Bradea C, Crumpei F, Anton R,
Moldovanu R:Multiple peritoneal hydatid disease after rupture
of multivesicular hepatic hydatid cyst. Case report. J Gastro-
intestin Liver Dis 2006, 15:301-305.
5. Polat C, Dervisoglu A, Hokelek M, Yetim I, Buyukkarabacak Y,
Ozkutuk Y, Erzurumlu K: Dual treatment of albendazole in
hepatic hidatidosis: New therapeutic modality in 52 cases.
J Gastroenter 2005, 20:421-425.
6. Aktan AO, Yalin R: Preoperative albendazole treatment for
liver hydatid disease decreases the viability of the cyst. Eur J
Gastroenterol Hepatol 1996, 8:877-879.
7. Bildik N, Cevik A, Altintas M, Ekinci H, Canberk M, Gülmen M:
Efficacy of preoperative albendazole use according to
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41:312-316.
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Management of abdomen hydatidosis after rupture of a hydatid splenic cyst: a case report

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