Identification and comparison of intestinal microbial diversity in patients at different stages of hepatic cystic echinococcosis.

There is a significant focus on the role of the host microbiome in different outcomes of human parasitic diseases, including cystic echinococcosis (CE). This study was conducted to identify the intestinal microbiome of patients with CE at different stages of hydatid cyst compared to healthy individuals. Stool samples from CE patients as well as healthy individuals were collected. The samples were divided into three groups representing various stages of hepatic hydatid cyst: active (CE1 and CE2), transitional (CE3), and inactive (CE4 and CE5). One family member from each group was selected to serve as a control. The gut microbiome of patients with different stages of hydatid cysts was investigated using metagenomic next-generation amplicon sequencing of the V3-V4 region of the 16S rRNA gene. In this study, we identified 4862 Operational Taxonomic Units from three stages of hydatid cysts in CE patients and healthy individuals with a combined frequency of 2,955,291. The most abundant genera observed in all the subjects were Blautia, Agathobacter, Faecalibacterium, Bacteroides, Bifidobacterium, and Prevotella. The highest microbial frequency was related to inactive forms of CE, and the lowest frequency was observed in the group with active forms. However, the lowest OTU diversity was found in patients with inactive cysts compared with those with active and transitional cyst stages. The genus Agatobacter had the highest OTU frequency. Pseudomonas, Gemella, and Ligilactobacillus showed significant differences among the patients with different stages of hydatid cysts. Additionally, Anaerostipes and Candidatus showed significantly different reads in CE patients compared to healthy individuals. Our findings indicate that several bacterial genera can play a role in the fate of hydatid cysts in patients at different stages of the disease.

Echinococcus ortleppi infection in humans: An emerging zoonosis in Asia.

Echinococcus ortleppi is the genotype G5 of Echinococcus granulosus sensu lato and is a zoonotic canine tapeworm of which larvae causes cystic diseases in domestic animals and also humans. While this species is highly endemic and widely spread in domestic animals, human infection is extremely rare and only sporadic, and thus, entire picture of human cystic echinococcosis due to infection with E. ortleppi is unclear. We have made an extensive literature review on the cases of E. ortleppi infection in humans and found a total of 19 cases from various places in the world with recent emergence in Asian countries.

A Case of Spinal Echinococcosis in a Japanese Woman Living in Tokyo: Diagnostic Challenges in Non-endemic Areas and Public Health Implications.

Echinococcosis, caused by Echinococcus spp., often affects the lungs and liver, and spinal involvement is rare. Echinococcus multilocularis is prevalent in Japan, particularly in Hokkaido. We herein report a rare case of spinal echinococcosis in a 31-year-old woman who was diagnosed in Tokyo. Spinal echinococcosis is uncommon and often leads to misdiagnoses. The patient likely contracted the disease via contaminated fresh produce transported from an endemic region. This study emphasizes the diagnostic challenges of spinal echinococcosis in non-endemic regions and highlights the public health concerns related to the spread of infections in non-endemic areas.

First confirmed case of hydatid disease of the male breast: A case report.

Introduction: Hydatid disease of the male breast has not been documented in literature to date. Case report: We present a case of a 46-year old male patient who presented with a painful breast lump for 1 year. On imaging, it was suspicious for hydatid disease of the breast and was successfully diagnosed preoperatively on fine-needle aspiration cytology and histopathology as hydatid disease of the breast. Discussion: Incidence of hydatid disease of the breast contributes to a minute number of total cases of hydatid disease. Male breast hydatid disease was unknown prior to this report. We did find similarities in the imaging features of our case and the ones described for female breasts. On ultrasonography, we found it analogous to the 'water-lily' sign which has been described in hepatic hydatic cysts. Conclusion: Keeping hydatid disease as a differential in the setting of a suspicious breast mass even in a male patient proved to be rewarding as it negated the need for further cross-sectional imaging and a definitive diagnosis could be sought based on the fine-needle aspiration cytology and histopathological findings.

