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Nondestructive three-dimensional (3D) micromorphological imaging technique is essential for hepatic alveolar echinococcosis (HAE) disease to determine its damage level and early diagnosis, assess relative drug therapy and optimize treatment strategies. However, the existing morphological researches of HAE mainly depend on the conventional CT, MRI, or ultrasound in hospitals, unfortunately confronting with the common limit of imaging resolution and sensitivity, especially for tiny or early HAE lesions. Now we presented a phase-retrieval-based synchrotron X-ray phase computed tomography (PR-XPCT) with striking contrast-to-noise ratio and high-density resolution to visualize the HAE nondestructive 3D structures and quantitatively segment different pathological characteristics of HAE lesions without staining process at the micrometer scale. Our experimental results of the HAE rat models at early and developed pathological stages and albendazole liposome (L-ABZ) therapeutic feeding models successfully exhibited the different HAE pathological 3D morphological and microstructural characteristics with excellent contrast and high resolution, demonstrating its availability and superiority. Moreover, we achieved the quantitative statistics and analysis of the pathological changes of HAE lesions at different stages and L-ABZ therapeutic evaluation, helpful to understanding the development and drug treatment of HAE disease. The PR-XPCT-based quantitative segmentation and characterization has a great potential in detection and analysis of soft tissue pathological changes, such as different tumors., (© 2018 Wiley Periodicals, Inc.)

Ex vivo liver resection combined with autotransplantation is a recently introduced approach to cure end-stage hepatic alveolar echinococcosis (HAE), which is considered unresectable by conventional radical resection due to echinococcal dissemination into the crucial intrahepatic conduits and adjacent structures. This article aims discuss the manipulation details and propose reasonable indications for this promising technique. All patients successfully underwent liver autotransplantation with no intraoperative mortality. The median weight of the autografts was 636 g (360-1300 g), the median operation time was 12.5 hours (9.4-19.5 hours), and the median anhepatic phase was 309 minutes (180- 460 minutes). Intraoperative blood loss averaged 1800 mL (1200-6000 mL). Postoperative complications occurred in 13 patients during hospitalization; 5 patients experienced postoperative complications classified as Clavien-Dindo grade III or higher, and 2 patients died of intraabdominal bleeding and acute cerebral hemorrhage, respectively. Twenty-nine patients were followed for a median of 14.0 months (3-42 months), and no HAE recurrence was detected. The technique requires neither an organ donor nor any postoperative immunosuppressant, and the success of the treatment relies on meticulous preoperative assessments and precise surgical manipulation., (© 2017 The Authors. American Journal of Transplantation published by Wiley Periodicals, Inc. on behalf of The American Society of Transplantation and the American Society of Transplant Surgeons.)

Objective: To investigate the distribution characteristics of gallbladder diseases in children with hepatic alveolar echinococcosis, and to analyze the related factors for hepatic alveolar echinococcosis and gallbladder diseases., Methods: The clinical data of hepatic alveolar echinococcosis patients under 18 years were collected in Qinghai Provincial People's Hospital-from January 2012 to December 2017, and the gallbladder-related complications in the surgically treated patients with hepatic al-veolar echinococcosis were analyzed., Results: The clinical data of 51 child patients with hepatic alveolar echinococcosis were collected and analyzed. According to the PNM classification criteria of WHO, P1, P2 and P3 in the 51 patients accounted for 37.25% (19/51), 41.18% (21/51) and 19.60% (10/51), respectively. According to the diagnostic criteria (WS 257-2006) of China for echinococcosis, the patients of infiltration type, calcification type and liquefaction cavitary type accounted for 66.67% (34/51), 21.57% (11/51) and 11.76% (6/51), respectively. Among the 51 patients, 78.43% (40/51) of the patients had clinical symptoms of biliary tract disease, and 58.82% (30/51) had gallbladder-related complications. The operations were performed on 40 patients, and 77.50% (31/40) of them had the postoperative complications. The results of binary multivariate logistic regression analysis showed that the clinical image classification, the liver segment distribution, size, location and number of lesions of hepatic alveolar echinococcosis were important correlative factors for the occurrence of gallbladder complications in hepatic alveolar echinococcosispatients., Conclusions: The incidence of complications related to gallbladder diseases is higher in the children with hepatic alveolar echinococcosis in Qinghai Province, and it has great influence on the operation mode and the occurrence and prognosis of postoperative complications. The early diagnosis and treatment of alveolar echinococcosis in children is particularly important.

Surgical treatment of liver cystic echinococcosis (LCE) could be conservative or radical. Radical surgery includes liver resection, but usually are minor hepatectomy in favourable segments. Experience in major hepatectomy (MH) for LCE is limited., Methods: Retrospective study., Period: January 2007-December 2014., Inclusion Criteria: liver infestation with Echinococcus granulosus causing active or complicated cysts. Epidemiological, clinical, radiological and surgical data were studied., Results: 145 patients underwent surgery for LCE. MH was performed in 49 patients (34%) with 81 cysts. 51% of patients were women. Mean age: 56 years. Sixteen patients (32.7%) had recurrent disease. The mean diameter cyst was 9.9 cm. The MH performed were right hepatectomy (n = 15), left hepatectomy (6) and others (n = 28). The reason for MH was occupation of the entire lobe (14), severe vascular or biliary involvement (17), or a combination of the two (18). Major morbidity (Clavien III-V) was 26%. Mortality was 2%. Mean hospital stay: 15.3 days. At follow-up (mean: 31 months) the rate of liver recurrence after MH was 0%., Conclusions: MH is feasible in LCE, with a major morbidity rate of (26%), and zero recurrence. Indications of MH are occupation of an entire lobe, extreme biliary or vascular involvement or recurrent cysts., (Copyright © 2018. Published by Elsevier Ltd.)

