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Cerebral alveolar echinococcosis is rare and has a poor prognosis. We report an unusual case presenting with disseminated intracranial lesions secondary to primary hepatic infection.
Cerebral alveolar echinococcosis is rare. We report a case with multiple intracranial masses which show cauliflower-like contrast enhancement pattern on MRI. The lesions originated from hepatic involvement with invasion of the inferior vena cava.
Cutaneous parasitic lesions, associated with a dense fibrous reaction, markedly improved under albendazole treatment in one case of supraumbilical skin localization of alveolar echinococcosis. Since collagen cross-linking increases during fibrogenesis and contributes to the stability of fibrotic lesions, we monitored the level of the cross-links pyridinoline and pentosidine in skin lesions from this patient to determine if they would reflect the changes occurring during treatment. We looked at the deposition of cross-linked type I collagen by immunohistochemistry and also measured the serum concentrations of pentosidine and of a fragment of type I collagen (ICTP), which contains a site of pyridinoline formation. Albendazole treatment did not affect either the collagen content of skin lesions or the serum concentrations of ICTP and pentosidine, but it led to a pronounced decrease in pyridinoline level concomitant with the disappearance, observed by immunohistochemistry, of extensively cross-linked fibrotic type I collagen. The follow-up of collagen cross-linking by pyridinoline in skin tissue thus appears to be useful in reflecting the improvement of fibrotic skin diseases during therapy.
Human alveolar echinococcosis (AE) is a rare and highly pathogenic helminthic zoonosis due to infection with the intermediate stage of the small fox tapeworm, Echinococcus multilocularis. Parasite transmission is restricted to northern latitudes, including central and north China, from where few clinical and no detailed community studies have been reported. In August, 1991, 65 (5%) of 1312 people residing in three rural communes of Zhang County, Gansu Province, China were diagnosed as having hepatic AE after mass ultrasound scanning with serological back-up. This represents one of the highest prevalence rates of AE ever recorded. It is also the first time that mass ultrasound scanning and serology have been used together in an AE endemic region. The region was selected one year earlier, when a preliminary serosurvey on 606 unselected people in the same locality resulted in an 8.8% serum antibody positive rate with a 76% rate of confirmation of hepatic AE in 37 individuals who could be followed up in 1991. Seropositivity rates varied for villages between 0 and 20.5%. Overall, females (7.8%) had a significantly greater risk of infection than males (2.5%), especially in the 31-50 age group, a difference which may be related to contact with dogs and dog faeces over many years. Age-specific prevalence of AE increased from 0% in the under 5-year group to 8.2% in those aged 31-50. The youngest case was 11 years and the mean age of diagnosis was 40 years. Adult tapeworms of E multilocularis were identified from the small intestines of 10% of domestic dogs. Sylvatic animal hosts of the parasite have not yet been identified. The high prevalence of human AE in this region of central China is most probably due to semi-domestic transmission of E multilocularis between wild rodents and dogs, together with the poverty and poor hygiene in these rural communities.
BACKGROUND/AIMS: Long-term chemotherapy with benzimidazoles is beneficial in non-resectable alveolar echinococcosis (AE). Criteria to track early therapeutic efficacy are lacking and the clinical impact of immunosurveillance is unsettled. We aimed to analyze this issue particularly for assessing the putative parasitocidal efficacy of chemotherapy. METHODS: The present study is part of our prospective Swiss trial outlined previously and comprises 57 patients with a median follow-up of 18.5 (3-30) years and with repeated tests of humoral and cell-mediated immunity. The series was subdivided into group A (n=23; curative surgery) and group B (n=34: non-resectable AE). RESULTS: Long-term survival was 87% (group A) and 76% (group B). The profiles of specific antibodies against EmII/3-10 antigen normalized within 3 years in most group A-patients, but remained above the cut-off value in 40% of group B-patients. This lack of normalization was associated with lower bioavailability of mebendazole. AE-recurrence after 'radical' surgery (up to 13 years) was associated with high anti-EmII/3-10 concentrations in 7 of 8 cases. Following abrogation of longterm chemotherapy in group B, no AE-recurrence occurred in 9/18 patients, suggestive of parasitocidal efficacy and documented by a normal EmII/3-10 profile. CONCLUSIONS: The EmII/3-10 profile is of value in monitoring AE after surgery and/or chemotherapy.
Globally human alveolar echinococcosis (AE) is a rare zoonotic helminthic disease confined to the Northern Hemisphere as sporadic infections in rural populations, principally in some areas of North America, west-central Europe, the Near East, Siberia, Central Asia, Japan and China. In China the first human cases were reported from western regions in the 1960s, but most hospital records remain fragmented and inadequate. From the mid-1990s mass screening surveys using portable ultrasound scanners recorded higher prevalences (up to 6% by county) than in any other areas of the world with some village rates as high as 15%. Risk factors identified for AE cases included ethnicity, sex, age and occupation. The role of the dog in transmission of Echinococcus multilocularis to humans now appears to be significant and may be one of the most important risk factor, in combination with landscape/land-use features conducive to maintaining wildlife host populations.
