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J Parasit Dis (July-Dec 2012) 36(2):265268

DOI 10.1007/s12639-012-0127-y

SHORT COMMUNICATION

Hydatid disease in childhood: revisited report of an interesting


case
Zeeba Shamim Jairajpuri Sujata Jetley
Md. Jaseem Hassan Musharraf Hussain

Received: 2 April 2012 / Accepted: 26 May 2012 / Published online: 17 June 2012
Indian Society for Parasitology 2012

Abstract Hydatid disease is a zoonosis caused by the


tapeworm of Echinococcus spp. The disease is widely
endemic in many sheep and cattle rearing locales. However, hydatidosis does not remain restricted to endemic
geographical locales anymore but rather is a global health
concern. It is a major public health burden causing significant morbidity and mortality. Echinococcus granulosus
involvement in children has a different pattern than adults.
Children of all age groups are susceptible and localization
of the disease in the lungs is more commonly seen. Multiple liver cysts in the paediatric age group is relatively
uncommon. We report an interesting case of multiple liver
cysts in a 5-year old boy which was diagnosed as hydatid
cysts on histopathological examination.
Keywords

Hydatid  Cyst  Children  Liver

Z. S. Jairajpuri  S. Jetley (&)  Md. J. Hassan


Department of Pathology, Hamdard Institute of Medical
Sciences and Research, Jamia Hamdard, Hamdard Nagar,
New Delhi 110062, India
e-mail: sujatajetley@rediffmail.com
Z. S. Jairajpuri
e-mail: zeebasj@rediffmail.com
Md. J. Hassan
e-mail: jaseemamu@gmail.com
M. Hussain
Department of Surgery, Hamdard Institute of Medical Sciences
and Research, Jamia Hamdard, Hamdard Nagar,
New Delhi 110062, India

Introduction
Hydatid disease is a zoonosis caused by the tapeworm of
Echinococcus spp. It is prevalent where livestock is raised
in association with dogs. The disease is widely endemic in
many sheep and cattle rearing locales such as Australia,
Latin America, Africa and Middle Eastern countries like
Iran. (Akhter et al. 2011). India, though not primarily a
sheep and cattle rearing country, hydatid disease has been
reported from many diverse locations such as Madras,
Ahmedabad and some parts of Himachal Pradesh (Vamsy
et al. 1991). With increased migration of populations the
prevalence of the disease has spread to other regions also
(Sumer et al. 2009). Hence, contrary to the popular perception, hydatidosis does not remain restricted to endemic
geographical locales anymore but rather is a global health
concern, particularly in the rural countryside and semiurban localities. It is a major public health burden causing
significant morbidity and mortality. Hydatid disease is a
parasitic infestation caused by Echinococcus spp., the most
common being Echinococcus granulosus; others are E.
multilocularis and E. vogeli. The primary hosts are dogs
while humans are accidental intermediate hosts infected
through ingestion of contaminated food with dog faeces or
through direct contact with dogs (Sherwani et al. 2003).
Most infected persons are asymptomatic and clinical
manifestations vary according to the anatomic location of
the cyst (Zargar-Shoshtari et al. 2007). The disease can
occur in any organ and many unusual presentations of
Echinococcus have been reported (Abu-Eshy 1998).
Echinococcus granulosus involvement in children has a
different pattern than adults. In adults, liver is the most
common site of localization of the larval forms whilst in
children it is more common in the lungs. However, multiple liver cysts in the paediatric age group is relatively

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J Parasit Dis (July-Dec 2012) 36(2):265268

uncommon. We report an interesting case of multiple liver


cysts in a 5-year old boy which was reported as hydatid
cysts in the liver. This report also attempts to review literature on hydatid disease with reference to the pediatric
age group.

Case report
A 5-year old male child, resident of the nearby slum,
presented to the surgical outpatient department with history
of abdominal distension and pain. History of fever off and
on accompanied by loss of appetite and jaundice was
elicited. His past history revealed a small fall from a
staircase a week ago. No history of canine contact was
obtained on direct questioning. On examination, evidence
of external injury was not seen and a palpable mass in the
right hypochondrium was noted. Laboratory investigations
and radiological imaging were ordered. Peripheral blood
findings revealed a haemoglobin of 11.2 g/dl and leukocytosis with a total leukocyte count of 13,000 cells/mm3
Neutrophilia (85 %) was predominant on differential leukocyte count and erythrocyte sedimentation rate was marginally elevated. Other routine tests including liver
function test and renal function test along with urine
examination were within limits. Abdominal ultrasonography was non specific, demonstrated multiple cystic liver
masses with few internal echoes. A primary diagnosis
suggestive of a simple liver cyst with haemorrhage was
made. Computerized tomographic scan (CT scan) was
requested for a more conclusive opinion. It showed multiple rounded hypodense lesions with peripheral enhancement in segments II, III, IV and VIII of the liver (Fig. 1),
suggesting a possibility of liver abscess or hydatid cyst.
Hence, keeping in view the clinical symptoms and imaging
findings, the patient was taken up for surgery. Exploratory
laparotomy with removal of cyst and omental packing was
done. All three liver cysts were removed and sent for histopathological evaluation. On gross examination, three
pearly white membranous cysts were seen (Fig. 2) along
with fibrous capsule i.e. the pericyst layer (Fig. 3). Histopathological examination of the cyst wall showed a chitinous lamellated layer (Fig. 4), germinal layer was not
distinctly evident. A confirmed diagnosis of hydatid cyst
liver was made.

