Case Report

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Title:
Primary Iliac Bone Hydatid Disease: A Case Report

Author(s):
Mohammad Shabanpour Haghighi MD 1, Abolfazl Bagherifard MD 2, Mohamad Qoreishy MD 3, Tahmineh Mokhtari PhD 4*

1 Department of Orthopedic Surgery, Iran University of Medical Sciences, Tehran, Iran
2 Department of Orthopedic Surgery, Iran University of Medical Sciences, Tehran, Iran.
3 Department of Orthopedic Surgery, Shahid Beheshti University of Medical Sciences, Tehran, Iran
4 Department of Anatomy,Tehran University of Medical Sciences, Tehran, Iran

* Corresponding Author

Vol 2, Num 2, April 2015

 

   

Abstract

Hydatidosis is a parasitic caused by the larva of the tape worm Echinococcosis granulosus. In this study, an old man with progressive hydatidosis in iliac bone was reported. The X-ray radiography showed multiple cystic lesions. The Computed tomography scan results showed a multiple erosive lesions. The biopsy was performed and the result of biopsy confirmed the diagnosis of hydrated cyst in this patient. Now he is put on a medical treatment with mebendazole.

Keyword:Hydatid cyst, Hydatidosis, Pelvis, Orhtopaedics, A.5, A.14

 

   

Introduction

Echinococcosis (hydatidosis, hydrated cyst) is a parasitic and zoonotic disease affects human and other mammals and its common form in human caused by the larva of the tape worm Echinococcosis granulosus.[1]

Hydatidosis commonly involves lung (18-35%) and liver (55-70%).[2-4] The musculoskeletal involvement was reported in several studies.[5] The incidence of primary bone involvement is rare and is about 0.5 to 2.5 %.[6] However, this infection usually occurs at more vascularized sites, such as the vertebrae [7-9], long bone [4,10], and pelvic.[11-15] In these cases, larva reached the bone, penetrates the spongy tissue of bone and matures in that area and can damage the tissue like a tumor.[16] In bone involvement, the prognosis of this disease is poor. Even, these cases require to extensive medical treatments and surgical procedures.[14]

The parasitology and pathology of the parasite in patient with hydatidosis should be understood for better medical and surgical treatments. In addition, the surgeons should be fully aware of the management limitations and possible complications of these patients.[10,17,18]

In this study, an old man with progressive hydatidosis in iliac bone was reported. The biopsy was performed and the result of biopsy confirmed the diagnosis of hydrated cyst in this patient.

Case Report

In the present study, a 51 year old Afghan man living in Iran as a case of Hydatid disease in left iliac bone was evaluated. He complained from a painful hip joint after motor accident about two years ago and this pain had progressively increased from six month ago. This pain was aggravated by walking and he referred to more evaluations two month ago. Loss of appetite or history of tuberculosis was not explained. In the physical exams, there was no associated fever and in local examination showed a cystic, non-tender swelling of almost 10-cm in diameter in the right flank area. Patient had antalgic gait.

In abdominal exam, no swelling or mass could be felt and spinal tenderness or deformity weren't observed. In addition, physical examination of the left hip joint revealed limited internal rotation with mild pain.

For more evaluations plain radiography were performed for this patient. On the radiography of the pelvis, multiple cystic lesions were observed throughout the left iliac bone (Figure 1).

The sonography of the liver, spleen, both kidneys, and the urinary bladder was normal increased perfusion, blood pool and bone up-take in the left iliac bone were observed in the bone scanning this patient (Figure 2). No other abnormalities were detected.

In the laboratory tests, white blood cell count was 12,100 cells/mm3 (with 73% polymorphonuclear cells), the erythrocyte sedimentation rate (after the first hour) was 117 mm and C-reactive protein was 3+. In addition, the indirect hemagglutination and Casoni tests were normal.

Computed tomography (CT) of liver and lungs were normal. Then, CT scan of the pelvis was performed (Figure 3) and it revealed a large hypodense cystic lesion all over the left iliac bone which involved the soft tissue. Its view was multifocal. The results of the first CT scan suggested malignancy, metastatic lesions or osteomyelitis.

In the laboratory tests, white blood cell count was 12,100 cells/mm3 (with 73% polymorphonuclear cells), the erythrocyte sedimentation rate (after the first hour) was 117 mm and C-reactive protein was 3+. In addition, the indirect hemagglutination and Casoni tests were normal.