An unusual female case, with alveolar echinococcosis (AE) disseminated from the primary hepatic lesion to the brain by metastasis formation, was retrospectively identified during a community survey in Ningxia Hui Autonomous Region, northwest China in 2003. Among possible metastases of hepatic AE, locations to the brain are rare and usually fatal; and they have especially been assigned to concomitant immune suppression. An enhancing role of pregnancy, which may be suspected in this case, the favourable outcome after surgery and chemotherapy, and also a mental disability in a child following long-term intrauterine exposure to mebendazole, make the report particularly unique.
Cystic echinococcosis (CE), caused by the larval stage of Echinococcus granulosus sensu lato (s.l.), is a parasitic zoonotic disease recognized by the World Health Organization as a neglected tropical disease of significant public health concern. Despite ongoing control programs, CE remains endemic, underlining the need for integrated control strategies that involve new diagnostic and therapeutic tools. Recent investigations have spotlighted microRNAs (miRNAs) as key regulators in parasite development, immunomodulation, and as potential diagnostic and therapeutic targets. In the present research, a molecular study was conducted to investigate hydatid cyst samples (protoscoleces and germinal membranes) collected in southern Italy from different ruminant species (sheep, cattle, and water buffaloes), naturally infected with CE, with the ultimate goal of establishing a more comprehensive picture of miRNA expression patterns in these intermediate hosts. The bioinformatic analysis of hydatid cyst samples revealed 168 mature miRNAs. Among these, egr-miR-10-5p, egr-let-7-5p, and egr-miR-71-5p were the most abundant, with egr-miR-10-5p showing particularly high expression levels. No significant differences in miRNA abundance between host species were found. In contrast, when focusing on the comparison between protoscoleces and sterile germinal membranes, 24 miRNAs were found to be differentially expressed. Targeted qPCR of four selected miRNAs (egr-miR-71-5p, egr-let-7-5p, egr-miR-125-5p, and egr-miR-10-5p) showed clear overexpression in protoscoleces and in fertile germinal membranes compared with sterile ones. The differential miRNA expression patterns provide insight into the molecular mechanisms controlling the parasite's lifecycle and may guide the development of novel intervention methods to enhance CE control in endemic areas.
The probability of disease given the results of a test, is called the predictive value of the test. The predictive value of a test is not a property of the test itself but will vary according to the prevalence of the disease in the studied population. The positive predictive value (PPV) is the probability that the subject tested has the disease given that a positive result is obtained. The negative predictive value (NPV) is the probability that the subject tested is normal given that a negative result is obtained. As the prevalence of a disease in a population approaches zero so does the PPV and most of the positive cases will be 'false positives'. Conversely the NPV will be very high at low prevalences and there will be few 'false negative' results. The sensitivity and specificity of a test are properties of the test and do not vary with prevalence. The higher the sensitivity and specificity of a particular test the greater the predictive values will be at any given prevalence of the disease. Ultrasound (US) is increasingly used for detecting lesions due to cystic and alveolar echinococcosis (CE and AE) and portable US scanners facilitate community based mass screening surveys in remote rural communities. Screening is justified with AE and CE in endemic areas as diagnosis at an early stage can lead to a better prognosis following treatment. The sensitivity and specificity of US has been reported to be between 88-98% and 95-100% respectively for CE and the sensitivity is a little higher for AE. Both species have pathognomonic signs on US and the technique is considered to be the 'gold standard' although it is still an imperfect test. Clinical, laboratory and epidemiological data also play an important role in the diagnosis of CE and AE. US results where possible, should be evaluated in relation to these findings. Suspected CE and AE images, may benefit from the use of other imaging techniques such as magnetic resonance imaging, computerised tomography and in the case of AE angiography or cholangiography. Immunological tests or molecular biological techniques also provide a useful back up, especially for AE. As sensitivity and specificity are properties of the US diagnostic test they should not vary if the case mix reported in different studies remains the same. The use of the WHO standardised US classifications for CE and AE should be used so that the properties of the test are standardised. Quality control of field based studies will depend on geographical variations in the case mix and the relative proportions of cyst types without pathognomonic signs. The latter will have the most bearing on variations in specificity, as would the use of different classifications. Inter- and intra-observer variability and differences in prevalence will affect the performance of US in different endemic settings. Community based surveys must adhere to the highest ethical standards and the outcome of surveys should result in appropriate treatment and follow-up strategies for all infected individuals and suspected cases found during the surveys.