To evaluate the relationship between cyst activity and calcification degree in cystic echinococcosis (CE) in humans, 99 hepatic cysts at successive stages of involution, surgically excised from 72 Sardinian patients, have been analyzed. Cysts were classified into 4 groups according to calcification extent: CALC 0 (no calcification); CALC 1 (scattered punctate calcifications); CALC 2 (large coarse segmental/partial calcifications); CALC 3 (complete or nearly complete circumferential ring of calcification up to thick wall of osseous consistency/calcified content of cyst). In addition the possible correlation with antibody response has been explored analyzing IgG1, IgG4 and IgE produced against somatic PSCAg. Results showed that calcification is not restricted to the inactive WHO cyst types CE4 and CE5, but occurs to a varying extent in all morphotypes of metacestode, from active classic unilocular or multivesicular cysts to the more complicated and highly degenerate stages, where cyst wall appears massively calcified. Prevalence of calcification increases with progression of cyst degenerative process, but is not synonymous with parasite inactivity and can be misleading as signs of calcification may coexist with still metabolically active cysts. On the contrary, detection of entirely firmly solidified content seems a reliable indication of cyst inactivity. IgG4 is the dominant isotype associated particularly with the evolutive phase. Positive rates and OD levels, higher in active vs inactive stages, are stable or increase slightly in weakly and moderately calcified cysts (CALC 1/CALC 2), compared to non-calcified ones (CALC 0), strongly decreasing in highly calcified forms (CALC 3). In conclusion, evaluation of calcification extent may be pertinent for staging CE, and immunological tests, particularly for IgG4, and IgE may help to better define cyst activity., (Copyright © 2018 Elsevier B.V. All rights reserved.)

The main goal of the research was to study echinococcosis liver cyst laparoscopic treatment's effectiveness. The retrospective analysis of liver echinococcosis cyst surgical treatment in the period from 2003 to 2013 years was conducted. 348 patients underwent surgical treatment, among them 283 patients - laparoscopic procedure, 65 - open surgery. Medial age was 42,3±7,9 years. Female - 214, male - 134. 249 patients underwent laparoscopic partial (conservative) cystectomy, 34 - laparoscopic radical cystectomy with liver resection, 3 (1%) - conversion. 47 patients underwent partial (conservative) cystectomy using open approach. 18 - radical cystectomy using either typical or atypical hemihepatectomy. A long term analysis of 226 (79,8%) patients in a period from 6 month to 7 years was conducted. Comparison of the results revealed the advantage of laparoscopic operations. It was manifested in less blood loss, duration of the procedure, pain syndrome, and a smaller number of bed-days. Infections of the residual cavity after open surgery were twice as much comparing to ones after laparoscopic surgery. Complications after open surgery were significantly higher than after laparoscopic procedures due to postoperative wound infections. After open surgery frequency of echinococcosis recurrence was higher than after laparoscopic one (8,5% vs 2,5%) (р<0,05). Nowadays it is possible to say that treatment of choice of noncomplicated liver echinococceal cysts is laparoscopic one. Correctly made laparoscopical procedure has a good long term results, few postoperative compications, less frequent recurrences and the excellent cosmetic effect. From the principles of radical surgical treatment's point of view laparoscopic approach is the same as an open surgery.

Liver hydatid cyst represents one of the most frequent localizations of Echinococcus granulosus tapeworm in humans. The disease progresses symptom-free in most patients, due to a slow growth rhythm of the cyst. When it reaches large sizes, the hydatid cyst causes discomfort or pain in the liver bed, low appetite, phenomena of hepatitis or cholangitis. The patients are most often diagnosed with liver hydatid cyst after imagistic investigations (radiological or ultrasound) for symptoms that, most commonly, are not related to a hydatid cyst. We present the case of an 11-year-old girl, with no pathological history, who presented forme fruste symptoms, not acknowledged by her parents, and deceased after an anaphylactic shock caused by the rupture of a liver hydatid cyst.

Background: Hepatic echinococcosis (HE) is a zoonosis and depicts a rare but potentially lethal disease caused by larval infestation of E. multilocularis (alveolar echinococcosis, AE) and E. granulosus (cystic echinocococcosis, CE). In many countries, HE is a critical public health problem. Clinically, HE patients initially are often asymptomatic for years. Depending on the echinococcal manifestations patients can later develop unspecific symptoms as fatigue, abdominal pain and may present with elevated transaminases, jaundice and hepatomegaly. The combination of grey scale ultrasound and serological tests has been the gold standard for the screening and diagnosis of HE. Besides MRI, CT and FDG-PET scans, safe and directly accessible contrast-enhanced ultrasound (CEUS) may easily help to indirectly describe perilesional inflammation. Upon diagnosis of HE, an appropriate therapeutical strategy should be evaluated in a multidisciplinary way., Objective: The aim of the present retrospective monocenter study is to assess the diagnostic performance of CEUS examination in the evaluation of hepatic echinococcal manifestation by comparison with CT, MRI, FDG-PET scans and histopathology., Methods: Out of 36 patients with echinococcal disease (16 patients with E. multilocularis infection, 12 patients with E. granulosus infection and 8 patient with unspecified Echinococcus infection) 8 HE patients (4 patients with E. multilocularis, 2 patients with E. granulosus and 2 patients with unspecified echinococcal liver disease) were included in this study on whom CEUS was performed between 2008-2016. The applied contrast agent was a second-generation blood pool agent (SonoVue ®, Bracco, Milan, Italy). CEUS examinations were performed and interpreted by a single experienced radiologist with more than 15 years of experience., Results: All patients were examined without occurrence of any side effects. In all 4 AE patients, contrast enhancement could be detected by means of CEUS and was confirmed by MRI or PET-CT scan. In the remaining 4 patients (CE and unspecified echinococcosis), doppler ultrasonography, CEUS and corresponding CT or MRI scans could not detect any hypervascularization of the lesions of interest. The histopathological analysis did not reveal any viable parasite material. CEUS showed a sensitivity of 100% and a specificity of 100% compared to MRI, CT or FDG-PET-CT., Conclusions: CEUS depicts a safe method for the evaluation of echinococcal liver disease. In addition to serological tests and grey scale ultrasound, CEUS imaging could be integrated as an easily accessible tool helping to describe hypervascularization as a sonomorphological correlate for active perilesional inflammation of echinococcal manifestations. CEUS may further help to differentiate between CE and AE and also to evaluate treatment outcome.

