Osseous hydatidosis of the proximal femur: a rare diagnosis in revision total hip arthroplasty.

Musculoskeletal hydatidosis is a rare but severe disease in central Europe. This case report presents the incidental finding of an osseous hydatidosis after cementless revision total hip arthroplasty in a patient without a preoperative history of hydatidosis or any clinical symptoms. Revision total hip arthroplasty had been necessary due to a septic osteonecrosis of the femoral head 2 years after osteosynthesis of a traumatic proximal femur fracture with a sliding hip screw. The positive sample was taken out of the greater trochanter in the area of the possible former entry point for the lag screw, which was macroscopic inconspicuous. Sero-analysis could afterwards confirm the suspected diagnosis. Postoperative chemotherapy with albendazole was performed for 6 months. A full-body MRI did not reveal any further cysts. This case demonstrates a possible impact of migration on the expected pathogens in revision arthroplasty. This demonstrates that in revision arthroplasty, an infection with this parasite also has to be taken into account, if the patients come from an area endemic for hydatidosis.

Treatment challenges associated with bone echinococcosis.

OBJECTIVES: In this literature review, we concentrate on epidemiology and therapy of osseous echinococcosis, with an emphasis on the recurrence risk. METHODS: Literature review 1930-2012. RESULTS: We retrieved 200 publications based upon single case reports or case series, mostly from resource-poor settings. Among the 721 rural patients (22% females; median age 37 years), 60% of all reported cases were from the Mediterranean region and almost all patients were immune competent. Echinococcus granulosus was identified as the most frequent species. Most infections involved a single bone (602/721; 83%) and often the spine (321 cases; 45%). In eight cases (8/702; 1%), a secondary bacterial surgical site infection was reported. Surgical intervention was performed in 702 cases (97%), with single intervention in 687 episodes (95%). Complete excision of the lesion was possible in only 117 episodes (16%). Albendazole was by far the most frequently used agent in monotherapy with various dosages, while mebendazole in monotherapy was less frequent (32 cases). The median duration of antihelminthic therapy was 6 months (range 0.7-144 months). There were 124 recurrences (17%) after a median delay of 2 years (range 0.4-17 years). In multivariate analysis, the presence of visceral organ involvement increased the odds of recurrence by 5.4 (95% CI 3.1-9.4), whereas the number of surgical interventions, the duration of antihelminthic therapy or the use of hypertonic saline did not influence recurrence. CONCLUSIONS: Bone echinococcosis is a rare parasitic disease. While treatment modalities vary considerably, combined surgical and medical approaches are the standard of care with a 17% risk of recurrence.

Skeletal manifestations of hydatid disease in Serbia: demographic distribution, site involvement, radiological findings, and complications.

Although Serbia is recognized as an endemic country for echinococcosis, no information about precise incidence in humans has been available. The aim of this study was to investigate the skeletal manifestations of hydatid disease in Serbia. This retrospective study was conducted by reviewing the medical database of Institute for Pathology (Faculty of Medicine in Belgrade), a reference institution for bone pathology in Serbia. We reported a total of 41 patients with bone cystic echinococcosis (CE) during the study period. The mean age of 41 patients was 40.9±18.8 years. In 39% of patients, the fracture line was the only visible radiological sign, followed by cyst and tumefaction. The spine was the most commonly involved skeletal site (55.8%), followed by the femur (18.6%), pelvis (13.9%), humerus (7.0%), rib (2.3%), and tibia (2.3%). Pain was the symptom in 41.5% of patients, while some patients demonstrated complications such as paraplegia (22.0%), pathologic fracture (48.8%), and scoliosis (9.8%). The pathological fracture most frequently affected the spine (75.0%) followed by the femur (20.0%) and tibia (5.0%). However, 19.5% of patients didn't develop any complication or symptom. In this study, we showed that bone CE is not uncommon in Serbian population. As reported in the literature, therapy of bone CE is controversial and its results are poor. In order to improve the therapy outcome, early diagnosis, before symptoms and complications occur, can be contributive.

Cystic echinococcosis of the pelvic bone with recurrences: a case report.

Hydatid cysts commonly affect the liver and the lung. However, they rarely involve bones with vertebral column. We hereby report a case of a female patient with cystic echinococcosis of the hip bone and ilium. She presented with a long history of frequent recurrences highlighting the dismal prognosis at this rare site. Resection of the hydatid cyst from the sacroiliac region was done with allograft and autograft (rib graft) with lumbosacroiliac fixation. Follow-up of the patient at 6 months showed no detectable abnormality on radiology and the patient was doing well.