The case of a 26-year-old woman with cardiac and pulmonary echinococcosis is presented. Surgical treatment of the intrathoracic disease was done in a one-stage operation. Under cardiopulmonary bypass, a left ventricular hydatid cyst and a lower lobe pulmonary cyst were removed. The patient is completely asymptomatic and is leading a normal life two years after operation.
A case of hepatic alveolor echinococcosis with contiguous invasion of the pancreas, vertebrae, and spinal cord and intrathecal metastases to the brain stem is reported. The long course after a hemihepatectomy may be a factor in the modification of the natural course of the disease and in the induction of this unusual mode of spread.
Latex agglutination test (LAT) was standardized and evaluated to detect hydatid antigen in fluid samples aspirated from 6 surgically proved human cases of cystic echinococcosis (CE), 4 suspected human cases of CE (2 cases of cysts in the liver which were not confirmed surgically and 2 cases of pelvic cysts later confirmed as abscesses) and 7 cases of hydatid cysts of liver in cattle. Echinococcus granulosus scolices and hook lets were seen in aspirated fluid by microscopy and the characteristic germinal layer of the cyst wall was demonstrated by histopathology in 6 human hydatid cysts operated and removed by surgery. In case of cattle hydatid liver cysts no scolices or hook lets were seen in aspirated fluid as they were sterile cysts but characteristic laminated layer of the cyst wall was demonstrated by histopathology of these cysts. The LAT could detect antigen in fluid samples collected from all 6 human cases of surgically proved CE and 7 cases of hydatid cyst liver in cattle, thus showing sensitivity of 100%. The LAT could detect antigen in fluid samples collected from 2 suspected cases of CE liver in humans, which were not operated. The LAT was found to be specific. No cross reactivity was observed. The results of the study showed that LAT could be employed as a simple and rapid diagnostic procedure, as an alternate to microscopy, to confirm the hydatid etiology of a suspected cyst.
OBJECTIVE: Human echinococcosis remains a serious health problem for the Mediterranean countries, among them Greece. As there is no effective medical therapy, surgery is still the treatment of choice. MATERIAL AND METHODS: We present our experience in the surgical management of hydatidosis by a transthoracic approach, based on 85 patients (49 male, 36 female, aged 4-86 years) treated during 1986-1996. RESULTS: Twenty-one patients (26.3%) appeared with complications as: hydatidemesis (n = 5), hydropneumothorax (n = 3), cyst infection (n = 3), empyema thoracis (n = 8), cholebronchial (n = 3) and cholebronchopleural fistula (n = 1). The location of the cysts was: 61 in the lungs (right, 29; left, 24; bilateral, eight), 31 on the liver dome, six in the pleural cavity, two in the mediastinum, and one in each of pericardium, chest wall, and right pararenal space. Surgical approach involved a thoracotomy or median sternotomy in all cases. Pulmonary endocystectomy and capitonnage was the procedure of choice in the surgical management. Hepatic cysts were approached through a right thoracophrenotomy and were managed with evacuation of the main and daughter cysts, suture of the diaphragm to the margins of the cyst, and drainage of the cystic and pleural cavities. There was no in-hospital mortality. Major postoperative complications were: empyema thoracis (n = 3), biliary fistula (n = 2), and bronchopleural fistula (n = 1). Five patients presented later with seven recurrences of the disease. CONCLUSION: Transthoracic approach is a good and safe choice in surgical treatment of both the intrathoracic and the (concomitant or not) hydatid cysts on the upper surface of the liver.
In an abattoir study, 514 camels, slaughtered for meat production in different areas of northern Libya were examined for the presence of cystic echinococcosis (CE). In addition, 367 sheep and 184 goats were examined. The overall prevalence of infection with CE was 48% in camels, 15.8% in sheep and 3.8% in goats. The infection rate, number and size of cysts were significantly higher in older camels. In six city abattoirs across northern Libya, i.e. Zawia, Tripoli, El-Khumes, Mesurata, Sirt and Benghazi, the prevalence rate of infection in camels ranged from 38.7% to 55.2%, in comparison with sheep and goat rates which were between 0% and 37.9% and 0% and 8.2%, respectively. In camels, the lungs were the most frequently infected organs (85.4%) with liver cysts occurring at a significantly lower rate (33%). In contrast, the liver was the predominant infected site with prevalence values of 86% and 100% in sheep and goats, respectively. More than 90% of camel hydatid cysts were fertile. The possible role of camels in the transmission of CE in Libya is discussed.
