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International Journal of Infectious Diseases (2009) 13, e480—e482 http://intl.elsevierhealth.com/journals/ijid CASE REPORTS Extensive hydatidosis of the femur and pelvis with pathological fracture: A case report Ramchander Siwach a, Roop Singh a,*, Virender Kumar Kadian a, Zile Singh a, Mantu Jain a, Harnam Madan a, Sunita Singh b a b Department of Orthopaedic Surgery, Paraplegia and Rehabilitation, Pt B.D. Sharma PGIMS, Rohtak 124001, Haryana, India Department of Pathology, Pt B.D. Sharma PGIMS, Rohtak, Haryana, India Received 9 September 2008; received in revised form 11 December 2008; accepted 26 December 2008 Corresponding Editor: William Cameron, Ottawa, Canada KEYWORDS Hydatid cyst; Pelvis; Femur Summary Background: Hydatid cysts caused by Echinococcus sp can produce tissue cysts anywhere in the body. Skeletal cystic lesions are rare, yet because of their unusual presentation diagnosis can be missed. Case report: We report a case of extensive hydatidosis of the femur with pathological fracture and involvement of the pelvis bone, without involvement of abdominal viscera, in a 51-year-old woman. The patient presented with swelling and deformity of the upper and middle third of the left thigh. The diagnosis was confirmed clinico-radiologically and the patient was treated with hindquarter amputation and chemotherapy. The patient died of sepsis and extensive bedsores one month after surgery. Conclusions: Orthopedic surgeons should be alert to this morbid condition and this disease should be suspected in cystic lesions affecting any organ of the body in pathological fractures with nonunion, especially in endemic areas of the world. Early diagnosis helps in eradication and salvage of the bone; misdiagnosis and delayed diagnosis are always fraught with the danger of amputation, recurrence, and sepsis. # 2009 International Society for Infectious Diseases. Published by Elsevier Ltd. All rights reserved. Introduction Hydatid disease (echinococcosis) is endemic in many parts of the world, particularly in sheep-raising districts of Australia, South America, Central Asia, the Middle East, and the Mediterranean countries. In cases of hydatid cysts the incidence * Corresponding author. Tel.: +91 1262 213171. E-mail address: [email protected] (R. Singh). is 59—75% in the liver, 27% in the lung, 3% in the kidney, and 1—2% in the brain.1 Bone involvement in hydatidosis occurs in less than 1% of the patients,2,3 yet it is the most debilitating form of echinococcosis in humans. Although compatible with long-term survival, the disease is not easy to eradicate and perhaps impossible to cure.2 Skeletal involvement is usually secondary to hepatic or pulmonary hydatidosis, however it may on occasion occur as the primary disease. Cases have been reported in the vertebrae, the femur, the tibia, and the pelvis.4—7 Intraosseous lesions usually began at the epiphysis 1201-9712/$36.00 # 2009 International Society for Infectious Diseases. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.ijid.2008.12.017 Extensive hydatidosis of the femur and pelvis e481 and may be polycystic or may occur, though less frequently, in the form of a solitary hydatid cyst.2 The polycystic type occurs because the cyst is unable to expand and fragments, causing diffuse spreading of the daughter cyst and scolices along the bone canals owing to bone rigidity. In both types of hydatid cyst, destruction of bone occurs through three mechanisms: compression, ischemia, and osteoclast proliferation around the compressed bone tissue causing thinning and fracture and extension into soft tissues.2 We present in this paper a case of extensive hydatidosis of the femur and pelvis with pathological fracture of the femur. Case report A 51-year-old female was referred to our tertiary center with complaints of pain, swelling and deformity of the upper and middle third of the left thigh of two-year duration. She was anemic, in poor general condition, and had a sacral bedsore. Local examination revealed flexion contracture of the left hip and knee. Diffuse swelling was present at the left mid thigh; painful abnormal mobility was present in the left mid thigh. X-ray revealed a segmental