After paraclinical evaluations, an open biopsy surgery was performed and the sample was prepared for pathological studies. The pathology results revealed the hydrated cyst in this case (Figure 4). He explained that had a discharge history 12 days after biopsy and beginning the treatment. After this condition, his pain was relieved. Now he is on a medical treatment with mebendazole.

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    Figure 1: The X-ray scan of Pelvis in patien with primary iliac bone hydatid disease. (White arrow: multiple cystic lesions)
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    Figure 2: Bone scanning of patient with primary iliac bone hydatid disease
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    Figure 3: Pathologic results of biopsy. laminated membrane of hydatid cyst (black stars). Cancellous bone (gray stars).
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    Discussion

    The most common areas for infestation of hydatidosis are in South America, Mediterranean, Central Asia, the Middle East and North, Australia and East Africa.[4,19] Hydatidosis caused by the larval stage of the tape worm Echinococcosis granulosus which affects human and other mammals and is a cosmopolitan parasitic zoonotic disease.[1,20] Human is considered as accidental and intermediate host in the cycle of Echinococcus granulosus , in which the liver and lungs have been predominantly involved.[2-4,21] The affection of bone is rare.[6,22] However the bone involvement in sites of vertebra, femur and pelvis is common [6] and the cystic echinoccocosi lesions in bone may be single or multiple and similar to a bone neoplasia.[16]

    In man, infection to the parasite is usually acquired in childhood about five to 20 years before the diagnosis and the symptoms of hydatidosis present several years after exposure.

    The symptoms of pain and tenderness at the affected sites appear in the advanced stages and recorded rarely with fractures.[6]

    In the present study a 51 year old Afghan man living in Iran as a case of Hydatid disease in left iliac bone was evaluated. Pathological studies confirmed the present of hydrated cyst in the iliac bone.

    Martínez et al (2001) reported eight cases (five men and three women) with primary pelvis and hip hydatidosis. They treated all patients by curettage and albendazole therapy. In three cases, in which only the ilium was involved, the outcome was satisfactory. The remaining patients required several debridement procedures in combination with chemotherapy and two developed chronic lesions. they conclude that treatment for this condition is difficult and when the osseous involvement is extensive the prognosis is poor.[23]

    Sometimes, primary pelvic hydatid cyst can present with abdominal symptoms. These complications are related to compression of the rectum and urinary tract.[14] In some cases, primary pelvic hydatid cyst can cause sciatica and foot drop.[24]

    Also, in these cases, deaths have been reported. The reasons of death in these cases were anaphylactic shock (because of spillage during excision) or biopsy (because of mistake in diagnosis of a retroperitoneal tumor).[25]

    In the present study, radiography and CT were performed for primary diagnosis. In endemic areas, hydatid disease should always be in the differential finding list of any growing mass in the body because of the variety of its presentation. radiography, ultrasonography, computed tomography, magnetic resonance imaging and immunologic tests are Valuable techniques for primary diagnosis of hydatidosis.[26]

    In this study, treatment of patient was performed by mebendazole.In the present study, serous discharge has been reported 12 days after biopsy and beginning the treatment and patient has pain relief after this discharge.

    The choice treatment of osseous hydatidosis is complete surgical excision. Isolated medical therapy with mebendazole or albendazole is not adequate for controlling the process and can be used after the complete excision of cyst.[17] However, in pelvic and hip radical surgery is impossible. So, it was suggested to treat the patients with mebendazole or albendazole for control the hydatidosis, and use the complete excision in frequent recurrence. Also, long term follow-up is strongly recommended.[27,28]

    In the present study, a 51 case with Hydatid disease in left iliac bone was evaluated. It can be concluded that the masses with characteristics of hydatid disease should be considered in endemic regions.



    Mohammad Shabanpour Haghighi MD
    Resident of Orthopaedic Surgery, Iran University of Medical Sciences, Tehran, Iran

       

    Abolfazl Bagherifard MD
    Assistant Professor, Orthopaedic Surgeon, Iran University of Medical Sciences, Tehran, Iran

       

    Mohammad Qoreishi MD
    Orthopaedic surgeon, Assistant professor, Shahid Beheshti Medical University, Tehran, Iran
    qoreishy@gmail.com

       

    Tahmineh Mokhtari PhD
    Student, Department of Anatomy, Medical school, Tehran University of Medical Sciences, Tehran, Iran
    Corresponding Author
    mokhtari.tmn@gmail.com

     
     

    Acknowledgements:
    None declared.

     
     

    Financial disclosure:
    None declared.

     
     

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