Proton Nuclear Magnetic Resonance (1H NMR) Metabolomics Study in Serum, Urine, and Cystic Fluid for Differentiating Fertility and Staging of Intra-abdominal Hydatid Cyst in Adults.

Background: Cystic echinococcosis (CE) is a parasitic zoonosis caused by the tapeworm Echinococcus granulosus. Over the past few years, a lot of research has been done on liver illnesses using metabolomics techniques to identify biomarkers which could identify the diseases in its early stages. The present study was done to explore biomarkers in serum, urine, and cystic fluid which would help in differentiating, staging, and assessing fertility of intra-abdominal hydatid cyst by using proton nuclear magnetic resonance (1H NMR) metabolomics. Materials and methods: In the study, 28 subjects (16 cases and 12 controls) were enrolled. Staging of hydatid cysts was performed using ultrasonography. In patients complying with case and control definition, blood, urine, and cystic fluid were collected for complete blood count, urine culture, Echinococcus IgG enzyme-linked immunosorbent assay (ELISA), and metabolomic analysis. The 17, 15, and 11 metabolites in serum, urine, and cystic fluid samples were quantified, respectively, to differentiate between case and control group. Results: In this study, we observed that there was a significant downregulation of succinate metabolite in urine samples of cases, down-regulation of five metabolites (isoleucine, valine, histidine, tyrosine and formate) and upregulation of alanine in cystic fluid of cases. Conclusion: Current study demonstrates that metabolomics can be used non-invasively for rapid diagnosis of CE. This is one of the very few studies, which used 1H NMR spectroscopy, to analyze the profile of metabolites in serum, urine, and cystic fluid in cases of CE and controls. How to cite this article: Raj N, Pandey A, Roy R, et al. Proton Nuclear Magnetic Resonance (1H NMR) Metabolomics Study in Serum, Urine, and Cystic Fluid for Differentiating Fertility and Staging of Intra-abdominal Hydatid Cyst in Adults. Euroasian J Hepato-Gastroenterol 2024;14(1):30-34.

A rare pathology in the neck: Hydatid cyst.

Neck cysts can be classified as congenital, infectious-inflammatory, and neoplastic. Hydatid disease is a parasitic infection caused by Echinococcus, is usually seen in the liver and lung and, is rare in the head and neck region even in endemic areas. If not treated, a life-threatening condition may be encountered. In this study, a case of hydatid cyst operated due to a cystic lesion with a diameter of approximately 8 cm in the neck was presented by reviewing the literature.

Comparative Analysis of Nanos and Ago Genes Expression in the Germinative Cells Isolated from Germinal Layer and the Neck Region of Echinococcus granulosus.

Background: We aimed to evaluate the differential expression of nanos and ago genes in the protoscoleces, germinal layer, the neck, and the sucker regions of adult Echinococcus granulosus. Methods: The study was conducted in 2018 at the Research Center for Hydatid Disease in Iran, Kerman University of Medical Sciences, Kerman, Iran. In the present study E. granulosus protoscoleces were cultured in a di-phasic medium to obtain strobilated worms. The strobilated worms were harvested and using a sterile razor blade, the neck region was separated. In the molecular study the neck sections were compared with the tissues derived from the suckers from the same worm. The primers were specifically designed for RT-qPCR on nanos and ago. The germinative cells were isolated from the cyst germinal layer and cultured in DMEM for further molecular studies. The Immunohisto-chemical profile was designed to explore the nature of nanos protein in the strobilated worms. Differences between and within groups were statistically assessed relative to the protoscoleces. Results: An increasing nanos gene expressions were found in sucker, neck, cells and germinal layer in comparison to the protoscoleces. The expression of ago gene was decreased in sucker, cell and germinal layer, and increased in the neck region in comparison to the protoscoleces. The results showed that both genes were expressed in all developmental stages of E. granulosus. Conclusion: nanos and ago genes were differentially expressed at different developmental stages of E. granulosus and may contribute to differentiation of the parasite.

Echinococcal disease can present with giant abdominal cysts at very young age: A case report.