Congenital or acquired obstruction of the inferior vena cava may lead to the development of collateral pathways with tributary vein to portal system. We present a patient with intrahepatic cavoportal collateral pathway due to hepatic hydatid cyst.

Rationale: Alveolar echinococcosis (AE) is a rare but highly malignant form of echinococcosis caused by Echinococcus multilocularis. There have been very few reports on multiple-organ AE, especially AE in bones. Here we report 3 rare cases of disseminated multiple-organ AE from western China and its neighboring areas., Patient Concerns: Patient 1 had back and left hip pain, headache, and weakness in left lower limb, often with minor epilepsy and fluctuation of blood pressure. Lower limbs Babinski sign was positive and muscular tension was above normal range. The second patient had pain in lower limbs and chest discomfort without fever, cough, sputum, chest tightness, or hemoptysis. Patient 3 had masses and pain in the back side of his right shoulder., Diagnoses: The patients had been treated for AE multiple times and were positive for serum hydatid antigens. They were diagnosed as multiorgan AE involving liver, spinal cord, and many other organs., Interventions: The patients had undergone surgeries to decompress the spinal cord, remove lesions from tissues as required, and were put on albendazole for at least 2 years., Outcomes: The patients responded well and AE recurrence has not occurred., Lessons: All 3 cases experienced multiple recurrences of AE due to missed diagnosis, misdiagnosis, or inappropriate treatment, which resulted in metastatic multiorgan AE. These cases demonstrated the need for more policy attention to battle AE endemic in western China.

The local immune mechanisms responsible for either self-healing or sustained chronic infection are not clear, in the development of E. multilocularis larvae. Here, we developed a suitable experimental model that mimics naturally infected livers, according to the parasite load. We demonstrated that local cellular immunity and fibrogenesis are actually protective and fully able to limit metacestode growth in the liver of low or medium dose-infected mice (LDG or MDG), or even to clear it, while impairment of cellular immunity is followed by a more rapid and severe course of the disease in high dose-infected mice (HDG). And recruitment and/ or proliferation of memory T cells (including CD4 Tem, CD8 Tcm and CD8 Tem) and imbalance of T1/T2/T17/Treg-type T cells in liver were not only associated with clearance of the parasite infection in LDG, but also with increased hepatic injury in HDG; in particular the dual role of CD8 T cells depending on the parasite load and the various stages of metacestode growth. Besides, we first demonstrate the association between LAG3- or 2B4-expressing T cells exhaustion and HD inocula in late stages. Our quantitative experimental model appears fully appropriate to study immunomodulation as a therapeutic strategy for patients with Alveolar Echinococcosis.

Propagation-based phase-contrast computed micro-tomography (PPCT) dominates the non-destructive, three-dimensional inner-structure measurement in synchrotron-based biomedical research due to its simple experimental setup. To quantitatively visualize tiny density variations in soft tissues and organs closely related to early pathological morphology, an experimental study of synchrotron-based X-ray PPCT combined with generalized phase and attenuation duality (PAD) phase retrieval was implemented with the hepatic echinococcosis (HE) infection rat model at different stages. We quantitatively analyzed and evaluated the different pathological characterizations of hepatic echinococcosis during the development of this disease via our PAD-based PPCT and especially provided evidence that hepatic alveolar echinococcosis invades the liver tissue and spreads through blood flow systems with abundant blood supply in the early stage. Additionally, the infiltration of tiny vesicles in HE lesions can be clearly observed by our PAD-PPCT technique due to the striking contrast-to-noise ratio (CNR) and mass density resolution, which cannot be found by the medical imaging techniques, such as magnetic resonance imaging (MRI), computed tomography (CT), and ultrasound, in hospitals. The results demonstrated that our PAD-PPCT technique has a great potential for indicating the subtle structural information of pathological changes in soft biomedical specimens, especially helpful for the research of early micro-morphology of diseases.

Background: Alveolar echinococcosis (AE) is caused by the metacestode stage of Echinococcus multilocularis. The inflammatory response to this infection is influenced by the interaction of the parasite with the host. We aimed to analyze human liver lesions infected with Echinococcus multilocularis and the changes of the cellular infiltrates during albendazole (ABZ) treatment., Methodology/principal Findings: We analyzed liver tissue samples from 8 untreated patients, 5 patients treated with two daily doses of 400 mg ABZ for up to two months and 7 patients treated for more than two months with the same ABZ therapy. A broad panel of monoclonal antibodies was used to characterize the lesion by immunohistochemistry. A change in the cellular infiltrate was observed between the different chemotherapy times. During the initial phases of treatment an increase in CD15+ granulocytes and CD68+ histocytes as well as in small particles of Echinococcus multilocularis (spems) was observed in the tissue surrounding the metacestode. Furthermore, we observed an increase in CD4+ T cells, CD20+ B cells and CD38+ plasma cells during a longer duration of treatment., Conclusions/significance: ABZ treatment of AE leads to morphological changes characterized by an initial, predominantly acute, inflammatory response which is gradually replaced by a response of the adaptive immune system.

Echinococcosis is a serious parasitic disease that ends lethally in 95% of untreated infected patients. It was first diagnosed in Slovakia in the year 2000. It is caused by the larval stage of a tapeworm belonging to the genus Echinococcus, which was assigned to the group "A" of zoonoses in the year 2004. The number of new infections is rising because of increasing percentage of infected red foxes (Vulpes vulpes). Early and accurate diagnosis of infections with this parasite is essential for proper initiation of adequate therapy. Thanks to professional multidisciplinary efforts and new laboratory procedures, the number of correctly diagnosed cases has increased. Antimicrobial therapeutic approaches can lead to improved quality of life and better prognosis even if radical surgery is refused by the patient.

The postoperative period of cystic echinococcosis was studied in 13 children. Demographic, epidemiological, clinical diagnosis, treatment, number location, and development of cysts and serologic data were analyzed. Age of children at diagnosis range 5 to 17 years. All patients with cystic echinococcosis had abdominal cysts. The liver was the main organ involved in ten patients (76,9%) - they had cysts located in the liver, two patients (15,4%) had lung cyst, one patient had concomitant lung and liver cysts. Twelve patients had single cysts and one had more than one abdominal cysts. Surgical treatment was performed in 23,1% cases. Ultrasound studies (US) were performed during the monitoring period. Evaluation of cysts was assessed by monitoring US changes. Positive dynamics was revealed in all patients; relapse of the disease was not noticed. Proceeding from the fact that in all patients echoarchitectonics of the hepatic tissue was lumped with a non-uniform structure and uneven ultrasound distribution, it is assumed that these changes are indicative of the development of connective tissue in the liver.

