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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 16  |  Issue : 1  |  Page : 75-77

Musculoskeletal hydatid masquerading as malignancy: A diagnostic dilemma


1 Department of Surgical Oncology, Sher-i-Kashmir Institute of Medical Sciences, Srinagar, Jammu and Kashmir, India
2 Department of Radiation Oncology, Government Medical College, Srinagar, Jammu and Kashmir, India

Date of Web Publication5-Jul-2017

Correspondence Address:
Mir Abdul Wahid
Department of Surgical Oncology, Sher-i-Kashmir Institute of Medical Sciences, Srinagar, Jammu and Kashmir
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2384-5589.209546

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  Abstract 

Hydatid cyst in the iliac bone is rare and is usually an incidental finding at any site other than liver and lung. It is a zoonotic disease which is due to infection with larval stage of Echinococcus granulosus. The disease is chronic, and cysts can be lodged in different organs. It has cosmopolitan distribution and impacts on health and economic indices in many countries throughout the world. Therefore, accurate information on the distribution of the disease is first step for the control and prevention. We present an unusual case of hydatid cyst found in the right iliac bone extending into the gluteal region.

Keywords: Hydatid, malignancy, musculoskeletal


How to cite this article:
Wahid MA, Ahmed SZ, Gowher HA, Butoo AJ, Ahmed SA, Nabi MG. Musculoskeletal hydatid masquerading as malignancy: A diagnostic dilemma. Afr J Med Health Sci 2017;16:75-7

How to cite this URL:
Wahid MA, Ahmed SZ, Gowher HA, Butoo AJ, Ahmed SA, Nabi MG. Musculoskeletal hydatid masquerading as malignancy: A diagnostic dilemma. Afr J Med Health Sci [serial online] 2017 [cited 2021 Jan 24];16:75-7. Available from: http://www.ajmhs.org/text.asp?2017/16/1/75/209546


  Introduction Top


Echinococcus granulosus is a cestode (tapeworm) that causes hydatid infection. These cestodes have a worldwide distribution, but the prevalence is higher in developing countries. This disease is endemic in cattle and sheep rearing regions of the world. The close association of people with sheep and dogs and the unavailability of clean potable water supply in developing countries such as India, Pakistan, New Zealand, and Mediterranean Countries make them endemic to the disease.[1] Most of the people contract the disease during their childhood but do not present until late adulthood. The natural progression of an untreated cyst may include calcification and death of the worm. However, more frequently the cyst enlarges gradually.[2] The disease is caused by echinococcal larva following ingestion of its eggs. The definitive hosts are dogs while sheep are intermediate hosts and humans are accidental intermediate hosts. The adult worms produce eggs that are released with feces and spread in various ways such as through the wind, water, or flies.[3] The disease is manifested by the presence of one or more hydatid cysts usually located in the liver or lung. The cysts of variable sizes are constituted by an external acellular cuticle and an inner cellular “germinal” layer that produces the brood capsules. The larvae (scolices) develop from the germinal layer.[4] Involvement of sites other than the liver and lung is rare, but no site is immune. Involvement of the musculoskeletal system occurs in 1– 4% of all cases.[5] In general, vascularized bones such as vertebrae, long bones, and epiphysis are infected. Other sites that may be involved include soft-tissues, ovaries, pancreas, scrotum, inguinal canal, chest wall, neck, and breast, but Iliac bone is a very rare site.[1]

Diagnosing hydatid disease in such uncommon sites is a real challenge.


  Case Report Top


A 61-year-old female from a rural area of Kashmir (Northern Part of India) with no previous history of tuberculosis but a history of trivial trauma 6 years back presented in the Medical Oncology Department with a huge swelling involving the right buttock and pain in the iliac bone. Fine needle aspiration cytology (FNAC) of the swelling done outside hospital was reported as having features suggestive of myxoid liposarcoma. X-ray of the pelvis showed a lytic lesion with reactive osteogenesis in the right iliac bone [Figure 1]. A computed tomography (CT) scan of the pelvis with hips was advised which showed an expansile destructive lesion in the right iliac bone with soft tissue hypodense lesions in the overlying gluteal region, likely to be collections. Contrast enhanced magnetic resonance imaging (MRI) and FNAC of the bone lesion was advised for further evaluation as it appears to be a mitotic lesion. MRI pelvis was done which showed an expansile mitotic lesion involving right iliac bone with multiple well circumscribed uniformly hyperintense lesions on T2-Weighted and T1-Weighted images with hypointense foci within it, features suggestive of liposarcoma. The patient was referred to our Surgical Oncology Department for assessment. Contrast enhanced CT chest and abdomen was advised to rule out metastasis and trucut biopsy performed. CT scan was again reported as lytic destructive lesion of (R) iliac bone with large soft tissue component with fat attenuation and hypodense lesion with fat attenuation and subtle internal enhancement in (L) kidney, metastasis. Trucut biopsy was initially inconclusive, but repeat was reported as tiny fibrocollagenous and adipose tissue bits showing ill-defined granuloma, chronic inflammation, and giant cells. Meanwhile, the patient started discharging pus from the biopsy site. Ultrasound abdomen was repeated to characterize the renal lesion, which was now reported as angiomyolipoma. Finally, it was decided to explore the lesion during which hydatid membranes were found, and formal exploration was done to remove all the cysts [Figure 2].
Figure 1: X-ray pelvis showing a lytic lesion with reactive osteogenesis in (R) iliac bone

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Figure 2: Hydatid cyst (R) gluteal region