Percutaneous Treatment of Bone Hydatid Cyst.

Cystic echinococcosis (CE) may be encountered in almost every site of the body, but bone involvement is relatively rare. The vertebral column and pelvis are the most affected areas. The combined medical and surgical approach is the main treatment option in current literature. Although percutaneous treatment of CE cysts located in the liver, spleen, kidney, and soft tissues has become a serious alternative to surgery, there is no bone CE cyst treated percutaneously in the literature. This case report aims to point out that percutaneous treatment can be an effective treatment choice and alternative to surgery.

[Echinococcosis of the rib with epidural extension]. / Kyste hydatique costovertébral : pathologie bénigne ou maligne ?

Osseous hydatidosis, especially when located in the rib, is a very rare disease. Less than 50 cases of costal echinococcosis have been reported in the literature to date. The authors report a case of echinococcosis of the rib with epidural extension in a 76-year-old patient presenting paraparesis. In addition, the patient presented a large posterior and thoracic soft tissue mass measuring about 30 centimetres in diameter. A chest x-ray, a CT thoracic scan and an MRI of the dorsal spine were performed. The imaging suggested echinococcosis of the rib with epidural extension. The cyst was completely resected. Histopathology of the resected specimen confirmed the diagnosis of echinococcosis. The patient died due to postoperative complications. Accurate presurgical diagnosis allows for appropriate management and helps eradicate the disease. This also prevents the dissemination of parasites and further complications.

Pelvic echinococcosis presenting as a mass in the gluteal region: a case report.

Hydatid disease of the skeletal system is a rare entity. We present one such case of pelvic echinococcosis in a 62-year-old male who presented with the chief complaints of pain and swelling in the left gluteal region for the past twelve years. The patient was planned for and underwent en-bloc excision of the lesion. Albendazole was used pre- and post-operatively to reduce parasitic load. At the last follow-up, the patient was disease-free and able to carry out his daily activities without much difficulty. We would conclude that, although uncommon, echinococcosis should always be ruled out, especially when dealing with other indolent pathologies of the hip joint.

Osseous cystic echinococcosis: A case series study at a referral unit in Spain.

BACKGROUND: Cystic echinococcosis (CE) is present in all continents, except for the Antarctica. Characteristically, CE lesions are found in the liver and the lungs, but virtually any part of the body may be affected (the spleen, kidneys, heart, central nervous system, bones, among others). It is estimated that the incidence of bone involvement in CE is 0.5% to 4%. METHODOLOGY: A retrospective study was performed of patients with osseous CE treated at the National Reference Unit of Tropical Diseases of the Ramon y Cajal Hospital, Madrid, Spain, between 1989 and December 2017. Epidemiological, clinical, diagnostic and therapeutic data of patients with long-term follow-up were collected. MAIN FINDINGS: During the study period, of the 104 patients with CE, 27 exhibited bone involvement (26%). The bones most frequently affected were the spine, followed by the ribs, pelvis, femur, tibia and the scapula. The most common symptom was pain followed by medullar syndrome and pathologic fracture. In total, 81.5% of patients underwent surgery for osseous CE at least once. As many as 96% received albendazol either in (mostly long-term) monotherapy or in combination with praziquantel. CONCLUSIONS: The diagnosis and management of osseous CE is challenging. In many cases osseous CE should be considered a chronic disease and should be managed on a case-by-case basis. Lifelong follow-up should be performed for potential recurrence and sequels.

Cystic Echinococcosis of the Bone: A European Multicenter Study.

Cystic echinococcosis (CE) is a zoonosis caused by the larval stage of the tapeworm Echinococcus granulosus. In humans, the infection induces the formation of parasitic cysts mostly in the liver and lungs, but virtually any organ can be affected. CE of the bone is one of the rarest forms of the disease, yet it is also extremely debilitating for patients and hard to manage for clinicians. Unlike abdominal CE, there is currently no expert consensus on the management of bone CE. In this study, we conducted a survey of the clinical records of seven European referral centers for the management of patients with CE and retrieved data on the clinical management of 32 patients with a diagnosis of bone CE. Our survey confirmed that the patients endured chronic debilitating disease with a high rate of complications (84%). We also found that diagnostic approaches were highly heterogeneous. Surgery was extensively used to treat these patients, as well as albendazole, occasionally combined with praziquantel or nitaxozanide. Treatment was curative only for two patients, with one requiring amputation of the involved bone. Our survey highlights the need to conduct systematic studies on bone CE, both retrospectively and prospectively.

