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Humoral and cellular immunity in patients with hepatic alveolar echinococcosis. A 2 year follow-up with and without flubendazole treatment.

Parameters of humoral and cellular immunity were assessed in 12 patients with alveolar echinococcosis (AE) of the liver before, during and after discontinuation of treatment with flubendazole (FZ). In infected patients, before any medical treatment values of serum IgG, IgA, total haemolytic complement and C4 were significantly higher than those observed in control subjects; IgA levels were higher in jaundiced patients. Specific antibodies assayed by indirect haemagglutination and immunoelectrophoresis were present only in infected patients and were shown to decrease by the sixth month of treatment; however, similar fluctuations were observed without treatment. The percentage and absolute number of B lymphocytes, and total circulating lymphocytes, were significantly lower in patients with AE. An impairment of functional activity of T cells assayed by the leucocyte migration test, with PPD and Candidin as antigens, was demonstrated despite a normal percentage of SRBC rosettes. The 'score' of migration index still decreased during FZ treatment and returned to initial values after the year of follow-up without treatment. These results suggest that human AE is associated with important immunological disturbances. Changes in humoral immunity can be unequivocally considered to be a consequence of the parasite infection. The primary or secondary nature of the impairment of cellular immune responses and its mechanisms remain to be elucidated. Flubendazole could be responsible for an increase of cellular immune alterations in these patients.

Adult↗

Nephrectomy for benign disease? A case of isolated renal echinococcosis.

Cystic hydatid disease may be found in virtually any organ, although involvement of the urinary tract is relatively uncommon. We report a case of isolated renal hydatid disease presenting with hydatiduria. A short review of the literature regarding diagnosis and management of renal echinococcosis is also presented.

Aged↗

Stereological evaluation of treatment response in patients with non-resectable hepatic alveolar echinococcosis using computed tomography via the Cavalieri method.

INTRODUCTION: The purposes of this study were to describe and adapt the relevant methods of computed tomography (CT) and stereology to estimate parasitic volume in the liver, to compare the efficiency of benzimidazole treatment in hepatic alveolar echinococcosis (AE), and to determine whether the response rates measured by the stereological method are correlated with those measured by simple volumetric measurements (SVM). METHODS: Nine eligible patients with non-resectable AE were included in the study. By using their abdominal CT at the baseline and after a year of treatment, treatment responses of the cases were evaluated both by the stereological method via a software and by SVM, retrospectively. The volume estimation was performed in our study using a different approach that consisted of three separate stages combined with the Cavalieri method of modern design stereology. RESULTS: The response rates were -17 +/- 55% and -12 +/- 37% by the Cavalieri method and SVM, respectively; however, they were not statistically significant (P = 0.59 and 0.21 for the Cavalieri method and SVM, respectively). Although some cases had comparable results, others had different response rates, and the two methods showed no significant correlation (r = -0.31, P = 0.41). CONCLUSIONS: Because this modified method provides accurate results by reducing margin of errors, even in case of bizarre shape of AE, a correct, unbiased, and reliable management of the cases with AE via this method may be possible. Owing to lack of a correlation with SVM, it is suggested that a measurement via SVM may be wrong and its use in the evaluation of the treatment response in such cases will not be sufficient and completely true.

Adult↗

Progress of untreated massive cardiac echinococcosis--echocardiographic follow-up.

A 56-year-old man was admitted with chest pain and dyspnea. Echocardiographic evaluation revealed a giant cystic cardiac mass with multiple loculations at interventricular septum extended to inferoposterior region protruding inside the cavity. The patient refused surgical therapy. His complaints persisted without significant changes after 5 months. Second echocardiographic evaluation revealed conjugation of previous multiple cyst to gigantic intramyocardial cyst and minimal pericardial effusion. We intend to illustrate herein an unusual echocardiographic appearance and progress of an untreated massive "cardiac echinococcosis." If cardiac hydatid cyst is left untreated it may transform to large cavity with a high risk of rupture.

Animals↗

A new therapeutic approach for the treatment of cystic echinococcosis: percutaneous albendazole sulphoxide injection without reaspiration.