Echinococcus granulosus larvae can cause cystic echinococcosis (CE, also known as hydatid disease) in humans. The latent phase of hydatid disease lasts for years as a result of the slow growth of the cysts, which only become symptomatic when they are large. Therefore, CE is seldomly seen in very young children. Here we present a 4-year-old boy with two giant asymptomatic abdominal cysts. Ultrasound was inconclusive in regard to the nature of the cysts and serology for echinococcosis was negative, rendering CE improbable also in view of the young age. Nevertheless, in the absence of other conclusive explanations, the patient was started on albendazole. A subsequent diagnostic percutaneous puncture with direct microscopy of cyst fluid revealed parasitological evidence of echinococcosis. This case report shows that CE can present with giant cysts also at very young age and should be considered as a possible diagnosis in all children with giant abdominal cysts.

Two hydatid cysts in the psoas muscle of a 40-year-old female: A rare case report.

INTRODUCTION AND IMPORTANCE: Hydatid disease can affect any human organ. The occurrence of hydatid cysts in psoas muscle is rare, accounting for only 1-3 % of cases, and even in endemic regions. However, the presence of multiple cysts is even more rare. CASE PRESENTATION: A 40-year-old female from a rural area presented with abdominal pain. Physical examination revealed tenderness in the right hypochondrium. Her medical history pulmonary hydatid cystectomy seven years ago. Radiology investigation showed a two-hydatid cyst within the left psoas major muscle. Cystectomy of two hydatid cyst was performed. The patient was prescribed albendazole at a dose of 15 mg/kg, and at the latest month's follow-up appointment, the patient did not experience any relapse. CLINICAL DISCUSSION: This case presents a rare instance of multiple hydatid cysts in the psoas muscle, highlighting the importance of considering atypical presentations in endemic regions. CONCLUSIONS: This case highlights the rarity of psoas major muscle hydatid cysts in a middle-aged woman from a rural area. Early recognition, accurate radiological assessment, and proper surgical treatment are crucial for positive outcomes and averting complications like infection, compression of nearby structures, or anaphylactic reactions.

Encysted Odyssey: A Clinical and Pictorial Analysis of Hydatid Cysts From Head to Toe.

INTRODUCTION: Cystic echinococcosis, a zoonotic disease caused by the larval form of Echinococcus granulosus, predominantly affects the liver and lungs, with humans acting as accidental hosts. METHODS: Our retrospective study at the Department of Radiology and Imageology, Nizam's Institute of Medical Sciences, included 187 histopathologically or serologically proven cases. The mean age of presentation was 49.4 years. RESULTS: Liver involvement was most prevalent, accounting for 83.4% (n=156) of cases, followed by sporadic involvement of other organs such as the mesentery, spleen, pancreas, thalamus, kidney, lung, spine, and omentum. Characteristic diagnostic features observed on imaging included peripheral calcifications in 33% of cases, internal septations in 25% (n=47), dense calcifications in 15% (n=29), daughter cysts in 6% (n=11), and floating membranes in 5% (n=10). Among hepatic lesions, 90% (n=141) were showing involvement of a single lobe. Notably, 78% (n=110) of lesions were limited to the right lobe, 21% (n=30) to the left lobe, and 1% (n=1) to the caudate lobe. The most affected hepatic segment was segment VIII, while the least common was segment I (caudate lobe). Complications were identified in 13% (n=25) of cases of hepatic hydatidosis. CONCLUSIONS: The findings of our study emphasize the systemic nature of E. granulosus infection which can affect various organs in the body. It also illustrates the invaluable insights imaging provides for timely and accurate diagnosis of hydatid disease.

A case report of definitive endoscopic management of a large right liver lobe hydatid cyst - A novel approach.