Clinical differentiation between dogs with benign and malignant spirocercosis.

Spirocerca lupi is a nematode infesting the canine oesophagus, where it induces the formation of a nodule that may transform into a malignant sarcoma. The current, retrospective study compared the clinical presentation, haematology, serum albumin and globulin and radiology of benign cases (n=31) and malignant cases (n=31) of spirocercosis. Dogs with spirocercosis-induced sarcoma were significantly older (6.4+/-1.91 years) than benign cases (4.93+/-2.87). In the malignant cases there were significantly (p=0.03) more sterilized females (10/31) and fewer intact males (4/31) compared to 2/31 and 13/31, respectively, in the benign cases. Hypertrophic osteopathy was observed in 38.7% of malignant cases and in none of the benign cases (p=0.0002). Common clinical signs included weight loss, regurgitation, anorexia, pyrexia (T>or=39.5 degrees ), respiratory complications and salivation but did not differ in prevalence between groups. On haematology, the malignant group had significantly (p<0.05) lower haematocrit (0.34+/-0.08 vs. 0.41+/-0.07) and higher white cell count (31.6+/-27.83 vs. 17.71+/-13.18 x 10(3)microl(-1)), mature neutrophil count (26.06+/-26.08 vs. 12.23+/-9.96 x 10(3)microl(-1)) and thrombocyte count (493.15+/-151.61 vs. 313.27+/-128.54 x 10(9)microl(-1)). There were no differences in the mean corpuscular volume and immature neutrophil count. On radiology, the mass length was not significantly different, but the height and the width of the malignant masses were significantly larger (62.59+/-15.15 mm and 73.93+/-20.94 mm) compared to the benign group (46.43+/-23.62 and 49.29+/-25.56, respectively). Spondylitis was more prevalent in the malignant group (67.86% vs. 38.46%, p=0.03). Examining secondary pulmonary changes revealed significantly higher prevalence of bronchial displacement in the malignant group (52% vs. 17%, p=0.008). Hypertrophic osteopathy appeared to be a very specific but relatively rare (poor sensitivity) marker of malignancy. Female gender, anaemia, leukocytosis, thrombocytosis, spondylitis and bronchial displacement are significantly more common in malignant cases, but appear in benign cases as well. However, if found together in a specific case, they should increase the index of suspicion for malignancy in a diagnosed spirocercosis case.

[Hydatid disease of bone: a report of 2 cases]. / Kyste hydatique primitif intraosseux: à propos de deux cas.

INTRODUCTION: The hydatid cyst of Echinococcus granulosus tends to develop in liver. The primary bone hydatidosis is rare. EXEGISIS: The authors report 2 cases of primary hydatidosis of bone in a 57 year old men and 60 year old diabetic women. The symptoms and signs were not specific. The ultrasound investigation: standard radiography and computed tomographic scan, was suggestive of the lesion. The chest radiograph and the abdominal ultrasound were normal. Serological tests for hydatid disease were positive. The two patients have surgical excision and medical treatment: Albendazole administrated for 6 months. Histologic evidence confirmed the diagnosis. The outcome was good for both patients without recurrence after 2 years. CONCLUSION: Through these 2 cases and a review of the literature, the authors analyse the epidemiological and clinical aspects of bone hydatidosis and discuss the therapeutic procedures.

A varied presentation of hydatid cyst: a report of four cases with review of literature.

Contrary to the popular perception, hydatosis does not remain restricted to certain geographical locales but is a global health concern, particularly in the rural countryside. Although liver and lung involvement account for the majority of the lesions, primary breast, bone, kidney and soft tissue hydatosis with features of cystic echinococcosis are extremely rare. We report four such rare cases of cystic echinococcosis which were diagnosed and treated in our institute.

[Primary hydatidosis of the chest wall. Report of 5 cases]. / Hydatidose osseuse primitive de la paroi thoracique. A propos de 5 cas.

