International Surgery Journal

Porwal R et al. Int Surg J. 2016 Aug;3(3):1538-1542

http://www.ijsurgery.com pISSN 2349-3305 | eISSN 2349-2902

DOI: http://dx.doi.org/10.18203/2349-2902.isj20162743
Research Article

Queer localizations of primary hydatid disease:

experience from a single institute
Rekha Porwal, Hanuman Prasad Gupta, Amit Singh*, Rajesh Kumar Singh

Department of Surgery, JLN Medical College, Ajmer, Rajasthan, India

Received: 23 May 2016

Accepted: 02 July 2016

*Correspondence:
Dr. Amit Singh,
E-mail: [email protected]

Copyright: © the author(s), publisher and licensee Medip Academy. This is an open-access article distributed under
the terms of the Creative Commons Attribution Non-Commercial License, which permits unrestricted non-commercial
use, distribution, and reproduction in any medium, provided the original work is properly cited.

ABSTRACT

Background: Cystic hydatid disease is a zoonosis caused by Echinococcus granulosus. It may affect any organ and
tissue in the body, in particular the liver and Lung. Primary extrahepatic hydatid cysts are rare, can have atypical
presentations and can pose a diagnostic challenge.
Methods: This retrospective study was carried out in the Department of Surgery at tertiary care hospital in Ajmer,
Rajasthan, India. Twenty patients diagnosed with hydatid cysts without involving liver were included in this study.
All patients were managed surgically from January 2009 to December 2015 and evaluated retrospectively collected
data.
Results: The cysts were present in lungs (30%), kidney (15%), spleen (10%), subcutaneous (10%), inguinal canal
(5%), breast (5%), intramuscular (10%), thyroid (5%), retroperitoneal (5%), parotid (5%)..
Conclusions: The occurrence of Hydatid cystic disease in locations sites other than liver and lungs is very rare.
Symptoms are related to size, location or possible complication of the cyst. Hydatid disease is a differential diagnosis
of cystic lesions, especially for the cystic lesions encountered in patients who live in or have come from endemic
region.

Keywords: Queer locations, Hydatid, Echinococcosis, Zoonosis

INTRODUCTION frequently, the spleen, kidneys, heart, bone and central

nervous system and head and neck region.2
Echinococcosis (hydatid disease) is a zoonosis caused by
the larval stage of Echinococcus granulosus. Humans are METHODS
accidental intermediate hosts, whereas animals can be
both intermediate hosts and definitive hosts. Hydatid Hydatid disease in atypical region without primary
disease is endemic mainly in the Mediterranean countries, involvement of liver is very rare even in endemic areas,
the Middle East, the Baltic areas, South America, India, and only a few case reports were found in the literature.
China; however, owing to increased travel and tourism all Herein, we report cases diagnosed with Hydatid cysts in
over the world, it can be found anywhere, even in atypical locations without involving liver and discuss
developed countries.1 In India, Hydatid disease is diagnosis and management. Twenty patients with primary
common in most of the states of which Andhra Pradesh, Hydatid cysts in atypical locations managed surgically
Tamil Nadu and Jammu-Kashmir predominate. from January 2009 to December 2015 at J.L.N Medical
College, Ajmer, Rajasthan, India were evaluated
The most frequently involved organ is the liver (65%), retrospectively. Preoperative diagnosis was established
other organs involved are the lungs (25%) and, less by the history; clinical examination, complete blood

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 Porwal R et al. Int Surg J. 2016 Aug;3(3):1538-1542