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  Discussion Top


Hydatid cyst at unusual sites has been reported throughout the world. Musculo cystic cysts account for 0.7– 3% of total cases of hydatidosis. Spine is the most affected part of the Skelton with 50% incidence of all bones hydatidosis. Hydatid disease in the iliac bone is quite rare, even in areas where the disease is endemic is still common in the countries including the Mediterranean countries, the Middle East, South America, New Zealand, Australia, and South Asia. The liver is the most frequently involved organ (75%), followed by the lung (15%), and the remainder of the body (10%). The diagnosis of Echinococcus granulosis in an atypical location can be difficult. Histopathological evaluation of the excised specimen and FNAC usually leads to the diagnosis. Since puncture of the cyst may lead to an anaphylactic reaction due to spillage of hydatid fluid, the use of fine needle aspiration is controversial at present. Determining the ideal therapeutic approach for a musculoskeletal hydatid cyst not responding to medical treatment can be quite challenging. Conservative treatment, complete excision, and simple drainage have all been suggested as treatment options.[6] Radical surgical excision is especially indicated in cases of unilocular manifestations as only this method offers hope of permanent cure. Neelapala et al. reported a 35-year-old female patient who had lived in the United Kingdom all her life and presented with pain in the right side of pelvis in 1997.[7] She was investigated for back and hip problems. All her blood results including inflammatory markers were normal. Radiographs and bone scan were normal. She was thought to have hip dysplasia, and an MRI revealed abnormal signal changes in the right ilium suggestive of neoplasia or infection. She was referred to the oncology team for further opinion and management in May 1998. She was afebrile, and there was no history of infections and exposure to tuberculosis. She did not have any pets, and there was no history of contact with farm animals. Biopsy of the pelvic bone was carried out, and the histology showed necrotic bone with microsequestra surrounded by a foreign body reaction of histiocytes and giant cells with a thin fibrous wall. Special stains for mycobacterium and fungi were negative. Hydatid disease was considered as a diagnosis and core biopsy of the abnormal region and aspiration of the cysts lateral to the ilium were performed, and samples were sent for histology, microbiology and hydatid immunotests. Histology suggested cyst with laminar wall, reaction to nonhuman tissue and inflammation not typical of an abscess.

Hydatid disease involving other osseous locations typically manifests as different-sized areas of pure osteolysis that may become confluent and cause thinning of the cortex. Lesion growth causes breaking of the bone cortex and allows extension to the surrounding soft tissues.[8]

Sreeramulu et al. has earlier observed that intramuscular infestation may mimic a soft tissue tumor leading to inappropriate cyst rupture with the attendant risks of anaphylaxis and dissemination to other organs. He reported a case of a 34-year-old male patient with a cystic gluteal swelling turning out to be hydatid cyst on surgical exploration. Surgical excision with complementary treatment with antihelementhics is necessary to achieve complete healing.[9] Although the diagnosis is confirmed by cytology or histology, imaging modalities such as ultrasonography, CT, and MRI are more sensitive than serologic tests and in these cases, the involvement of other organs should be investigated. Treatment options of hydatid infection include drug therapy (albendazole, mebendazole, praziquantel), puncture, aspiration, injection, re-aspiration technique, and surgery. Although radicalsurgical excision offers hope of a permanent cure, the cyst should be excised as a whole without being ruptured to prevent any complications.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Hussain A, Maqbool A, Hussain S, Athar M, Shakoor A, Amin MK. Studies on prevalence and organ specificity of hydatidosis in ruminants slaughtered at Karachi and Faisalabad abattoir, Pakistan. Indian J Dairy Sci 1992;45:454-6.  Back to cited text no. 1
    
2.
Pezeshki A, Kia EB, Gholizadeh A, Koohzare A. An analysis of hydatid cyst surgeries in Tehran Milad Hospital, Iran, during 2001-2004. Pak J Med Sci 2007;23:138-40.  Back to cited text no. 2
    
3.
Lewall DB. Hydatid disease: Biology, pathology, imaging and classification. Clin Radiol 1998;53:863-74.  Back to cited text no. 3
[PUBMED]    
4.
Harandi MF, Hobbs RP, Adams PJ, Mobedi I, Morgan-Ryan UM, Thompson RC. Molecular and morphological characterization of Echinococcus granulosus of human and animal origin in Iran. Parasitology 2002;125(Pt 4):367-73.  Back to cited text no. 4
    
5.
Merkle EM, Schulte M, Vogel J, Tomczak R, Rieber A, Kern P, et al. Musculoskeletal involvement in cystic echinococcosis: Report of eight cases and review of the literature. AJR Am J Roentgenol 1997;168:1531-4.  Back to cited text no. 5
[PUBMED]    
6.
Torricelli P, Martinelli C, Biagini R, Ruggieri P, De Cristofaro R. Radiographic and computed tomographic findings in hydatid disease of bone. Skeletal Radiol 1990;19:435-9.  Back to cited text no. 6
[PUBMED]    
7.
Neelapala VS, Chandrasekar CR, Grimer RJ. Revision hip replacement for recurrent Hydatid disease of the pelvis: A case report and review of the literature. J Orthop Surg Res 2010;5:17.  Back to cited text no. 7
[PUBMED]    
8.
Braithwaite PA, Lees RF. Vertebral hydatid disease: Radiological assessment. Radiology 1981;140:763-6.  Back to cited text no. 8
[PUBMED]    
9.
Sreeramulu PN, Krishnaprasad, Girish Gowda SL. Gluteal region musculoskeletal hydatid cyst: Case report and review of literature. Indian J Surg 2010;72 Suppl 1:302-5.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2]



 

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