BACKGROUND: Hydatidosis is an endemic affection in Tunisia. Bone echinococcosis is a relatively rare entity accounting for only 0.5-2% of all hydatid cysts in humans and chest wall is an uncommon site for the disease. AIM: Report of a new case CASE: We report about this talk 5 cases concerning 2 men and 3 women (mean of age 35.4 years) explored for parietal mass (4 cases), or chest pain (1 case). Diagnosis was suspected on radiologic findings in all cases. All patients underwent surgery. Medical treatment was associated in 2 cases. Histopathology of resected specimen confirmed diagnosis of echinococcosis. No recurrence was observed during follow-up period.

A case of pelvic bone hydatidosis--diagnosis and treatment.

INTRODUCTION: Echinococcosis is a frequent helminthiasis in Bulgaria. Hydatid cysts commonly affect the liver and the lung. Echinococcosis rarely involves bones (0.5 to 2.5%) and vertebral column is affected in 50% of the cases. PATIENTS AND METHODS: We report a case of a 67-year-old female with echinococcosis of the pelvic bone clinically manifested by pain and swelling of the right iliac region and complicated by abscess of the surrounding soft tissue. The patient underwent surgery at the Department of Orthopedics and Traumatology, University hospital "St. George", Medical University, Plovdiv. Diagnosis was based on imaging findings, serologic and histological methods. Biopsy material was investigated by standard histological methods hematoxilin-eosin (H-E),PAS and hydrargyrum impregnation after Gommori, which presented both chitin and germinative membranes of the hydatid cyst. RESULTS: The patient underwent complete resection of the bone lesion along with the surrounding soft tissue. Purulent collection of 1200 ml was evacuated. A six months follow-up of the patient revealed no pathology of the cyst and other organs. She walked unaided. No echinococcosis recurrence was observed on control examinations. CONCLUSIONS: Hydatid bone disease is usually difficult to diagnose. Its treatment includes excision of bone lesion, curettage and oral therapy. It is not always possible to fully remove all the affected bone fragments especially when cysts are located in the femoral and pelvic bones. In the reported case the hydatid cyst was fully removed by resection of the right iliac ala and the patient's mobility was preserved.

Hydatid bone disease: a case report and review of the literature.

Hydatid disease may develop in almost any part of the body and can be identified with a combination of clinical history, imaging findings, and serologic results; however, the diagnosis of bone hydatidosis is primarily based on radiographic findings. Bone hydatid disease is often asymptomatic, and its diagnosis is usually made at an advanced stage when lesions have become extensive. We present a case of a 45-year-old woman who was admitted to the University Hospital, Stara Zagora, Bulgaria complaining of pain in her left tibia. Radiographs revealed an oval cyst with a diameter of 3.5 cm, located in the diaphyseal part of the tibia. The cyst was excised, and no recurrence was observed on follow-up. Functional outcome was excellent.

Cytological diagnosis of rhinosporidiosis with skeletal involvement--a case report.

A young Hindu male presented with painful swelling of left lower thigh for 6 months. The provisional diagnosis both clinically and radiologically was osteosarcoma. FNAC and biopsy proved the lesion to be a case of rhinosporidiosis. The present case is reported due to rare incidence of skeletal rhinosporidiosis.

Infection by Henneguya sp. (Myxozoa) in the bone tissue of the gill filaments of the Amazonian catfish Hypophthalmus marginatus (Siluriformes).

This study describes aspects of the infection caused by the myxosporean genus Henneguya, which forms cysts in the bony portion of the gill filaments of Hypophthalmusmarginatus. Specimens of this catfish were acquired dead from artisanal fishermen near the town of Cametá, state of Pará, northern Brazil, between July 2011 and May 2012. They were transported in refrigerated containers to the Carlos Azevedo Research Laboratory at the Federal Rural University of Amazonia, in Belém, where analyses were performed. After confirmation of parasitism by the genus Henneguya, observation were made using optical and differential interference contrast (DIC) microscopy. The histological technique of embedment in paraffin was used. Ziehl-Neelsen staining was applied to the histological sections. Necropsy analyses on specimens of H. marginatus showed that 80% of them (40/50) had cysts of whitish coloration inside the bony portion of the gill filaments, filled with Henneguya spores. The present study found inflammatory infiltrate in the vicinity of the cysts. Furthermore, the special Ziehl-Neelsen staining technique made it possible to mark the Henneguya sp. cysts in the bone tissue and in spore isolates in the gill tissue structure. The descriptions of these histopathological findings show that this parasite is very invasive and causes damage to its host tissues.