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.

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.

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.

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.

Eradication of advanced pelvic hydatid bone disease after limb salvage surgery - 5-year follow-up: a case report.

INTRODUCTION: Echinococcosis is produced by the larval stage of Echinococcus granulosus; it is a parasitic disease which is seen rarely in humans and has adverse outcomes. We report a case of advanced pelvic hydatid bone disease with successful limb salvage surgery. Our patient had a 5-year follow-up without recurrence which is a rarity as per the literature. Early diagnosis and prompt medical therapy are necessary for effective management whereas delayed diagnosis is always fraught with the risk of recurrence and sepsis. CASE PRESENTATION: In 2009, a 30-year-old woman, native of Karachi (Sindhi ethnicity), presented at our clinic with history of a pathological fracture 11 years earlier. Her fracture was initially misdiagnosed and fixed. Subsequently she had persistent disease that progressed with time. Following this she underwent multiple surgeries and the diagnosis of hydatid disease was made but despite multiple debridements and medical therapy she was not cured and finally she was offered a hemipelvectomy (limb sacrifice). On presentation to our hospital she was counseled regarding options of hemipelvectomy versus a limb salvage form of modified internal hemipelvectomy and wide margin resection. She opted for limb salvage. She underwent internal hemipelvectomy with wide margin resection of soft tissue and proximal femur along with postoperative albendazole therapy. She was able to walk again after a very long period. Currently she is 5-years postreconstructive surgery. She is infection free and ambulant without support. CONCLUSIONS: Hydatid bone disease is a rare entity in our part of the world but a careful history and thorough look at the initial images of our patient would have led to the suspicion of pathologic fracture and subsequent early diagnosis of this difficult problem. A second important learning point in this case was the lack of early referral to a center where this difficult problem could have been handled effectively. This could have minimized the physical, mental and financial stress to the patient and her family.

Infection by Henneguya sp. (Myxozoa) in the bone tissue of the gill filaments of the Amazonian catfish Hypophthalmus marginatus (Siluriformes) / Infecção por Henneguya sp. (Myxozoa) em tecido ósseo do filamento branquial do bagre amazônico 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.

O presente estudo descreve os aspectos da infecção causada por mixosporídio do gênero Henneguya, formando cistos na porção óssea dos filamentos branquiais de Hypophthalmus marginatus. Espécimes desse bagre foram adquiridos mortos de pescadores artesanais perto da cidade de Cametá, Estado do Pará, Brasil, entre julho de 2011 e maio de 2012. Os animais foram transportados em contêineres refrigerados até o Laboratório de Pesquisa Carlos Azevedo, na Universidade Federal Rural da Amazônia, em Belém, onde se procederam as análises. Após a constatação do parasitismo pelo gênero Henneguya, foi realizada a observação em microscópio óptico e em microscópio de contraste de interferência diferencial (DIC). Foi realizada técnica histológica de impregnação em parafina e coloração dos cortes histológicos em Ziehl-Neelsen. As análises necroscópicas dos espécimes de H. marginatus revelaram que 80% (40/50) destes apresentavam cistos esbranquiçados na porção óssea dos filamentos branquiais, repletos de esporos do gênero Henneguya. O presente estudo revelou infiltrado inflamatório nas imediações dos cistos. Além disso, a técnica especial de coloração em Ziehl-Neelsen possibilitou marcar os cistos de Henneguya sp. no tecido ósseo e de esporos isolados na estrutura de tecido branquial. As descrições desses achados histopatológicos mostram que esse parasita é muito invasivo e produz danos aos seus tecidos do hospedeiro.

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.

Recurrent primary hydatid disease of tibia / Quiste hidatídico primario recurrente de la tibia

Hydatid disease is caused by a cestode, Echinococcus. Its intermediate hosts are herbivores but humans can be accidental hosts. Hydatid disease is endemic in some parts of America, Australia, the Mediterranean region, Central Asia, and Central and Eastern Europe. The organs most frequently affected by Echinococcus are the liver and the lungs. Primary involvement of the skeleton is rare. Cases have been reported in the vertebrae, pelvis, humerus and femur. The location of hydatid cysts in the tibia is rarely described in the medical literature. We, herein, report a case of primary hydatid cyst of the tibia presenting with a pathologic fracture simulating benign bone cystic lesion. The diagnosis of hydatid bone disease was not suspected preoperatively. This case emphasizes the importance of considering hydatid disease in the differential diagnosis of cystic bone lesions, especially in individuals coming from regions where the disease is endemic.

La enfermedad hidatídica es causada por un céstodo del género equinococo. Sus huéspedes intermediarios son animales herbívoros, pero los seres humanos pueden ser huéspedes accidentales. La enfermedad hidatídica es endémica en algunas partes de América, Australia, la región mediterránea, Asia central, y Europa central y oriental. Los órganos más frecuentemente afectados por los equinococos son el hígado y los pulmones. Es raro un compromiso primario del esqueleto es rara. Se han reportado casos en las vértebras, pelvis, húmero y fémur. La localización de los quistes hidatídicos en la tibia, raramente se describe en la literatura médica. Aquí reportamos un caso de quiste hidatídico primario de la tibia acompañado de fractura patológica simulando una lesión quística benigna del hueso. El diagnóstico de la enfermedad hidatídica ósea no fue sospechado preoperatoriamente. Este caso destaca la importancia de considerar la hidatidosis en el diagnóstico diferencial de las lesiones óseas quísticas, especialmente en individuos procedentes de regiones donde la enfermedad es endémica.