Background/aims: Cystobiliary communication (CBF) with hepatic hydatid disease is responsible for postoperative bile leakage after surgical management. This study aims to detect various predictors of CBF and its outcome after surgical management., Materials and Methods: This is a retrospective, cohort study of all patients who underwent surgical management for hydatid disease of the liver. Patient data were recorded on an internal web-based registry system supplemented by paper records. Patients were classified into two groups according to the presence of CBF: group (A) patients with CBF and group (B) patients without CBF., Results: There were 123 patients with a hepatic hydatid cyst with a mean age of 39.92±14.59 years. Patients were classified into group (A), 26 patients (21.1%) with CBF, and group (B), 97 patients (78.9%) without CBF. The age group (p=0.04), presence of jaundice (p=0.001), serum glutamic-pyruvic transaminase (SGPT) (p=0.001), cyst size (p=0.0001), and cyst size group (>10 cm) (p=0.0001) were associated with CBF. That cyst size was the only independent predictor of the occurrence of CBF. Intraoperative suturing and the T tube led to complete healing of CBF, and postoperative endoscopic retrograde cholangio-pancreatography (ERCP) and tubal drainage led to a rapid reduction in the bile output and the healing of the fistulas after 9±2.6 days., Conclusion: That cyst size was the only independent predictor for the occurrence of CBF. Management is related to the size of the fistula, the site of the cyst, and the experience of the hepatobiliary surgeon. ERCP is an important option for the management of CBF.

Serology is important for the diagnosis and follow-up of human alveolar echinococcosis (AE). However, patient conditions are highly variable among those with AE, and antibody responses in serological follow-up have not been well-defined. We recently described a new AE rat model established by implantation of small AE tissue into a single arbitrary location in the liver; no metastasis and dissemination were observed. In the present study, we examined the serological characteristics in our rat model before and after surgical treatment. The results showed that antibody responses against crude antigens were increased at one month after transplantation and similar to those of other model animals. For the antigen Em18, antibody responses were slower in our rat model than in other animal models. After surgical resection, changes in antibody responses against Em18 were similar to those observed in human patients with AE. Because of the slow growth of lesions, establishment of a single hepatic lesion and patterns of antibody responses, our rat model may be useful for clarifying follow-up serodiagnoses in human AE and determining the mechanisms of multi-organ involvement by primary infection with oncospheres rather than metastasis.

Alveolar echinococcosis (AE) caused by Echinococcus multilocularis metacestode is frequently associated with deleterious zoonotic helminthiasis. The growth patterns and morphological features of AE, such as invasion of the liver parenchyme and multiplication into multivesiculated masses, are similar to those of malignant tumors. AE has been increasingly detected in several regions of Europe, North America, Central Asia, and northwestern China. An isoform of E. multilocularis antigen B3 (EmAgB3) shows a specific immunoreactivity against patient sera of active-stage AE, suggesting that EmAgB3 might play important roles during adaptation of the parasite to hosts. However, expression patterns and biochemical properties of EmAgB3 remained elusive. The protein profile and nature of component proteins of E. multilocularis hydatid fluid (EmHF) have never been addressed. In this study, we conducted proteome analysis of EmHF of AE cysts harvested from immunocompetent mice. We observed the molecular and biochemical properties of EmAgB3, including differential transcription patterns of paralogous genes, macromolecular protein status by self-assembly, distinct oligomeric states according to individual anatomical compartments of the worm, and hydrophobic ligand-binding protein activity. We also demonstrated tissue expression patterns of EmAgB3 transcript and protein. EmAgB3 might participate in immune response and recruitment of essential host lipids at the host-parasite interface. Our results might contribute to an in depth understanding of the biophysical and biological features of EmAgB3, thus providing insights into the design of novel targets to control AE.

Cystic echinococcosis (CE) is a pandemic infectious disease caused by the tapeworm Echinococcus granulosus that forms cysts in different organs such as lungs and liver. Imaging examination and serological tests have some drawbacks such as low sensitivity. In this study, we used an up-to-date workflow of laser microdissection-based microproteomics and matrix-assisted laser desorption/ionization time-of-flight imaging mass spectrometry in order to depict the proteomic pattern of CE in the liver. This investigation revealed specific markers of a parasitic cyst in liver. This proteomic pattern could facilitate diagnosis of CE in the future., (Copyright © 2016 Elsevier B.V. All rights reserved.)

Biliary leakage (BL) is the most common cause of postoperative morbidity after conservative liver hydatid cyst surgery. The objective of this study was to determine incidence of BL and related risk factors in patients with solitary liver hydatid cyst who underwent conservative surgery. A total of 186 patients were included in this study. Age, gender, cyst recurrence, diameter, and localization, World Health Organization Informal Working Group on Echinococcosis (WHO-IWGE) classification, cavity management techniques, cyst content, and alkaline phosphatase (ALP) levels were evaluated with univariate and multivariate analyses. Of these patients 104 were female and 82 were male. The mean age was 43.5 ± 14.7 years. Postoperative BL was detected in 36 (19.4%) patients. Cyst diameter (P = 0.019), cyst localization (P = 0.007), WHO-IWGE classification (P = 0.017), and ALP level (P = 0.001) were the most significant risk factors for BL. Independent risk factors for BL were perihilar localization, large cyst diameter, high ALP level, and advanced age according to WHO-IWGE classification.

G1 genotype of Echinococcus granulosus sensu stricto is the major cause of hydatidosis in Northern Africa, Tunisia included. The genetic relationship between lung and liver localization were studied in ovine, bovine and human hydatid cysts in Tunisia. Allozyme variation and single strand conformation polymorphism were used for genetic differentiation. The first cause of genetic differentiation was the host species and the second was the localization (lung or liver). The reticulated genetic relationship between the liver or the lung human isolates and isolates from bovine lung, is indicative of recombination (sexual reproduction) or lateral genetic transfer. The idea of two specialized populations (one for the lung one for the liver) that are more or less successful according to host susceptibility is thus proposed., (Copyright © 2016 Elsevier B.V. All rights reserved.)