pathological fracture of the left femur with multiple osteolytic lesions with honeycomb appearance, narrow transitional zone without reactive bone in the whole of the left femur, and left hemipelvis with complete resorption of the femoral head and neck. Soft tissue shadows with multiple punctuate opacities were also noted (Figure 1). Abdominal sonography did not reveal hepatic or other visceral involvement. Ultrasound of the neck showed no mass lesion in the thyroid or parathyroid. A computed tomography scan revealed multiple lytic expansile lesions involving the left hemipelvis and sacrum, the spinal canal, and the whole of the left femur with pathological fracture. A break in the cortex was seen at multiple places in the pelvis and femur (Figure 2). Laboratory findings showed raised eosinophils with an erythrocyte sedimentation rate of 35 in the first hour and hemoglobin of 8 g%. A hemagglutination test was positive. Hindquarter amputation was performed and histopathology of the resected specimen revealed the characteristic trila- Figure 1 Plain radiograph showing intraosseous cystic lesions in the left pelvis and femoral bones, destroyed hip joint, and pathological fracture of the femur. Figure 2 Computed tomography scan showing multiple cystic lesions involving the pelvic cavity and ileum. mellar hydatid cyst wall and scolices of Echinococcus granulosus scattered amidst fragments of bone and bone marrow (Figure 3). The patient was given albendazole 10 mg/kg/day and measures were taken to improve her general condition. However, the patient died after one month due to sepsis and extensive bedsores. Discussion Hydatid disease is caused by the tapeworm Echinococcus. Species of the genus Echinococcus include Echinococcus vogeli, Echinococcus granulosus and Echinococcus multilocularis. In man and domestic animals, this parasitic infection is most commonly caused by the larval stage of Echinococcus granulosus.2,3 The adult worm resides in the intestine of the canine, which functions as a definitive host. Ingestion of ova passed with the feces from the definitive host by man and domestic animals as intermediate hosts, hatch in the small intestine and enter the blood circulation, locating in different tissues where they produce hydatid cysts.3 These cysts commonly occur in the lungs and liver, but can be found in any other organ or tissue including bones, spleen, heart, eye, brain, and genitourinary tract. Anatomoclinical changes are peculiar to localization in the bone.2 From the anatomopathologic stand Figure 3 Microscopy confirms the diagnosis and reveals classic hydatid cyst (H&E, 200). e482 point, this localization marks the torpid, insidious progression of the parasite into the bone tissue, leading to a diffuse, extensive, invasive process; so from the clinical stand point, wherever it is localized, its complete surgical eradication is rarely possible.2 Hydatid cysts of bone remain asymptomatic over a long period, and are usually detected after a pathologic fracture or secondary infection, or following the onset of compressive myelopathy in cases of vertebral lesions.4 However, a definite preoperative diagnosis without histological examination is often difficult as there are no pathognomonic signs, radiological findings may be confused with those of other tumoral lesions, and serological tests are of limited value.2 When a pathological fracture occurs in long bones due to hydatid cyst, non-union is common. The present case might have been harboring a symptomatic infestation for quite a long time and it was only the pathological fracture with non-union that brought the patient to us for treatment. Echinococcus joint disease is usually due to secondary extension from an adjacent bone. Transarticular extension from the pelvic bone to the femur or sacrum, similar to the present case, has been reported in the literature.8,9 The threat of anaphylactic reaction to the cyst fluid and spread of infection to the surrounding structure and soft tissues, sometimes by the seeding of hooklets, adds anxiety to both open biopsy and definitive surgery. Difficulty in both diagnosis and management are hallmarks of hydatidosis of bone. The disease mimics chronic osteomyelitis, fibrous dysplasia of bone