INTRODUCTION: Cystic echinococcosis is a public health concern worldwide and is endemic in rural communities in South Africa (Shaw et al., 2006). The management of hydatid liver disease is of vital socio-economic importance within the infected communities (Centers for Disease Control and Prevention [Internet]. Echinococcosis). Often, surgical intervention is needed, and this carries its own morbidity and economic burden in our low-to-middle income setting (Acta Trop., 2003). Definitive endoscopic management is rarely considered and offers an exciting option with decreased morbidity to the patient. PRESENTATION OF CASE: This is a case report of a 36-year-old male who presented with a large right lobe liver hydatid cyst causing abdominal discomfort and pain. He also described early satiety and weight loss with malaise. The symptoms had been present for approximately 8-months duration. The diagnosis of a hydatid liver cyst was made on positive serology and imaging (CE1). The disease was managed with medical treatment using a full course of albendazole initially and then endoscopic drainage into the duodenum using a cautery-enhanced lumen apposing metal stent. There has been no recurrence up to the present time and complete symptom and cyst resolution has been noted. DISCUSSION: Given the success of this unconventional management, this case report will help in providing a low-morbidity management option in this endemic disease in certain selected cases. It also provides in detail how to use this option as a definitive management pathway. CONCLUSION: This management option required dynamic thinking and a new application of a revolutionary technology which has changed endoscopic management of a variety of conditions.

Diagnostic Problem in a Child With an Isolated Renal Hydatid Cyst: A Case Report.

Cystic hydatid disease is a parasitic disease caused by the larvae of the small tapeworm Echinococcus granulosus. It is still a serious public health problem in endemic regions such as the Mediterranean basin, especially in the Balkans. Usually, the complaints caused by the cysts are non-specific and there are rarely abnormalities in routine laboratory tests. The most common is the involvement of the liver. The frequency of isolated kidney involvement, especially in a child, is uncommon. We describe a rare pediatric case of an isolated renal hydatid cyst presenting with a urinary tract infection-like clinical presentation, leading to misdiagnosis and delayed treatment.

Exploring rare locations of hydatid disease: a retrospective case series.

INTRODUCTION: Hydatid disease, endemic in Mediterranean countries, primarily affects the liver, but can manifest in diverse organs. Non-hepatic and non-pulmonary cysts often pose diagnostic challenges. This study examines patients with hydatid cysts in atypical locations. METHODS AND RESULTS: From 2013 to 2020, our center treated 250 echinococcosis patients, among whom 11 cases (4.4%) with hydatid disease in uncommon sites were retrospectively reviewed. The distribution of unusual cyst locations and their clinical implications are discussed. CONCLUSION: Diagnosing hydatid cysts in uncommon locations is a formidable challenge. Surgeons should always contemplate the prospect of an unconventional cyst location when encountering patients with cystic masses in endemic regions. Failing to consider this possibility could lead to unfavorable outcomes.

Comparison of cystectomy and lobectomy of lung hydatid cyst in pediatrics: a retrospective study.

Objective: Hydatid cyst is an endemic disease in Iran. The treatment of choice for paediatric lung hydatid cysts is surgical removal of the cyst. However, due to its high prevalence the risk of recurrence after the surgery, cystectomy with capitonnage, which preserves the lung tissue, is a favourable surgical approach compared to lobectomy. Herein, the authors compared the outcome of cystectomy and lobectomy of lung hydatid cysts. Methods and materials: This is a retrospective study conducted in the paediatric surgery department. Paediatric patients who had undergone surgery due to pulmonary hydatid cysts were enroled. The patients were divided into two groups including cystectomy and non-anatomic lobectomy. Then, the length of surgery, length of hospitalization, postoperative complications, and the time required to remove the chest tube were calculated in each group. Results: A total of 32 patients were enroled in this retrospective study. Age, sex, location, and size of cysts were not significantly different between the two groups. The duration of surgery in the lobectomy and cystectomy groups was 116.3±33.7 versus 116.1±28.2 min, respectively (P=0.53). Surgery complications including the need for blood transfusion, pneumothorax, need for bronchoscopy and atelectasis were not different between the study groups. The mean time for first chest tube removal was significantly different between the groups with the lobectomy group having a shorter time (P=0.02). The length of hospital and ICU stay were not different between the two surgical procedures. The time to remove the first chest tube was significantly higher in cystectomy compared to lobectomy (P=0.02). Conclusion: The complications and outcome of the cystectomy are comparable to the lobectomy technique. However, the cystectomy method has the advantage of preserving the lung tissue, therefore it's a favourable technique in endemic areas for hydatid cysts where reoperation may be indicated.