counts, liver and kidney function tests, X-ray chest, the cyst. Palpable lump was also a presenting symptom in
ultrasound and contrast enhanced computerized patients with subcutaneous and muscle cysts. In one case
tomography (CECT). The treatment for all the patients Hydatid cyst was present in inguinal canal and mimics
was surgical. All the patients received postoperative obstructed inguinal hernia, and in one case cyst was
Albendazole chemotherapy. present in breast which mimics breast abscess (Table 1).
Surgical treatments include complete cyst excision in
RESULTS most of the patients. Nephrectomy, splenectomy,
incision and drainage with cyst excision (breast) were
Table 1: Various queer locations of hydatid cystic performed whenever cysts invaded these organs. All the
disease, their clinical presentation and management in patients were given postoperative chemotherapy
present study. (Albendazole 10 mg/kg/day) for three cycles of 21 days
each with a gap of 1 week between each cycle. Morbidity
Cyst location Number Symptoms (n) Surgical was minimal, with a wound infection recorded
(%) treatment postoperatively in two cases. The diagnosis of Hydatid
Lungs 6 (30%) Chest pain-3 Thoracotomy cyst was confirmed on histopathological examination of
Cough with with the specimen in all cases.
expectoration- cystectomy
2
Hemoptysis-1
kidney 3 (15%) Lump Nephrectomy,
abdomen, cystectomy
upper
abdominal pain
Spleen 2 (10%) Splenomegaly, Splenectomy
upper
abdominal
pain,
subcutaneous 2 (10%) swelling cystectomy
Inguinal canal 1 (5%) Swelling cystectomy
mimicking
obstructed
inguinal
hernia.
breast 1 (5%) Lump breast Incision and
mimicking drainage of
abscess breast cyst with
cystectomy
retropritoneum 1 (5%) Lump cystectomy Figure 1: Intramuscular location of hydatid cyst in
abdoman
thigh region.
Intramuscular 2 (10%) Swelling cystectomy
Cold abscess
DISCUSSION
Thyroid 1 (5%) swelling cystectomy
Parotid 1 (5%) swelling cystectomy
Hydatid disease is seen endemically among sheep-raising
communities. As India is not a major sheep-rearing
Hydatid disease involving atypical sites without country, it seems that this disease is uncommon here.
involvement of liver was seen in a total of 20 patients. Of However the present study is a clinical study of Hydatid
these patients 7 were male and 13 females, the sex disease in central Rajasthan in which practice of sheep
incidence revealed female preponderance in the study and goat grazing is quite common and most of the rural
(M: F: 1: 2). Patient age ranged from 22 to 65 years. The background persons who have goat and sheep moves
mean age was 38.6 years. whole of the year in search of grass in desert area. In the
present study, most of the patients were from rural areas
Majority of the patients belongs to, fourth decade of life. associated with farming and field work and kept domestic
Majority of patients were from rural areas (14) and animals including goat, sheep and dog. Various studies
remaining (6) was from urban areas. History of contact have reported mean age of nearly 40 years which is
with dogs, goat and sheep was available in eight cases. similar to our study.3,4 Imaging modalities such as
The cysts were present in lungs (30%), kidney (15%), ultrasonography, CT scan, and MRI have been the
spleen (10%), subcutaneous (10%), inguinal canal (5%), methods of choice, especially the latter, which has been
breast (5%), intramuscular (10%), thyroid (5%), the diagnostic method of choice for the preoperative
retroperitoneal (5%), parotid (5%) (Table 1). diagnosis of the Hydatid cyst in most unusual
Nonspecific abdominal pain and a nontender palpable locations.5 In our study, ultrasonography was done in 16
abdominal lump were the most predominant symptoms; cases and proved diagnostic in only 11 cases and CECT
other symptoms varied according to the localization of scan was done in 13 (65%) cases, It was 100%

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 Porwal R et al. Int Surg J. 2016 Aug;3(3):1538-1542