OBJECTIVE: In this experimental study, the effectiveness of intracystic injection of albendazole sulfoxide solution was investigated as a new approach to percutaneous treatment in liver hydatid disease. METHODS: Ten naturally infected sheep were selected and divided into two groups: a treatment group (n = 7), and a control group (n = 3). Intracystic injection of albendazole sulfoxide was performed in the first group, whereas the control group received intracystic distillated water injection instead. No reaspiration was performed in any group. RESULTS: During the follow-up period of 6 months, serial sonographic examination revealed a significant decrease in the cyst size, progressive solidification, and complete separation of the germinal and the laminated membranes of hydatid cysts from the pericysts in the treatment group. In the control group, diameters and volumes of cysts were increased. All procedures were done without any complications. During the follow-up-period, liver function tests were normal. After 6 months, all sheep were killed and were examined for macroscopic and microscopic changes. Pathological examination showed pericyst hyalinization, inflammatory cells in the cyst wall, degeneration of laminated and germinal membranes, and necrotic material in the cyst cavity. No viable protoscoleces or daughter cysts were observed. CONCLUSION: Albendazole sulfoxide injection as a scolecidal agent in the percutaneous treatment of cystic echinococcosis seems to be effective in this animal model. Further studies are suggested to evaluate the effectiveness of this procedure in human subjects.

Albendazole↗

[Compile and evaluation of national surveillance on human echinococcosis in Japan, 1999 to 2002].

National surveillance on human ecinococcosis has been performed since April 1999 when it was stipulated as a Category IV Disease under the Infectious Diseases Prevention Law. During the last 4 years of surveillance, 3 cases with unilocular hydatidosis (age range 27-81 years; median age 55 years) and 51 cases with alveolar hydatidosis (age range 15-86 years; median age 64 years) have been reported. The numbers of reported cases with alveolar hydatidosis have been increased with age, and the largest number reported in the age group > or = 71 years. Three cases with unilocular hydatidosis have been reported from health centers in Honshu, and were likely to be imported cases. Seventeen reported cases had clinical symptoms. None of the reported cases had information on infection route. Fifty out of 51 cases with alveolar hydatid disease have been reported from health centers in Hokkaido. When analyzing the cases by subdividing Hokkaido into six regional districts, large number of cases were reported from health centers in Ishikari-Shiribeshi-Iburi region (20 cases) and in Nemuro-Abashiri-Kushiro region (15 cases). As detailed addresses of the cases were masked, we compared the number of cases per 100,000 residents in regions. Health center in Nemuro-Abashiri-Kushiro region (2.13/100,000) had largest rate and second was Souya-Rumoi region (2.05/100,000). The results from current surveillance data only suggest the generation situation of human echinococcosis in several years ago or more, so the generation situation between 1999 April and 2002 December in surveillance is unknown.

Adult↗

Increased activation and oligoclonality of peripheral CD8(+) T cells in the chronic human helminth infection alveolar echinococcosis.

Alveolar echinococcosis (AE) in humans is a chronic disease characterized by slowly expanding liver lesions. Cellular immunity restricts the spreading of the extracellular pathogen, but functional contributions of CD4(+) and CD8(+) T cells are not defined. Here we studied ex vivo the phenotype and function of circulating T-cell subsets in AE patients by means of flow cytometry, T-cell receptor spectratyping, and lymphocyte proliferation. AE patients with parasitic lesions displayed a significant increase of activation of predominantly CD8(+) T cells compared to healthy controls and AE patients without lesions. In vitro, proliferative T-cell responses to polyclonal stimulation with recall antigens and Echinococcus multilocularis vesicular fluid antigen were sustained during chronic persisting infection in all AE patients. Only in AE patients with parasitic lesions did T-cell receptor spectratyping reveal increased oligoclonality of CD8(+) but not CD4(+) T cells, suggesting a persistent antigenic drive for CD8(+) T cells with subsequent proliferation of selected clonotypes. Thus, our data provide strong evidence for an active role of CD8(+) T cells in AE.

Adult↗

Improved primary immunodiagnosis of alveolar echinococcosis in humans by an enzyme-linked immunosorbent assay using the Em2plus antigen.

Alveolar echinococcosis (AE) in humans is generally a fatal disease when not diagnosed early enough to provide curative treatment such as radical surgery. Immunodiagnosis for early detection of AE was improved by the isolation of an affinity-purified metacestode Em2 antigen and by the synthesis of recombinant Echinococcus multilocularis antigen II/3-10. Both antigens were individually assessed by enzyme-linked immunosorbent assay (ELISA) and demonstrated high specificities and diagnostic sensitivities, although both missed approximately 4 to 11% of diagnostic cases of AE. To provide an optimal serodiagnostic test, we investigated the two purified antigens by using a test employing a mixture of both purified antigens (designated Em2plus antigen) in one assay. For comparative purposes, crude E. multilocularis and Echinococcus granulosus metacestode antigens were investigated as well. The Em2plus ELISA proved to be the optimal diagnostic test with the highest diagnostic sensitivity, 97%, in serum samples from 140 patients with AE and an overall specificity of 99% for infections due to other Echinococcus and non-Echinococcus parasites. The new test combination (Em2plus ELISA) is suggested for the serodiagnosis of AE in patients and for seroepidemiological surveys.