Extracorporeal Membrane Oxygenation (ECMO) Use as Supportive Therapy in a Patient With Acute Respiratory Distress Syndrome Due to Rupture of a Pulmonary Hydatid Cyst.

Pulmonary echinococcosis is a parasitic infection that accounts for 20% of the infected cases with echinococcosis. Patients may present after a cyst rupture associated with a variety of complications, including acute respiratory distress syndrome (ARDS). Extracorporeal membrane oxygenation (ECMO) is known as supportive therapy for patients with respiratory and cardiac failure, including ARDS associated with multiple causes. Parasitic infection associated with ARDS due to cyst rupture managed with ECMO as bridging to definitive surgical intervention is documented in two previous case reports only. Here, we are presenting a 21-year-old female with a pulmonary hydatid cyst complicated by ARDS and managed with ECMO.

Concurrent Abdominal and Mediastinal Hydatid Cystic Disease: A Rare Case.

Hydatid disease in humans is caused by accidental ingestion of Echinococcus in its larval form. It mostly affects the liver and lungs, but rarely the mediastinum and other areas as well. The diagnosis is mostly confirmed intraoperatively in cases of mediastinal disease. The mainstay of treatment in such cases is surgery. This is a case report of a rare finding of hydatid disease in mediastinum along with the abdomen and its surgical management.

Clinical Pattern of Pediatric Hydatid Disease.

Context: Literature regarding hydatid disease in children is sparse. Aims: To highlight the peculiarities in the clinical pattern of pediatric hydatid disease (PHD). Settings and Design/Materials and Methods: Data were collected retrospectively from all children aged <18 years who presented to our tertiary care institute from July 2021 to June 2023 with hydatid disease involving any organ. Statistical Analysis Used: Simple statistical analysis involving sums, means, averages, and percentages. Results: Four of the 10 cases (40%) involved the lung, while only 2 (20%) involved the liver. There were five females and four males with an age range of 2-17 years. Four of the cases had primary extrahepatic extrapulmonary hydatid disease (40%), two involving the pancreas, one in the rectouterine pouch, and one intracranial. Conclusions: The clinical pattern of PHD is different from that of adults. Pulmonary echinococcosis is more common than hepatic involvement. Primary extrahepatic extrapulmonary hydatid disease is also more common in children than previously thought. A cystic lesion anywhere in a child warrants a differential of hydatid disease.

Immunomodulatory effects of hydatid antigens on mesenchymal stem cells: gene expression alterations and functional consequences.

Background: Cystic echinococcosis, caused by the larval stage of Echinococcus granulosus, remains a global health challenge. Mesenchymal stem cells (MSCs) are renowned for their regenerative and immunomodulatory properties. Given the parasite's mode of establishment, we postulate that MSCs likely play a pivotal role in the interaction between the parasite and the host. This study aims to explore the response of MSCs to antigens derived from Echinococcus granulosus, the etiological agent of hydatid disease, with the hypothesis that exposure to these antigens may alter MSC function and impact the host's immune response to the parasite. Methods: MSCs were isolated from mouse bone marrow and co-cultured with ESPs, HCF, or pLL antigens. We conducted high-throughput sequencing to examine changes in the MSCs' mRNA expression profile. Additionally, cell cycle, migration, and secretory functions were assessed using various assays, including CCK8, flow cytometry, real-time PCR, Western blot, and ELISA. Results: Our analysis revealed that hydatid antigens significantly modulate the mRNA expression of genes related to cytokine and chemokine activity, impacting MSC proliferation, migration, and cytokine secretion. Specifically, there was a downregulation of chemokines (MCP-1, CXCL1) and pro-inflammatory cytokines (IL-6, NOS2/NO), alongside an upregulation of anti-inflammatory mediators (COX2/PGE2). Furthermore, all antigens reduced MSC migration, and significant alterations in cellular metabolism-related pathways were observed. Conclusion: Hydatid disease antigens induce a distinct immunomodulatory response in MSCs, characterized by a shift towards an anti-inflammatory phenotype and reduced cell migration. These changes may contribute to the parasite's ability to evade host defenses and persist within the host, highlighting the complex interplay between MSCs and hydatid disease antigens. This study provides valuable insights into the pathophysiology of hydatid disease and may inform the development of novel therapeutic strategies.