Introduction: Alveolar echinococcosis is a life-threatening zoonotic parasitic disease. Its incidence is rare. In some cases, the correct and timely diagnosis can be difficult., Case Report: The authors present the case of a young patient with liver, diaphragm and lung involvement. The suspicion of echinococcus infection was made on the basis of medical history, clinical symptoms, and a combination of ultrasonography, computed tomography, magnetic resonance imaging tests and serological methods. The patient underwent multimodal treatment with albendazole and en-bloc resection of the liver, lung and diaphragm. The definitive diagnosis of alveolar echinococcosis was determined from samples of the resected tissues using histopathology and polymerase chain reaction methods. The patient has been followed regularly and is on life-long treatment with albendazole., Conclusion: The precise diagnosis and multimodal therapy of alveolar echinococcosis is fundamental from the point of view of patient long-term survival., Key Words: alveolar echinococcosis - diagnosis - multimodal treatment - follow-up.

Alveolar echinococcosis is a parasitic disease limited to the northern hemisphere. The disease occurs primarily in the liver and shows a profile mimicking slow-growing malignant tumors. Echinococcus multilocularis infection is fatal if left untreated. It can cause several complications by infiltrating the vascular structures, biliary tracts, and the hilum of the liver. As it can invade the adjacent organs or can spread to distant organs, alveolar echinococcosis can easily be confused with malignancies. We provide a brief review of epidemiologic and pathophysiologic profile of alveolar echinococcosis and clinical features of the disease. This article focuses primarily on the imaging features of alveolar echinococcosis on ultrasonogra-phy, computed tomography, magnetic resonance imaging, diffusion-weighted imaging and positron emission tomography-computed tomography. We also reviewed the role of radiology in diagnosis, management, and follow-up of the disease.

Objective: Contrast-Enhanced Ultrasound (CEUS) has been introduced as a promising imaging technique for diagnosis of hepatic alveolar echinococcosis (HAE). But the correlation between the image features and the underlying complex pathology of HAE has not been fully understood. In this study, we reviewed CEUS and pathological images of 31 lesions in 24 patients with HAE from Aba Tibetan Qiang Autonomous Prefecture, an epidemic area in China., Materials and Methods: 24 patients who received CEUS examination for suspicion of HAE and with pathologically confirmed HAE were retrospectively reviewed. Parasitic lesions obtained from surgery were sectioned and stained with hematoxylin and eosin (HE)., Results: US examination showed that the 12 lesions were hypoechoic and 19 lesions were hyperechoic. The hypoechoic images of HAE quite resemble the images of hepatocellular carcinoma and hemangioma. For the CEUS images of lesions ≥3 cm, 9 lesions (9/25, 36%) were hypoechoic with mixed content without circular rim enhancement; 16 lesions (16/25, 64%) had circular rim enhancement. All the lesions < 3 cm (n=6) were with circular rim enhancement and non-enhancement internal area. Pathological examination showed that the cysts are surrounded by an inner necrotic zone and peripheral granulomatous and fibrous tissues., Conclusions: CEUS images of large HAE lesions are more complex than that of the small lesions. Large HAE lesions can be hypoechoic with mixed content with or without circular rim enhancement, and no internal area enhancement with circular rim enhancement. The small lesions are more likely to show circular rim enhancement and non-enhancement internal area.

A cross sectional study compared the clinical features of the pulmonary and hepatic hydatid cysts in children and adults and evaluated IHAT and ELISA techniques in diagnosis. The results showed that the patients ages were 5-14 years (10.6 ± 3.7) in children and 16-75 years (32.2 ± 14) in adults, patients 34 (75.5%) had liver cysts, 25 (55.5%) had pulmonary cysts and 7 (15.5%) had both liver and lung cysts. In hepatic hydatidosis, 7/34 (20.5%) cases were asymptomatic while others showed variable clinical manifestations. The commonest symptom was localized right hypochondrial pain in 13 (38.2%) and the least one was jaundice in 4 (11.7%). The commonest sign was abdominal masses on the right hypochondrium in 88.2% and the least one was ascites in 5.8%.The commonest symptom of pulmonary hydatidosis was chest pain in 8 (34.7%) followed by cough and hemoptysis pn 4 (17.3%) and the least one was cough and fever (8.6%). Pulmonary hydatid cysts in children were significantly higher in males (17.3%) than females (4.3%), but without significance in adults (26% in male vs. 21.7% in females). Sex difference in hydatid cyst frequencies between adults and children was significant (P < 0.05). Mixed hepatic and pulmonary cysts were less in children than in adults (14.3% vs. 85.7%), with huge pulmonary cysts of 20 cm were more common in children (37.7%) than in adults (17.7%). The high sensitivity (95.5%) of ELISA-IgG recommended this test showed a dependable sero-diagnosing one.

Alveolar echinococcosis (AE) is a clinically very severe zoonotic helminthic disease, characterized by a chronic progressive hepatic damage caused by the continuous proliferation of the larval stage (metacestode) of Echinococcus multilocularis. The proliferative potential of the parasite metacestode tissue is dependent on the nature/function of the periparasitic immune-mediated processes of the host. Immune tolerance and/or down-regulation of immunity are a marked characteristic increasingly observed when disease develops towards its chronic (late) stage of infection. In this context, explorative studies have clearly shown that T regulatory (Treg) cells play an important role in modulating and orchestrating inflammatory/immune reactions in AE, yielding a largely Th2-biased response, and finally allowing thus long-term parasite survival, proliferation and maturation. AE is fatal if not treated appropriately, but the current benzimidazole chemotherapy is far from optimal, and novel options for control are needed. Future research should focus on the elucidation of the crucial immunological events that lead to anergy in AE, and focus on providing a scientific basis for the development of novel and more effective immunotherapeutical options to support cure AE by abrogating anergy, anticipating also that a combination of immuno- and chemotherapy could provide a synergistic therapeutical effect., (© 2015 John Wiley & Sons Ltd.)