The influence of pharmacological treatment on the immune response of patients with Echinococcus granulosus infection was evaluated by reverse transcriptase-polymerase chain reaction (RT-PCR) to determine mRNA expression for IL-12 p35, IL-12 p40, interferon-gamma (IFN-gamma), tumour necrosis factor-alpha (TNF-alpha) and IL-4 in PBMC from 12 patients before and after chemotherapy and from seven uninfected controls. Most patients' PBMC showed measurable amounts of IL-12 p35, IL-4, IFN-gamma and TNF-alpha mRNA in parasite antigen-stimulated and unstimulated cultures. Conversely, IL-12 p40 mRNA was detected almost exclusively in successfully treated patients (86%) after therapy. In these patients semiquantitative analysis of RT-PCR products showed a significant difference between IL-12 p40 mRNA mean levels before and after therapy (P = 0.03 in parasite antigen-stimulated cultures; P = 0.001 in unstimulated cultures). IL-4 mRNA was weakly expressed before therapy and more highly so after treatment in both groups of patients and under both culture conditions; IL-4 mRNA reached its highest level in post-therapy PBMC from patients in whom therapy failed (stimulated cultures). IFN-gamma and TNF-alpha mRNA expression increased in patients who responded to therapy and decreased in patients who did not. In contrast to IL-12 p35, IFN-gamma and TNF-alpha mRNAs, IL-12 p40 and IL-4 mRNAs were detected exclusively in patients, suggesting a close relationship between these two cytokines and cystic echinococcosis. Our findings indicate that chemotherapy influences the immune response, thus determining changes in Th1/Th2 cytokine mRNA patterns, predominantly in IL-12 p40 and IL-4 mRNA expression.
PURPOSE: To present the predominantly ultrasonographic (initial and follow-up) imaging in a disease that is rare among the Central European paediatric population--and to evaluate the role of ultrasound for initial staging and follow-up under antihelmintic therapy. METHOD: The imaging documents as well as the clinical record of a 10-year old Armenian girl with systemic hydatid disease (cystic echinococcosis) were analysed retrospectively. RESULTS: By means of ultrasound, the complete initial systemic spread of the disease with at least 11 cysts within the liver, 1 cyst in the left kidney, 1 peri-/2 intracardiac cysts, and 1 cyst in the dorsal musculature was detected. Repeated sonographic examinations allowed the estimation of successful medical treatment by the following criteria: size reduction of all cysts with changing internal structures from an initially echo-free to an increasingly homogeneous echodense character; no developing new cysts. In addition, CT and MRI enabled a more complete demonstration of especially the intra- and pericardiac lesions (preoperatively) and the exclusion of further intracranial cysts. CONCLUSION: In paediatric hydatid disease, ultrasonography yields important information not only with regard to the initial staging, but also to the antihelmintic therapeutic effects and thus helps to determine when to discontinue medical treatment.
Cystic echinococcosis (CE) is a zoonotic infection caused by echinococcus granulosus and is still frequent in endemic areas. CE is predominantly localised in the liver. The therapy of choice is the surgical sanitation of the lesions, increasingly through interventional procedures such as percutaneous punctation of the cysts, aspiration of the cystic fluid, instillation of a protoscolicidal solution and reaspiration of this solution (PAIR). However, in some cases, such as perforation into the biliary tract, PAIR is not feasible. We report on an eight-year-old girl who suffered from CE with perforation of one cyst into the biliary tract. The diagnosis was confirmed by ERCP and cholangioscopy and a right hemihepatectomy was performed. The girl received adjuvant chemotherapy with albendazol perioperatively and is still asymptomatic with no indications of disease 10 months after the operation.
In areas where human cystic echinococcosis (CE) is endemic, the results of ultrasonographic or X-ray examinations have revealed a surprisingly high prevalence of abdominal cysts in asymptomatic individuals. The results of preliminary studies indicate that the ratio of liver infection:lung infection (LI:LU) is much higher in the asymptomatic individuals (9:1 or 7:1) than is usual among symptomatic cases of liver CE (2:1). This difference may indicate that, compared with lung cysts, liver cysts rarely cause morbidity, perhaps because they grow at a slower rate than those in the lungs. In an attempt to explore this possibility, the published results of ultrasonographic and radiological surveys on general populations and the records of autopsies and hospital-based investigations of symptomatic cases of liver CE were reviewed. In general populations, the overall prevalence of cysts in the liver (2.5%) was found to be much higher than that of cysts in the lungs (0.3%), giving a LI:LU ratio of 8.3:1. In the symptomatic cases, however, LI:LU ratios were only 2.5:1 (based on hospital records) or 4.1:1 (based on autopsy records). In addition, the estimated mean growth rate of the cysts in 53 surgical cases of CE from the province of Río Negro in Argentina was found to be significantly higher than that of the cysts in 89 asymptomatic cases detected during ultrasonographic surveys in the same area.