Revision hip replacement for recurrent Hydatid disease of the pelvis: a case report and review of the literature
© Neelapala et al; licensee BioMed Central Ltd. 2010
Received: 2 December 2009
Accepted: 11 March 2010
Published: 11 March 2010
A case of a large recurrent hydatid cyst involving the right ilium and right hip treated with excision of the cyst, Total hip replacement and revision of the acetabular component with a Tripolar articulation for cyst recurrence and acetabular component loosening is presented along with a review of the relevant literature. To our knowledge there is no reported case of Total Hip replacement and revision for hydatid disease involving the bony pelvis.
Hydatid disease commonly involves liver and lung. There are many reports on Hydatid disease with the involvement of the musculoskeletal system [1–11]. Involvement of the musculoskeletal system occurs in 1% to 4% of all cases . Hydatid disease is a parasitic infection caused by tapeworm Echinococcus which inhabits in the small intestine of carnivores. The adult worms produce eggs that are released with the faeces and spread in various ways, such as through the wind, water or flies . After ingestion by the host, the embryos migrate through the intestinal wall and are either arrested in the capillary bed of the liver developing into liver cysts, or manage to penetrate into systemic circulation thus ending up in remote organs. Due to their physiologic role as capillary filters and their vast capillary volume, the liver and lung are most often affected. The brain, the muscles or the bones are the more frequently involved distant organs.
In this report, we present a case of a large recurrent hydatid cyst involving the right ilium and right hip treated with excision of the cyst and Total hip replacement which was functional for 80 months and revision with a Tripolar articulation for cyst recurrence and acetabular component loosening followed for 12 months is presented. To our knowledge there is no reported case of Total Hip replacement and revision for hydatid disease involving the bony pelvis.
A 35-year-old female patient who had lived in the United Kingdom all her life was referred with pain in the right side of pelvis in 1997. 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 a 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 May1998. 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 on 11/6/98 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. Propionibacterium was grown in culture sensitive to penicillin, amoxicillin, erythromycin and ciprofloxacin. She was advised to take of Penicillin-V 500 mg four times a day for six weeks. Nevertheless Propionibacterium bone infection was thought to be the unlikely cause of her hip pain. She was reviewed on 14/12/98 with worsening right hip pain. Examination showed limitation of right hip movements. Radiographs now showed abnormality in the right ilium with narrowing of the joint space. She was advised to have repeat blood tests including a FBC, ESR and Myeloma screen (all the blood tests were normal). MRI scan on 7/1/99 showed altered marrow signal change from inferior part of right sacro-iliac region to acetabulum and lobulated cyst in the soft tissue around the right hip region. 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 on 28/1/99 and samples were sent for histology, microbiology and hydatid immunotests. Histology suggested cyst with laminar wall, reaction to non-human tissue and inflammation not typical of an abscess. Cultures had grown coagulase negative staphylococci. ELISA test was positive for hydatid, 1:265.
Based on the MRI findings, histology report and positive serology a diagnosis of hydatid disease of the pelvis was made and she was referred to the Infectious disease unit for further management where she was started on treatment with Albendazole. Further investigations showed no evidence of liver or lung disease.
Despite the treatment with albendazole, symptoms persisted and a MRI on 24/9/99 showed progression of cysts in the right ilium and thigh with hip joint effusion. Although the cysts in the thigh region were thought not be of hydatid origin, due to the pain she was having she underwent an operation and had removal of two cysts one along the lateral side of rectus femoris and the other one deep to gluteus medius on 25/10/99. The operation was covered with Praziquental and Albendazole and she was discharged home on Albendazole. Histology again confirmed the diagnosis of hydatid disease. She was reviewed in the clinic on 6/6/00 and had an MRI on 27/6/00 as she had increasing pain in the right hip and she stopped taking albendazole as she was having hair loss. MRI was compared with the old one and was reported that the cystic lesion in the right ilium was getting bigger.
Reported sites of Hydatid disease of the musculoskeletal system.