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.

Multi-organ hydatidosis with extensive involvement of the hemi-pelvis and ipsilateral femur.

Bone involvement of hydatid disease is uncommon but when encountered, it presents few unique pathological features. The pattern of tissue involvement is largely different from that of visceral hydatid cyst. We describe the case of a 47 year-old man from northern India, a case of systemic hydatidosis including the liver and the right lung, presenting with an abscess like lesion in the left gluteal region with pathological fractures of the left femur. Radiographs and CT-scan images showed extensive invasion of the left hemi-pelvis and left proximal femur. Debridement of the honeycombed ilium yielded hydatid fluid, numerous small cysts and necrotic material. Multiple large devitalized and sequestrated bone pieces were recovered from the bone cavity of the affected ilium. A histopathological study of the bone sequestrums revealed the unique pattern of bone invasion by the characteristic laminated multi-layered cyst walls into areas of least resistance. Bone sequestration has not often been described or demonstrated elaborately in published studies of the past. The bone defects formed after debridement of the ilium and proximal femur were filled with bone cement along with augmentation of the femur using intra-medullary nail. The surgical technique adopted in our case although was not expected to be curative owing to the multi-system disease; it did result in significant functional improvement in the patient.

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.

What skeletons tell us. The story of human paleopathology.

Human skeletal paleopathology provides important insight regarding the antiquity of some diseases and their distribution in past human groups. The history of human skeletal paleopathology extends back more than 150 years. Rudolf Virchow published reports on the subject, and research on paleopathology has provided critical data on important topics such as the origin of syphilis. With the development of powerful new research tools, human paleopathology will continue to be a source of data on the development of disease and its effect on human biological and cultural development.

Hidatidosis ósea: Reporte de 5 casos y revisión de la literatura / Bone hydatidosis: Report of five patients

Bone location of hydatid cysts occurs in 0.5 to 3 percent of all cases of hydatidosis. The most common bones involved are spine, long bones and pelvis. We report five patients with bone hydatidosis. A 24-year-old male consulting for paraparesis and numbness of lower limbs; CAT scan showed hydatid cysts located in D2, D3 and D4 vertebral bodies. A 47-year-old male consulting for a fracture of the femur; X rays disclosed multiple hydatid cysts in the femur. A 13-year-old female consulting for claudication of the right lower limb; X ray examination showed a hydatid cyst in the iliac bone. A 21-year-old women presenting with weakness of the lower limbs and voiding problems; CAT scan showed a vertebral hydatidosis and spinal cord compression. A 67-year-old female presenting with weakness of the lower limb and loss of sphincter control; CAT scan showed hydatid cysts in D4 vertebral body.

Pelvic bone hydatidosis.

Bone hydatid disease is a rare pathology, characterized by its long clinical latency, the absence of radiological specificity as well as its challenging surgical treatment. We report an observation of pelvic bone hydatidosis in a 28-year-old patient, appearing with pain and stiffness of the hip as well as a degenerative aspect on the coxofemoral joint on X-ray. The treatment consisted of an extensive hemipelvectomy with femoro pelvic arthrodesis, completed by medical treatment. The functional result was excellent in spite of a break in the fixation material, which allowed a certain mobility at the proximal end of the femur. Hydatid disease of bone is infiltrating,diffuse, slow, and progressive, all characteristics explaining the often-delayed diagnosis. Medical imaging provides accurate analysis for planning a broad surgical resection. The quality of surgical resection is determined according to the risk of recurrence. Pelvic locations are particularly difficult to treat. After an enlarged and difficult surgical resection, reconstruction remains aleatory and poses many technical problems.

[Bone hydatidosis: report of five patients and review of the literature]. / Hidatidosis ósea: Reporte de 5 casos y revisión de la literatura.

Bone location of hydatid cysts occurs in 0.5 to 3% of all cases of hydatidosis. The most common bones involved are spine, long bones and pelvis. We report five patients with bone hydatidosis. A 24-year-old male consulting for paraparesis and numbness of lower limbs; CAT scan showed hydatid cysts located in D2, D3 and D4 vertebral bodies. A 47-year-old male consulting for a fracture of the femur; X rays disclosed multiple hydatid cysts in the femur. A 13-year-old female consulting for claudication of the right lower limb; X ray examination showed a hydatid cyst in the iliac bone. A 21-year-old women presenting with weakness of the lower limbs and voiding problems; CAT scan showed a vertebral hydatidosis and spinal cord compression. A 67-year-old female presenting with weakness of the lower limb and loss of sphincter control; CAT scan showed hydatid cysts in D4 vertebral body.