diagnostic. Three patients of pulmonary Hydatid disease duration. There was no history of injury, discharge from
in the present study presented with chest pain, two the nipple and family history of breast
patients presented with cough with expectoration, and cancer. Examination revealed a red inflamed tender area
one patient had minimal haemoptysis. Chest x-ray study measuring 5 cm × 5 cm in the subareolar region
is of more help in diagnosis of pulmonary hydatids than extending to upper outer quadrent of the left breast. The
liver. On chest film, an intact pulmonary Hydatid cyst nipple of the left breast was retracted. The right breast
appears as rounded homogenous shadow with well- and nipple were normal and there was no axillary or
defined margins.6 The reported prevalence of splenic cervical lymphadenopathy. USG study of the breasts
involvement by Hydatid disease varies from 0.9% to 8%.7 revealed a thick walled cavitary lesion with internal
Hydatid cyst is the only parasitic cyst of the spleen and it echoes in the subareolar region of the left breast
is said to be twice as common as the non-parasitic suggestive of abscess cavity. The patient underwent
variety. Any type of Hydatid cyst can be seen in the surgery for the abscess left breast. This case was
spleen. Isolated splenic involvement itself is very diagnosed as Hydatid cyst incidentally during surgery,
uncommon. Two cases of Hydatid cyst affecting the and confirmed by histopathology.
spleen were seen in the present study, an incidence of
10% and were managed by splenectomy. Primary Soft tissue Hydatid disease is rare even in endemic areas,
Hydatid of the kidney is rare entity and is responsible for and a primary focus within muscle in the absence of
only 2 to 3% of all Hydatid disease.8 Renal involvement pulmonary or hepatic involvement is most unusual.
could be primary or secondary, in primary Hydatid Muscle Hydatidosis accounts only for 3-5% of all cases.11
disease cyst passes through the portal system into the In present study A 45-year-old female patient presented
liver and retroperitoneal lymphatic. The Hydatid cyst of to our surgical outpatient department, with a history of
the kidney is considered closed if all three layers of the painless, slow growing swelling in posterior aspect of her
cyst are intact. When the cyst goes outside the pericyst left leg since 10 years. On physical examination, the
confined by the lining of collecting system it is swelling was non-tender, measuring approximately 10 cm
considered to be an exposed cyst. If all the three layers of x 6 cm in size in the calf region of left leg. A clinical
the cyst have ruptured resulting in free communication diagnosis of a lipoma or a large sebaceous cyst was made
with the calyces and pelvis, it is called an open or and FNAC was done which was suggestive of
communicating cyst. Three cases of Hydatid disease suppurative inflammatory pathology. The diagnosis of
affecting the kidney were seen in this study, an incidence Hydatid cyst was set intraoperatively. A postoperative
of 15%. In present study one case of renal Hydatid was CTscan of the thorax and USG abdomen revealed no
associated with massive renal destruction which was signs of other echinococcal cysts. Thus, the case was
treated with nephrectomy, remaining two cases of considered as a primary Hydatid cyst of the calf. Another
isolated renal Hydatid treated with cyst excision alone. In case with intramuscular Hydatid disease in thigh region
present study one case of Hydatid cyst was found in clinically resembles cold abscess and diagnosed
inguinal canal associated with outside the sac of indirect intraoperatively (Figure 1).
hernia, signs and symptoms were in favour of obstructed
inguinal hernia and sonography was inconclusive, The mechanism of primary subcutaneous localization is
inguinal canal exploration presumed diagnosis and cyst unclear. There are two potential mechanisms; (a) direct
excision with standard hernia repair was done, diagnosis subcutaneous contamination through an injured skin or
confirmed by histopathology. The parotid gland Hydatid (b) subcutaneous colonization of ingested eggs after
cysts are always primary.9 In present study A 36-year-old passing liver and lungs, incidence of subcutaneous
woman presented with slowly progressive swelling in the Hydatid disease is 1.5%.12 In the present study two case
right periauricular region for 6 month duration. It was was diagnosed subcutaneous Hydatid, both were leg
non-tender and mobile. On ultrasonography a cystic mass swelling and wrongly labelled as soft tissue mass or
was reported. Fine needle aspiration of a parotid mass sebaceous cyst, diagnosed intraoperatively and confirmed
revealed inflammatory swelling. Computed tomography by histopathology.
(CT) scan of the head and neck revealed a round, well-
demarcated water-density cystic mass in the right parotid The thyroid gland is an extremely rare site of Hydatid
gland. A provisional clinical diagnosis of a cyst or a cyst, even in endemic regions.13 In the present study a 26
neoplasm of the parotid gland was made. At the year old male present with solitary thyroid swelling since
operation, the cystic mass replacing most of the one year. The local examination showed a right solitary
superficial part of right parotid gland was demonstrated. thyroid nodule, measuring 2 × 1 cm, cystic, nontender,
Superficial parotidectomy was carried out. and not fixed to the surrounding structures with no
Histopathological examination confirmed the diagnosis cervical lymph node. Thyroid function tests were within
of Hydatid disease. Hydatid cyst of the breast is very normal limits. Thyroid ultrasonography (US) showed a 2
rare. The breast can be a primary site or part of a × 1.5 cm diameter cystic nodule in the right thyroid lobe.
disseminated Hydatidosis. It might mimicked FNAC revelled thyroid cyst. Cyst was excised and
fibroadenoma, phyllodes tumors, chronic abscesses, or diagnosis was confirmed by
even carcinoma.10 A 28-year-old woman presented with histopathology. Postoperative radiologic examination of
gradually progressive, painful left breast since one month

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 Porwal R et al. Int Surg J. 2016 Aug;3(3):1538-1542

the chest and US examination of the abdomen were CONCLUSION

normal.
Echinococcosis can appear at any site of the human body,
Hydatid cyst which develops only in the retroperitoneal and so should always be considered in the differential
space without an accompanied lesion in other organs is diagnosis of cystic space-occupying lesions or
defined as primary retroperitoneal Hydatid cyst. It is unidentified tumor formations in patients from endemic
extremely rare, seen in 0.8% of the cases.14 In the present areas. Hydatid diseases at unusual sites are like iceberg
study a 28 year women comes with lump lower abdomen because actual incidences are much higher than reported.
since one year. On examination 10 cm 12 cm globular Proper surgical and medical management to avoid any
nontender firm mass arising from pelvis and moves with recurrences, and a regular follow-up, are of utmost
horizontal direction with restricted mobility in vertical importance to detect any late complications such as local
direction. Ultrasound (USG) of the whole abdomen recurrence of the disease and development of Hydatidosis
revealed a 10.4 cm × 6.4 cm right adenexal at the primary sites.
multiloculated mass occupying the right pelvis with
internal debris. The liver, spleen and kidneys showed a Funding: No funding sources
normal echo pattern. On exploration both ovaries and Conflict of interest: None declared
uterus was seen normal and bulging seen from Ethical approval: The study was approved by the
retroperitoneal space, on exploration of retroperitoneal institutional ethics committee
space a cystic mass seen which was enblock removed.
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Cite this article as: Porwal R, Gupta HP, Singh

A, Singh RK. Queer localizations of primary
hydatid disease: experience from a single institute.
Int Surg J 2016;3:1538-42.

International Surgery Journal | July-September 2016 | Vol 3 | Issue 3 Page 1542