Purpose: To investigate whether dual-energy computer tomography(DECT) could determine the angiographic vascularity of alveolar echinococcosis lesions by comparing the quantitative iodine concentration (IC) with the microvascular density (MVD)., Material and Methods: Twenty-five patients (16 men, 9 women; mean age, 40.9 ± 13.8 years) with confirmed hepatic alveolar echinococcosis (HAE) underwent DECT of the abdomen, consisting of arterial phase (AP), portal venous phase (PVP), and delayed phase (DP) scanning, in dual-source mode (100 kV/140 kV). Image data were processed with a DECT software algorithm that was designed for the evaluation of iodine distribution in the different layers (marginal zone, solid and cystic) of the lesions. The CT patterns of HAE lesions were classified into three types: solid type, pseudocystic type and 'geographic map' (mixed) type. The IC measurements in different layers and different types of lesions were statistically compared. MVD was examined using CD34 immunohistochemical staining of the resected HAE tissue and scored based on the percentage of positively stained cells and their intensity. Pearson's correlation analysis was used to evaluate the potential correlation between DECT parameters and MVD., Results: A total of 27 HAE lesions were evaluated, of which 9 were solid type, 3 were pseudocystic type and 15 were mixed type. The mean lesion size was 100.7 ± 47.3 mm. There was a significant difference in the IC measurements between different layers of HAE lesions during each scan phase (p < 0.001). The IC in the marginal zone was significantly higher than in the solid and cystic components in AP (2.15 mg/mL vs. 0.17 or 0.01 mg/mL), PVP (3.08 mg/mL vs. 0.1 or 0.02 mg/mL), and DP (2.93 mg/mL vs. 0.04 or 0.02 mg/mL). No significant difference was found among the different CT patterns of HAE lesions. Positive expression of CD34 in the marginal zones surrounding HAE lesions was found in 92.5% (25/27) of lesions, of which 18.5% (5/27) were strongly positive, 62.7% (17/27) were moderately positive, and 11.1% (3/27) were weakly positive. In contrast, 7.4% (2/27) of the lesions were negative for CD34. There was a positive correlation between IC measurements and MVD in the marginal zone of HAE lesions (r = 0.73, p < 0.05)., Conclusions: The DECT quantitative iodine concentration was significantly correlated with MVD in the marginal zones surrounding HAE lesions. Dual-energy CT using a quantitative analytic methodology can be used to evaluate the vascularity of AE.

An acute postresection hepatic insufficiency (PHI) constitutes a necessary moment before the hepatic resection planning, permits in some situations to conduct prophylactic measures and to avoid this severe complication. Possibility of PHI occurrence was prognosticated for results of surgical treatment improvement in patients, suffering focal hepatic affection, using introduction of certain preoperative preparation and surgical tactics. The main task of the investigation was to determine the diagnostic and prognostic value of the investigation methods and elaboration of prognostic algorithm of an acute PHI occurrence.

The aim of the study was to investigate ultrasound features (US) of liver after post operative anti-parasite recurrence treatment of patients with echinococcosis. The clinical analyses of 50 patients were carried out. It was concluded that the use of ultrasound can provide valuable data to the clinician to assess and monitor anti parasitic therapy echinococcosis of liver in post operative period. During the monitoring the positive dynamics of disease was observed in 94,5% of cases, in 5% оf cases toxic hepatites with septic complication was diagnosed, and in 0,5% of cases the disease recurrence was revealed. Ultrasound semiotics of liver after post operative anti-parasite recurrence treatment of patients with echinococcosis was presented. Control and monitoring of patients in the postoperative period echinococcosis with appropriate antirelapse antiparasitic therapy should be held not less than 1-5 years.

Background: This study retrospectively analyzed the clinical data, laboratory results, imaging findings, and histopathological features of 28 patients who underwent ultrasound-guided core-needle biopsy from a hepatic lesion and were diagnosed with alveolar echinococcosis., Results: Among 28 patients included in the study, 16 were females and 12 were males. The mean age of the studied population was 53 ± 16 years, and the age range was 18-79 years. The most common presenting symptom was abdominal pain, which was observed in 14 patients. A total of 36 lesions were detected in the patients' livers, out of which 7 had a cystic appearance. Hepatic vascular involvement, bile duct involvement, and other organ involvement were depicted in 14, 5, and 7 patients, respectively. The average number of cores taken from the lesions was 2.7, ranging between 2 and 5. In histopathological evaluation, PAS+ parasitic membrane structures were visualized on a necrotic background in all cases. Regarding seven patients, who were operated, the pathological findings of preoperative percutaneous biopsies were in perfect agreement with the pathological examinations after surgical resections. None of the patients developed major complications after biopsy., Conclusion: Ultrasound-guided core-needle biopsy is a minimally invasive, reliable, and effective diagnostic tool for the definitive diagnosis of hepatic alveolar echinococcosis., (© M. Bulakci et al., published by EDP Sciences, 2016.)

Introduction: Hydatidosis is a parasitic disease with tumour-like development of a cystic mass. This has specific endemic areas, Romania being amongst them. Our hospital has national addressability and the collaboration between the Departments of Parasitology, General Surgery and Pathology ensures optimal multidisciplinary approach to cases of therapeutic and diagnostic standpoint. The study aims to test the hypothesis that the gallbladder is a hydatid reservoir, to identify signs of biliary fistulas in the pericyst and liver parenchyma; to identify inflammation elements in the pericyst and the gallbladder., Material and Methods: The study is a retrospective observational one, carried out between 2011-2014, on a total of 35 patients operated for hepatic hydatidosis in the General Surgery Department of "Colentina" Clinical Hospital. All the selected patients had sent to the Pathology Department: gallbladder, cyst and pericyst. Statistical analysis of the data was performed using SPSS package Statistics 19., Results: The main results of the study revealed no evidence to confirm the hypothesis that the gallbladder is a hydatid reservoir. Out of the 35 cases, in 16 we observed the tendency to include hepatic biliary ducts in the pericyst or the formation of new canals which lead to the formation of biliary fistulas. Using immunohistochemical techniques with mark of CK19 (cytokeratin 19), have been observed the pattern of fistulization and modification of local architecture through the formation of the pericyst, in 16/35 (45.7%) of cases., Conclusion: Although it is a benign pathology, the evolution of hepatic hydatidosis can lead to severe complications and a low quality of life for the patient, both before and after surgery. Better knowledge of the pathology behind the local evolution of the disease can influence the therapeutic approach.