Site of infection
Bal et al 
Bellil et al 
Merkle et al 
Iliopsoas, left adductor musculature, left femur, left gluteus medius muscle, musculature of right upper leg
Metcalf JE 
bone - humerus
Natarajan MV 
bone - femur
Torricelli et al 
bone infection with adjacent soft tissue involvement in 12 cases
Dahniya et al 
5 bone infections without soft tissue involvement, 2 primary intramuscular (lt. shoulder, rectus femoris and vastus lateralis)
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 . Hydatid disease progresses slowly and is rarely life-threatening, especially when located in the soft tissue or muscles, thus supporting a conservative therapeutic approach. However, if the Hydatid disease causes profound disabilities or mobilization problems, complete cystopericystectomy and even total joint replacement becomes an option. Radical surgical excision is especially indicated in cases of unilocular manifestations as only this method offers hope of permanent cure . Therapeutic dilemmas could arise in cases of extended disease with many muscles or muscle layers in different sites of the body which are communicating via fistulas. Communication between lesions should always be suspected and revealed, even if primary and daughter cysts are distant. Complete surgical treatment should include the primary lesion, the daughter cysts and the communicating fistulas as a whole specimen. Bony pelvis is a difficult location for radical surgical excision of the Hydatid cyst and the morbidity of a Hindquarter amputation can be considerable. Subtotal excision of the cyst and joint replacement is an acceptable option based on our case report. Subtotal excision of the Hydatid cyst of the pelvis and a hip replacement can be durable providing adequate function. Patients should be carefully monitored for cyst recurrence and component loosening. Loose components due to recurrent cysts can be successfully revised to provide good clinical outcome.
- Bal N, Kocer NE, Arpaci R, Ezer A, Kayaselcuk F: Uncommon locations of hydatid cyst. Saudi Med J. 2008, 29 (7): 1004-8.PubMedGoogle Scholar
- Bellil S, Limaiem F, Bellil K, Chelly I, Mekni A, Haouet S, Kchir N, Zitouna M: [Descriptive epidemiology of extrapulmonary hydatid cysts: a report of 265 Tunisian cases]. Tunis Med. 2009, 87 (2): 123-6. FrenchPubMedGoogle Scholar
- Dahniya MH, Hanna RM, Ashebu S, Muhtaseb SA, el-Beltagi A, Badr S, el-Saghir E: The imaging appearances of hydatid disease at some unusual sites. Br J Radiol. 2001, 74: 283-289.View ArticlePubMedGoogle Scholar
- Duncan GJ, Tooke SM: Echinococcus infestation of the biceps brachii. A case report. Clin Orthop Relat Res. 1990, 261: 247-250.PubMedGoogle Scholar
- Kural C, Ugras AA, Sungur I, Ozturk H, Erturk AH, Unsaldi T: Hydatid bone disease of the femur. Orthopedics. 2008, 31 (7): 712-10.3928/01477447-20080701-12.View ArticlePubMedGoogle Scholar
- Lewall DB: Hydatid disease: biology, pathology, imaging and classification. Clin Radiol. 1988, 53: 863-874. 10.1016/S0009-9260(98)80212-2.View ArticleGoogle Scholar
- Merkle EM, Schulte M, Vogel J, Tomczak R, Rieber A, Kern P, Goerich J, Brambs HJ, Sokiranski R: Musculoskeletal involvement in cystic echinococcosis: report of eight cases and review of the literature. AJR Am J Roentgenol. 1997, 168: 1531-1534.View ArticlePubMedGoogle Scholar
- Metcalfe JE, Grimer RJ: Tackling osseous hydatidosis using orthopaedic oncology techniques. Ann R Coll Surg Engl. 2000, 82 (4): 287-9.PubMed CentralPubMedGoogle Scholar
- Natarajan MV, Kumar AK, Sivaseelam A, Iyakutty P, Raja M, Rajagopal TS: Using a custom mega prosthesis to treat hydatidosis of bone: a report of 3 cases. J Orthop Surg (Hong Kong). 2002, 10 (2): 203-5.Google Scholar
- Siwach R, Singh R, Kadian VK, Singh Z, Jain M, Madan H, Singh S: Extensive hydatidosis of the femur and pelvis with pathological fracture: a case report. Int J Infect Dis. 2009, 13 (6): e480-2. 10.1016/j.ijid.2008.12.017. Epub 2009 Apr 1View ArticlePubMedGoogle Scholar
- 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: 435-439. 10.1007/BF00241799.View ArticlePubMedGoogle Scholar
This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.