Animals↗

Urinary hydatid antigen detection by coagglutination, a cost-effective and rapid test for diagnosis of cystic echinococcosis in a rural or field setting.

We describe here coagglutination (Co-A), a rapid slide agglutination test for the detection of hydatid antigen in the urine for the diagnosis of cystic echinococcosis (CE). Paired urine and serum samples were collected from 16 patients with surgically confirmed CE, 10 patients with ultrasound-proven CE, 14 patients with clinically diagnosed CE, 24 patients with various parasitic diseases other than CE, and 25 healthy control subjects. Co-A detected excreted hydatid antigen in the concentrated urine of 7 of 16 (43.75%) surgically confirmed cases, 6 of 10 (60%) ultrasound-proven cases, and 8 of 14 (57.14%) clinically diagnosed cases of CE. A false-positive reaction was observed with 12.50% of control urine specimens from patients with parasitic diseases other than CE and 12% of urine samples from healthy controls. The circulating antigen was detected in the serum in 13 of 16 (81.25%) surgically confirmed cases, 6 of 10 (60%) ultrasound-proven cases, and 13 of 14 (92.86%) clinically diagnosed cases of CE. False-positive reactions were observed with three sera (12.5%) from controls with other parasitic diseases. The low sensitivity of Co-A for detection of antigen in the urine of a patient whose serum was positive for the antigen is possibly due to low levels of antigen in the urine. Unlike the collection of blood for serum, which is an invasive procedure and also requires technical expertise and disposable syringes, urine can be collected easily and frequently without causing any inconvenience to the patient. Urine as a clinical specimen alternative to serum would be immensely useful in the diagnosis of CE, particularly in a rural or field setting. In such situations as well as in poorly equipped laboratories, Co-A has the potential to be used as a simple, rapid, and economical slide agglutination test for detection of urinary hydatid antigen in the diagnosis of CE.

Agglutination Tests↗

Cardiac echinococcosis--a rare echocardiographic diagnosis.

A 30 year old female admitted for evaluation of left chest pain was suspected to have multiple cardiac hydatid cysts. The diagnosis was established by cross sectional echocardiography and computed tomography, supported by enzyme linked immunosorbent assay (ELISA) for echinococcosis. Medical therapy altered the echopattern of the cysts but failed to reduce cystic masses. Surgery was advocated but refused by the patient.

Adult↗

Radiologic aspects of hepatic echinococcosis. Value of the intravenous viscerogram and computed tomography.

Over five years, 105 patients with hepatic echinococcosis were studied. Plain radiographs of the hepatic area yielded the correct diagnosis in 35% of patients. Intravenous viscerograms were diagnostic in all but 14 of the patients (an accuracy of 83%). Computed tomography was performed in 19 cases and proved the most efficient of the techniques--it was diagnostic in all 19 cases. The radiological signs of this entity are discussed.

Calcinosis↗

Hepatic alveolar echinococcosis: correlative US and CT study.

A total of 24 cases of hepatic alveolar echinococcosis (HAE) due to Echinococcus multilocularis was assessed by US and CT. The diagnosis was confirmed in all cases by immunologic and histologic study. Both US and CT patterns of HAE showed changes of liver morphology in both contour and size. Abnormal areas of parenchyma were nodular or in fields, irregular, heterogeneous, and basically echogenic. On CT these lesions were hypodense (30 to 40 HU) and did not show enhancement after administration of intravenous contrast medium. Clustered microcalcifications were encountered within the abnormal parenchymal fields in 50% of cases, and necrotized zones occurred in 40% of cases. Dilatation of intrahepatic bile ducts was commonly seen, especially on US; hilar involvement was frequent. Follow-up by both techniques can display increases of primary lesions, occurrence of new foci, and local or regional extensions. Precise evaluation of the lesions arising from correlative use of US and CT permits adequate therapeutic management.

Adult↗

Alveolar echinococcosis in Turkey. Experience from an endemic region.