Hydatid disease is a parasitic infestation caused by ingestion of eggs of echinococcal species. For Echinococcus granulosus, the definitive host is the dog, and sheeps are the usual intermediate hosts. Humans are accidental intermediate hosts, infected by ingestion of food contaminated with eggs shed by dogs or foxes. The most common organs that hydatid disease encountered are the liver and lungs. Involvement of the kidney is rare and usually accompanies the other organ involvements. Cardiac involvement of echinococcosis is also very rare. We report the case of a 31-year-old woman with a 6-year history of asthma who collapsed after strenuous activity and died despite the interventions carried out. At autopsy, cystic masses were detected in the apex of the heart, in the right kidney, and in the liver. There were no macroscopic pathologic findings in the other organs. Microscopic examination revealed the diagnosis of hydatid cyst in the heart, right kidney, and liver besides medial hypertrophy of the lung vessels. Cause of death was attributed to hydatid cyst and its complications. Patients who have symptoms akin to asthma at clinical presentation have to be further investigated for organic cardiac and pulmonary diseases such as hydatid cyst, especially in endemic countries.

Background: The local immune responses to chronic echinococcal infections in various organs are largely unknown. Since the liver is the most frequently involved organ in such infections in human we aimed to characterize the inflammatory as well as immune cell infiltrate around hydatid cysts in the liver and compared to common inflammatory processes of the liver., Method: Surgical samples from the liver of 21 cystic echinococcosis (CE) patients were studied and the distribution of different types of inflammatory and immune cells were determined by immunohistochemistry. Furthermore, expression levels of costimulatory CTLA4, CD28, CD80 and CD86 molecules were measured at RNA level by PCR. Liver biopsy samples from patients with steatohepatitis (SH, n = 11) and chronic hepatitis (CH, n = 11) were used as non-inflammatory and chronic inflammatory controls, respectively. The composition and density of the inflammatory and immune cell infiltrates have been compared by using morphometry., Results: CD3+ T cells predominated the inflammatory infiltrate in all pathological processes, while in CE samples CD20+ B cells, in CH samples CD68+ macrophages were also frequent. Both myeloperoxidase (MPO) + leukocytes and CD68+ macrophages were found to be significantly decreased in CE as compared to either SH or CH samples. Concerning T cell subtypes, only CD8+ T cells were found to be significantly decreased in SH samples. CD1a + dendritic cells were almost completely missing from CE biopsies unlike in any other sample types. There were no differences detected in the mRNA expression of costimulatory molecules except decreased expression of CD28 in CE samples., Conclusion: In the hydatid lesions of the liver of chronic echinococcal infections T cell-mediated immunity seems to be impaired as compared to other types of chronic inflammatory processes, suggesting an immunosuppressive role for Echinococcus granulosus, which deserve further attentions.

This review gives a critical update of the situation regarding alveolar echinococcosis (AE) in Europe in humans, based on existing publications and on findings of national and European surveillance systems. All sources point to an increase in human cases of AE in the "historic endemic areas" of Europe, namely Germany, Switzerland, Austria and France and to the emergence of human cases in countries where the disease had never been recognised until the end of the 20th century, especially in central-eastern and Baltic countries. Both increase and emergence could be only due to methodological biases; this point is discussed in the review. One explanation may be given by changes in the animal reservoir of the parasite, Echinococcus multilocularis (increase in the global population of foxes in Europe and its urbanisation, as well as a possible increased involvement of pet animals as definitive infectious hosts). The review also focuses onto 2 more original approaches: (1) how changes in therapeutic attitudes toward malignant and chronic inflammatory diseases may affect the epidemiology of AE in the future in Europe, since a recent survey of such cases in France showed the emergence of AE in patients with immune suppression since the beginning of the 21st century; (2) how setting a network of referral centres in Europe based on common studies on the care management of patients might contribute to a better knowledge of AE epidemiology in the future., (Copyright © 2015. Published by Elsevier B.V.)

Background/aims: Benzimidazoles are efficacious for treating non-resectable alveolar echinococcosis (AE), but their long-term parasitocidal (curative) effect is disputed. In this study, we prospectively analyzed the potential parasitocidal effect of benzimidazoles and whether normalization of FDG-PET/CT scans and anti-Emll/3-10-antibody levels could act as reliable "in vivo" parameters of AE-inactivation permitting to abrogate chemotherapy with a low risk for AE-recurrence., Method: This prospective study included 34 patients with non-resectable AE subdivided into group A (n = 11), followed-up after diagnosis and begin of chemotherapy at months 6, 12 and 24, and group B (n = 23) with a medium duration of chemotherapy of 10 (range 2-25) years. All patients were assessed by FDG-PET/CT examinations and anti-EmII/3-10 serology. Chemotherapy was abrogated in patients with normalization of FDG-PET/CT and serum anti-EmII/3-10 levels. These patients were closely followed-up for AE recurrence. Endpoint (parasitocidal efficacy) was defined by the absence of AE-recurrence >24 months after stopping treatment., Results: Normalization of FDG-PET/CT scan and anti-EmII/3-10 levels occurred in 11 of 34 patients (32%). After abrogation of chemotherapy in these 11 patients, there was no evidence of AE-recurrence within a median of 70.5 (range 16-82) months. However, the patients' immunocompetence appears pivotal for the described long-term parasitocidal effect of benzimidazoles., Conclusions: The combination of negative FDG-PET/CT-scans and anti-EmII/3-10 antibody levels seem to be reliable parameters for assessing in vivo AE-larval inactivity after long-term benzimidazole chemotherapy., Trial Registration: clinicaltrials.gov: NCT00658294.