osteosarcoma, benign cystic lesion of bone, brown tumor (hyperparathyroidism), and various other neoplastic lesions.10—12 It should be considered in the differential diagnosis of expansile osteolytic lesions, especially in endemic areas.12,13 A conclusive diagnosis of hydatidosis has only been reached in half of the cases preoperatively.4 The indirect hemagglutination test is more reliable in diagnosing the condition than the Casoni skin test or Weinberg’s complement fixation test.8,14 Histopathological examination is confirmatory.12 The anaphylactic nature of the cyst fluid demands strict precautions and skillful handling during biopsy and definitive surgery. Osseous hydatidosis should be treated with radical resection with a wide margin of healthy tissue. This may be difficult, but incomplete removal results in recurrence.15 Radical resection in the pelvis and hip is extremely challenging,4,8,15 and total eradication of parasitic osteitis is almost impossible.15 Reconstructive surgeries after radical excision are almost technically impossible in the pelvis and hip, although in the past, hip arthroplasty3,7 and custom-made prosthesis4,16 have been tried. Extensive surgical approaches are always accompanied by the dangers of recurrence and infections. Even patients in a good general condition may not tolerate such surgeries. Sepsis may be a cause of death.7,15 Overall, a review of the literature reveals a poor prognosis if the disease is extensive in the pelvis and femur.10,11,13,15 R. Siwach et al. The purpose of this article is to alert orthopedic surgeons of this morbid condition and to emphasize the fact that this disease should be suspected in cystic lesions affecting any organ of the body in pathological fractures with non-union, especially in endemic areas of the world. Early diagnosis helps in eradication and salvage of the bone; misdiagnosis and delayed diagnosis are always fraught with the danger of amputation, recurrence, and sepsis. Conflict of interest No grants have been received for this study. No people or organizations are associated with this study. References 1. Fagzel F, Ghanbary H. Hydatid cyst of the orbit. J Isfahan Med School 2002;20. 65. 2. Zlitini M, Ezzaoula K, Lebib H, Karray M, Kooli M, Mesteri M. Hydatid cyst of bone: diagnosis and treatment. World J Surg 2001;25:75—82. 3. Szypryt EP, Morris DL, Mulholland RC. Combined chemotherapy and surgery for hydatid cyst of bones. J Bone Joint Surg Br 1987;69:141—4. 4. Morris B, Madiwale C, Garg A, Chavahan GB. Case report: hydatid cyst of bone. Aust Radiol 2002;46:431—4. 5. Duran H, Fernandez L, Gomez-Castresana F, Lopez-Duran L. Osseous hydatidosis. J Bone Joint Surg Am 1978;60:685—90. 6. Wirbel RJ, Mues PE, Mutschler WE, Salomon-Looijen M. Hydatid disease of pelvis and femur. A case report. Acta Orthop Scand 1995;66:440—2. 7. Sapkas GS, Stathakopoulos DP, Babis GC, Tsarouchas JK. Hydatid cyst of the bones and joints. 8 cases followed for 4—16 years. Acta Orthop Scand 1998;69:89—94. 8. Meziane A, Bachechar N, Benkirane A, Ouadfel J. Hydatid cyst of pelvis: a case report. Acta Orthop Belg 1987;53:517—9. 9. Belzunegui J, Maiz O, Lopez L, Plazaolu I, Gonzalez C, Figueroa M. Hydatid disease of bone with adjacent joint involvement. A radiological follow-up of 12 years. Br J Rheumatol 1997;36:133—5. 10. Martinez AA, Herrera A, Cuenca J, Herrero L. Hydatidosis of the pelvis and hip. Int Orthop 2001;25:302—4. 11. Agarwal S, Shah A, Kadhi SK, Rooney RJ. Hydatid bone disease of the pelvis. A report of two cases and review of the literature. Clin Orthop 1992;280:251—5. 12. Ozkan H, Dogramaci Y, Kose O, Esen E, Erdem H, Komurcu M. Primary hydatid cyst of the humerus. Ann Acad Med 2008;37: 440—1. 13. Merkle EM, Schulte M, Vogel T, Tomczak R, Reiber A, Kern P, et al. Musculoskeletal involvement in cystic echinococcosis: report of eight cases and review of the literature. Am J Roentgenol 1997;168:1531—4. 14. Tuzun M. Hekimoglu. CT findings in skeletal echinococcosis. Acta Radiol 2002;43:533. 15. Herrera A, Martinez AA. Extraspinal bone hydatidosis. J Bone Joint Surg Am 2003;85:1790—4. 16. Wirbel RJ, Schulte M, Maier B, Mutschler WE. Megaprosthetic replacement of pelvis: function in 17 cases. Acta Orthop Scand 1999;70:348—52.