BACKGROUND: Radical resection is the only potentially curative treatment for hepatic alveolar echinococcosis (AE). Although Turkey is an endemic region, population screening is not performed and early diagnosis is rare. Consequently, surgeons are compelled to explore possibilities such as near-total resection and biliodigestive anastomosis for palliation of jaundice. METHODS: Surgery was performed in 32 patients with hepatic AE with the following indications: (1) resection; (2) palliation of jaundice; (3) definite assessment of operability; (4) failure in the management of cavity infection by percutaneous methods. Curative resection (R0 = complete resection of all parasitic mass [n = 9], and R1 = a resection in which a small remnant was left on a vital structure [n = 8]) were performed in 17 patients, intrahepatic cholangiojejunostomy in 7, laparotomy-external drainage in 7, and debulking in 1. RESULTS: Perioperative mortality rates were 2/17, 0/7, 2/7 and 1/1, respectively. Twelve patients in the curative resection group are alive without recurrence/progression of the small remnant during a median follow-up of 59 (range 27-116) months. One patient developed an inoperable recurrence that was treated with albendazole. One patient was lost to follow-up. Long-term albendazole treatment was effective in all R1 patients except a patient who had slow asymptomatic progression. Successful palliation of jaundice was achieved in 5 of the 7 intrahepatic cholangiojejunostomy patients. CONCLUSIONS: The results of R1 resection in alveolar hydatid disease are similar to those of R0 resection; a small remnant is successfully controlled by albendazole. In patients with jaundice due to hilar invasion, biliary diversion from segment 3 or 5 is effective for palliation of the jaundice and facilitates albendazole treatment.

Adult↗

Immunological diagnosis of echinococcosis.

Our results with the immunofluorescence technique in the diagnosis of hydatid disease are reported. The sera of 28 patients with surgically proven hydatid disease, of 30 healthy individuals, and of 1 patient with cysticercosis were used. With a dilution of 1:100 a sensitivity of 96% was obtained in the patients affected by the disease. In the control group all but one of the sera were negative. A brief review of the immunological methods used at present for the diagnosis of echinococcosis is made. The most important problems connected with the immunodiagnosis of this disease are discussed.

Adolescent↗

Bronchocentric granulomatosis and pulmonary echinococcosis.

Bronchocentric granulomatosis is probably a nonspecific pathologic reaction to various types of intrabronchial agents. The presence of noninvasive fungi has been demonstrated in the airways of asthmatic patients with bronchocentric granulomatosis. However, an etiologic agent has not yet been identified in the respiratory tract of patients without asthma. In the present report, a patient without asthma but with bronchocentric granulomatosis and pulmonary echinococcosis is described.

Adult↗

Vascular echinococcosis.

Three patients with arterial echinococcosis presented with chronic or acute arterial occlusion. One case diagnosed as Leriche's syndrome was found at surgery to have aortic occlusion caused by hydatid cysts; arterial reconstruction was performed by aortoiliac interposition of a woven Dacron vascular graft. In the other 2 cases presenting with acute femoral or brachial artery occlusion, embolectomy revealed hydatid cysts as the cause of occlusion.

Adult↗

A case-control study of the risk factors for cystic echinococcosis among the children of Rio Negro province, Argentina.

In 1984 the prevalence of cystic echinococcosis (CE) in Rio Negro province in central Argentina reached alarming levels, with almost 6% of children aged 7-13 years infected with the causative agent, Echinococcus granulosus. Although the control activities developed between 1980-2000 have now lowered the prevalence of infection in this age-group to 1.1%, transmission of E. granulosus has clearly not ceased. The aim of the present study was to identify possible flaws in the control programme and the risk factors associated with CE. The 24 cases and 66 controls used were identified during a survey of the 1,070 schoolchildren attending 12 schools in Ingeniero Jacobacci, Rio Negro province. In interviews based around a standardized questionnaire, the adult female with responsibility for each subject (usually the mother but sometimes a grandmother or guardian) was asked 70 questions about the child and his or her immediate family, their contact with dogs and relevant environmentmental factors, and their level of contact with the control programme. Univariate and multivariate analyses were used to determine odds ratios (OR) and their 95% confidence intervals (CI). The main risk factors found to be significantly associated with CE were having a family member with the disease (OR = 3.11; CI = 0.92-10.47), spending the first years of life surrounded by a large number of dogs (OR = 2.11; CI = 1.2-3.5), and having a father who slaughtered sheep at his workplace (OR = 1.14; CI = 1.04-1.24). Obtaining drinking water from a tap (OR = 0.28; CI = 0.08-1.01) also remained in the final model, as a protective factor.

Adolescent↗