Background: Prognostic predictions in alveolar echinococcosis (AE) are usually based on relapse and metastasis. Thus, assessment of factors associated with metastasis and clinical outcomes in patients with alveolar echinococcosis are of significant important. The purpose of this study was to investigate the correlated factors for metastasis and prognosis for patients with AE., Methods: We analyzed 159 AE patients who underwent curative surgery at the First Teaching Hospital of Xinjiang Medical University from February 2003 to December 2013 by hematoxylin and eosin (HE) staining. Immunohistochemistry (IHC) staining for CD44 was performed in 159 AE and adjacent normal liver specimens. The clinicopathological parameters were analyzed for metastasis and prognosis, including gender, age, size, calcification, necrosis, foreign body granuloma and CD44 protein level., Results: The rate of metastasis was 22.1%. Chi-square test showed that low levels of CD44 was associated with metastasis (P = 0.004). However, multivariate analysis suggested that CD44 expression is not independent prognostic indicators for overall survival (P = 0.356). In addition, no correlations between prognosis and gender, age, size, calcification, necrosis, foreign body granulomatous, metastasis were identified., Conclusions: The finding that a low level of CD44 is associated with metastasis in AE patients. At present, it thus remains question whether CD44 expression is a valid prognostic marker for AE, further investigations are required.

Background: Echinococcus multilocularis, the causative agent of alveolar echinococcosis, is a fox tapeworm widely distributed in Europe with an increase of endemic area in recent years. Many mammal species including humans and non-human primates can be infected by accidental ingestion of eggs., Case Presentation: In March 2011, a 5-year-old zoo-raised male cynomolgus macaque (Macaca fascicularis) presented a paresis of the lower limbs which evolved into paralysis. Lesions in liver and vertebra were observed on tomography scan. E. multilocularis infection was diagnosed post-mortem by morphological and histological examination and detection of Em DNA by polymerase chain reaction. Serodiagnosis of other primates of the colony using enzyme-linked immunosorbent assay (ELISA) was negative. In June 2013, at necroscopy, a hepatic and a paravertebral masses were detected in a second cynomolgus macaque of the same colony. Serology and DNA isolated from hepatic and abdominal cysts confirmed E. multilocularis infection., Conclusions: We described hear vertebral and liver localization of alveolar echinococcosis in non-human primates. The animals lived in an indoor/outdoor housing facility, where the probable mode of contamination is by ingestion of food foraging around the enclosure which could be contaminated with fox feces. Serological survey in the facility should allow us to estimate the risk of human contamination and the zoonotic risk of monkey infection due to environmental contamination.

One of the most common and serious complications of hepatic hydatid cyst disease is communication between the cyst and the biliary tree. Surgical management of biliary fistula is associated with high morbidity and mortality. We retrospectively reviewed the effectiveness of endoscopic treatment of ruptured hydatid cyst into intrahepatic bile ducts. Diagnosis of intrabiliary rupture of hydatid cyst was mostly suspected by acute cholangitis, jaundice, pain, and/or persistent external biliary fistula after surgery. The diagnosis was confirmed by radiology and endoscopic retrograde cholangiopancreatography (ERCP) findings. We retrospectively reviewed clinical, laboratory, imagery, and ERCP findings for all patients. The therapeutic methods performed were endoscopic sphincterotomy, extraction by balloon or Dormia basket, stenting, or nasobiliary drainage. Sixteen patients with ruptured hepatic hydatid cyst into bile ducts were seen in 9 years. Nine of 16 patients had a surgical history of hepatic hydatid cyst and three patients had a percutanous treatment history. We carried out ERCP with sphincterotomy and extraction of hydatid materials (extraction balloon n = 11; Dormia basket n = 5) or biliary drainage (nasobiliary drainage n = 1; biliary stenting n = 1). The fistula healed in 80 % of patients with a median time of 6 weeks [range, 1-12] after endoscopic treatment. ERCP was an effective method of treatment for hepatic hydatid cyst with biliary fistula.

The aim of this study was to evaluate total antioxidant capacity (TAC), total oxidant status (TOS), and oxidative stress index (OSI) in sheep and lambs with cyctic eccinocoocosis (CE) diagnosed by ultrasonography and necropsy findings. A total of 9 sheep and 17 lambs with CE were used in this study and the findings were compared to those of 6 healthy control sheep. Ultrasonography were used for the diagnosis of CE in sheep and lambs, and necropsy was performed to check the presence of cysts in liver and lungs. Serum TOS and TAC were measured by a novel colorimetric method. The TOS-to-TAC ratios were also calculated as OSI values. Serum biochemical profiles were determined by conventional measurement methods as well. The mean values for TOS, TAC and OSI were significantly (p<0.001) lower in sheep and lambs with CE when compared with those of the control sheep, and they were also significantly lower in lambs with CE in comparison to the mean values obtained in sheep with CE. The levels of serum albumin, total cholesterol, creatinine, and triglycerides in lambs with CE were found out to decrease significantly (p<0.001) when compared with those of both sheep with EC and the control group. There were no significant differences between the groups in terms of other serum parameters. In addition, when clinically and some biochemical values were evaluated, CE was found to be more severe in lambs than in sheep. It was concluded that although common diagnostic cyst detection is performed by postmortem examination, ultrasonography could successfully be used in conjunction with serum biochemical profile detection and serum TOS, TAC and OSI measurements for diagnosis of cysts in liver and lungs of severely infected living sheep and lambs. Serum albumin, total cholesterol, creatinine, total protein and triglycerides might be used as indicators in sheep and particularly in lambs for the diagnosis of CE., (Copyright © 2015 Elsevier B.V. All rights reserved.)

Alveolar hydatid disease is a highly malignant form of echinococcosis caused by the larvae of the cestode echinococcus multilocularis. Alveolar hydatid disease always affects the liver and can metastasise to the lung and brain. Early diagnosis and precise evaluation of the localization as well as the extent of lesions are essential for treatment. In this report, we present ultrasound and computed tomography findings in a patient with hepatic alveolar echinococcosis. The patient, who was presented with hepatomegaly, jaundice, and an infiltrative solid tumor, diagnosed by ultrasound and computed tomography. In contrast to hydatid cyst caused by echinococcus granulosus, this is a rare disease in Iran.