Fig. 1 Computerized tomographic scan showing multiple liver cyst

Fig. 2 Gross picture of the hydatid cysts

Discussion
Hydatid disease is an important health problem in many
endemic countries and needs epidemiologic measures for
its eradication (Tantawy 2010). Accidental ingestion of the
embryonated eggs of E. granulosus following close contact

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Fig. 3 Gross appearance of the pericyst

J Parasit Dis (July-Dec 2012) 36(2):265268

Fig. 4 Photomicrograph of the chitinous laminated membrane

with dogs is usually the starting point of this disease.


Although many of the recorded cases of cystic Echinococcus are in adults, most human infections occur during
childhood and adolescence, the disease being more prevalent among the rural populations (Todorov and Boeva
2000). Most cysts found in adults are generally hepatic in
location and relatively few are seen in the lungs (Todorov
and Boeva 2000; Mirshemirani et al. 2011). Several studies
of hydatid disease in children however, have recorded a
higher incidence of pulmonary hydatidosis (Recep et al.
2000; Mamishi et al. 2007), while S ehitogullari (2007)
showed that hydatid cysts in the liver were more common
(S ehitogullari 2007). In contrast, an equal incidence of
hydatid cyst in lung and liver in pediatric patients was also
reported. (Talaiezadeh and Maraghi 2006). The authors
also postulated that the less dense nature of the liver tissue
in children as compared to adults allows the scoleces to
cross the liver barriers into the lungs. Our patient presented
with multiple liver cysts, an uncommon presentation as
compared to other pediatric case reports.
Despite an almost equal distribution of the disease in
both sexes in adults (Bulent et al. 2008) the predominance
in boys over girls has been described in many series
(Senyuz et al. 1999; Talaiezadeh and Maraghi 2006 Tantawy 2010; Mirshemirani et al. 2011) This has been
attributed to behavioral differences between both the sexes,
with more outdoor exposure of boys, as suggested by
Tantawy (2010). Patients age is known to range from 1 to
14 years although hydatid cyst in a 6 month old infant has
also been reported (Prashant et al. 2007). Hence children of
all age groups are susceptible. In concurrence with trends
in literature, our patient was a 5-year old male child.
Most of the cases of hydatid disease come from rural
areas or from people who have settled in urban localities
after spending a majority of their life in villages. Many

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patients usually acquire the disease during childhood, but


do not present with the clinical signs and symptoms until
late adulthood (Lewall and Mc Corkell 1986). Errors in the
clinical diagnosis of hydatid disease are frequent due to
lack of any typical clinical presentation of the disease and
imaging features which can also be variable. The variation
of clinical presentation depends on the size and site of the
lesion as well as the accessibility of the organ involved in
the clinical examination (Tekinbas et al. 2009). The two
most common sites of involvement are lungs and liver,
however no organ is spared. Abdominal mass is the most
common complaint in cases of liver involvement as seen in
our case, and similar to other studies (Talaiezadeh and
Maraghi 2006). The natural progression of untreated cyst
may include calcification and death of the cyst, however,
more frequently, the cyst gradually enlarges in size (Ammann and Eckert 1996). Hydatid cyst fluid is highly
allergenic and carries a risk of anaphylaxis, thus the
intraperitoneal rupture of a hydatid cyst is a life threatening
emergency that can be potentially fatal (Sumer et al. 2009).
Hydatidosis can affect the brain, heart, kidney, ureter,
spleen, uterus, fallopian tube, pancreas, diaphragm, bone,
and muscles (Abu-Eshy 1998). Mamishi et al. (2007), in a
retrospective study on hydatidosis in 31 children reported
multiorgan involvement in only three of the cases. In
contrast, higher incidences of multi-organ hydatid cyst
disease in adults have been reported by other workers
(Ishikawa et al. 2009; Mishra et al. 2010). Intra-cranial
hydatid cysts are more common in the paediatric age group
and cerebrospinal fluid cytosmears may provide confirmatory evidence of scolices (Sherwani et al. 2003). Diagnostic possibilities of cystic hepatic lesions are many
especially when confronted with the presence of a complex
hepatic cyst. Simple hepatic cysts are often encountered
and when found in children, tend to be small. However,
large cysts, greater than 4 cm, are also known to occur
(Blonski et al. 2006).They can be complicated by peripheral calcification and internal hemorrhage. The appearance
of hydatid cysts on sonography is variable and depends on
the stage of maturity (Caremani et al. 2003). Hydatid cysts
may at first appear similar to simple cysts on imaging.
Multiple echogenic foci due to hydatid sand may be seen
within the lesion and may be mistaken for simple cyst with
haemorrhage, as was in the present case. The diagnosis can
be difficult in these complex cases and therefore, differentiation from hydatid cyst is necessary.
We postulated that the history of fall and the ambiguous
sonographic findings led to the initial diagnosis of a complicated simple liver cyst. Surgical intervention was necessary to relieve the patients symptoms and also to
confirm the clinical diagnosis. In conclusion, though
hydatid cysts are seen in children of all ages, localization in
the lungs is more common than the liver and no organ is

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spared. This case had an unusual presentation in the form


of multiple liver cysts in a 5-year old boy. Prevention of the
disease by breaking the transmission cycle is of paramount
importance as with all zoonotic diseases. Measures like
proper education, creating awareness and maintaining
hygiene will help to control the spread of the disease.

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