Case Report of a Hidden Intruder-Extremely Rare Presentation of Hydatidosis as a Nuchal Tumoural Mass.

Introduction: The parasitic tapeworm impersonated by the larvae of Echinococcus granulosus represents the aetiology of the hydatid pathology. The predilect site of invasion is the liver, but there are other cases of different localization all over the body, regardless of the type of invaded tissue. Soft tissue hydatidosis can be a real challenge for the clinician in terms of the diagnosis, and it might generate various complications such as anaphylactic shock. The aim of the present work is to illustrate a unique case of primary hydatidosis located in the nuchal region. Case Report: We report the case of a 68-year-old male patient, a zootechnic, who presented at the hospital with a tumoural mass (dimension: about 12/10 cm) located in the nuchal region. The complex approach needed consisted of surgical therapy along with histopathological confirmation of the diagnostic and antiparasitic medication, which led to a complete recovery with a low probability of recurrence. Discussion: Encounters with patients with primary soft tissue hydatidosis are exceptionally rare, but the surgeon must take into consideration this clinical diagnosis, especially for patients located in an endemic region with occupations that might have exposed the patient to this type of parasite.

Exploration of altered miRNA expression and function in MSC-derived extracellular vesicles in response to hydatid antigen stimulation.

Background: Hydatid disease is caused by Echinococcus parasites and can affect various tissues and organs in the body. The disease is characterized by the presence of hydatid cysts, which contain specific antigens that interact with the host's immune system. Mesenchymal stem cells (MSCs) are pluripotent stem cells that can regulate immunity through the secretion of extracellular vesicles (EVs) containing microRNAs (miRNAs). Methods: In this study, hydatid antigens were isolated from sheep livers and mice peritoneal cavities. MSCs derived from mouse bone marrow were treated with different hydatid antigens, and EVs were isolated and characterized from the conditioned medium of MSCs. Small RNA library construction, miRNA target prediction, and differential expression analysis were conducted to identify differentially expressed miRNAs. Functional enrichment and network construction were performed to explore the biological functions of the target genes. Real-time PCR and Western blotting were used for miRNA and gene expression verification, while ELISA assays quantified TNF, IL-1, IL-6, IL-4, and IL-10 levels in cell supernatants. Results: The study successfully isolated hydatid antigens and characterized MSC-derived EVs, demonstrating the impact of antigen concentration on MSC viability. Key differentially expressed miRNAs, such as miR-146a and miR-9-5p, were identified, with functional analyses revealing significant pathways like Endocytosis and MAPK signaling associated with these miRNAs' target genes. The miRNA-HUB gene regulatory network identified crucial miRNAs and HUB genes, such as Traf1 and Tnf, indicating roles in immune modulation and osteogenic differentiation. Protein-protein interaction (PPI) network analysis highlighted central HUB genes like Akt1 and Bcl2. ALP activity assays confirmed the influence of antigens on osteogenic differentiation, with reduced ALP activity observed. Expression analysis validated altered miRNA and chemokine expression post-antigen stimulation, with ELISA analysis showing a significant reduction in CXCL1 expression in response to antigen exposure. Conclusion: This study provides insights into the role of MSC-derived EVs in regulating parasite immunity. The findings suggest that hydatid antigens can modulate the expression of miRNAs in MSC-derived EVs, leading to changes in chemokine expression and osteogenic capacity. These findings contribute to a better understanding of the immunomodulatory mechanisms involved in hydatid disease and provide potential therapeutic targets for the development of new treatment strategies.