Fifty-five cases of hepatic alveolar echinococcosis combined with lymph node metastasis: A retrospective study.

BACKGROUND: Lymph node metastasis is a specific type of metastasis in hepatic alveolar echinococcosis (AE). Currently, there is a scarcity of describing the clinical characteristics and lymph node metastasis rules of patients with hepatic AE combined with lymph node metastasis and its mechanism and management are still controversial. Radical hepatectomy combined with regional lymph node dissection is a better treatment. AIM: To analyse the clinical features of hepatic AE combined with lymph node metastasis to explore its treatment and efficacy. METHODS: A total of 623 patients with hepatic AE admitted to the First Affiliated Hospital of Xinjiang Medical University from 1 January 2012 to 1 January 2022 were retrospectively analysed. Fifty-five patients with combined lymph node metastasis were analysed for their clinical data, diagnosis and treatment methods, follow-up efficacy, and characteristics of lymph node metastasis. Finally, we comparatively analysed the lymph node metastasis rates at different sites. Categorical variables are expressed as frequencies and percentages, and the analysis of difference was performed using the χ 2 test. The Bonferroni method was used for pairwise comparisons when statistical differences existed between multiple categorical variables. RESULTS: A lymph node metastasis rate of 8.8% (55/623) was reported in patients with hepatic AE, with a female predilection (69.1%) and a statistically significant sex difference (χ 2 = 8.018, P = 0.005). Of the 55 patients with lymph node metastasis, 72.7% had a parasite lesion, neighbouring organ invasion, and metastasis stage of P3N1M0 and above, of which 67.3%, 78.2%, and 34.5% of hepatic AE lesions invaded the bile ducts, blood vessels, and distant metastases, respectively. Detection rates of lymph node metastasis of 16.4%, 21.7%, and 34.2% were reported for a preoperative abdominal ultrasound, magnetic resonance imaging, and computed tomography examinations. All patients were intraoperatively suspected with enlarged lymph nodes and underwent radical hepatectomy combined with regional lymph node dissection. After surgery, a routine pathological examination was conducted on the resected lymph nodes. A total of 106 positive lymph nodes were detected in six groups at various sites, including 51 single-group metastasis cases and four multi-group metastasis cases. When the metastasis rates at different sites were statistically analysed, we observed that the metastasis rate in the para-hepatoduodenal ligament lymph nodes was significantly higher than that of the other sites (χ 2 = 128.089, P = 0.000 < 0.05). No statistical difference was observed in the metastasis rate between the five other groups. Clavien-Dindo grade IIIa complication occurred in 14 cases, which improved after administering symptomatic treatment. Additionally, lymph node dissection-related complications were not observed. Recurrence after 2 years was observed in one patient. CONCLUSION: Lymph node metastasis is a rare form of metastasis in hepatic AE, which is more frequent in women. Para-hepatoduodenal ligament lymph nodes are commonly observed. Radical hepatectomy combined with regional lymph node dissection is a safe, effective, and feasible treatment for liver AE combined with lymph node metastasis.

Liver autotransplantation and atrial reconstruction on a patient with multiorgan alveolar echinococcosis: a case report.

BACKGROUND: Alveolar echinococcosis (AE) primarily affects the liver and potentially spreads to other organs. Managing recurrent AE poses significant challenges, especially when it involves critical structures and multiple major organs. CASE PRESENTATION: We present a case of a 59-year-old female with recurrent AE affecting the liver, heart, and lungs following two previous hepatectomies, the hepatic lesions persisted, adhering to major veins, and imaging revealed additional diaphragmatic, cardiac, and pulmonary involvement. The ex vivo liver resection and autotransplantation (ELRA), first in human combined with right atrium (RA) reconstruction were performed utilizing cardiopulmonary bypass, and repairs of the pericardium and diaphragm. This approach aimed to offer a potentially curative solution for lesions previously considered inoperable without requiring a donor organ or immunosuppressants. The patient encountered multiple serious complications, including atrial fibrillation, deteriorated liver function, severe pulmonary infection, respiratory failure, and acute kidney injury (AKI). These complications necessitated intensive intraoperative and postoperative care, emphasizing the need for a comprehensive management strategy in such complicated high-risk surgeries. CONCLUSIONS: The multidisciplinary collaboration in this case proved effective and yielded significant therapeutic outcomes for a rare case of advanced hepatic, cardiac, and pulmonary AE. The combined approach of ELRA and RA reconstruction under extracorporeal circulation demonstrated distinct advantages of ELRA in treating complex HAE. Meanwhile, assessing diaphragm function during the perioperative period, especially in patients at high risk of developing pulmonary complications and undergoing diaphragmectomy is vital to promote optimal postoperative recovery. For multi-resistant infection, it is imperative to take all possible measures to mitigate the risk of AKI if vancomycin administration is deemed necessary.

Liver cystic echinococcosis: A retrospective study on the demographics and clinical profile of patients managed at a single tertiary institution in central Eastern Cape Province, South Africa.

BACKGROUND: Cystic echinococcosis (CE) is a zoonotic disease with an Africa-wide prevalence of 1.7%. CE is caused by the tapeworm Echinococcus granulosus sensu lato, with the liver being the most commonly affected organ. In South Africa (SA), there is a paucity of data on liver CE demographics and management. OBJECTIVES: To describe the demographics and clinical profile of patients with liver CE in a single tertiary hospital in Eastern Cape Province, SA. METHODS: A 4-year (2019 - 2022) retrospective clinical record review study was conducted on patients presenting with liver CE to the Department of Surgery at Frere Hospital. The demographics, clinical characteristics and management of patients with liver CE are reported. RESULTS: A total of 56 patients diagnosed with and managed for liver CE were included in the study. The mean age of the patients was 37.5 years. Abdominal pain (n=39; 69.6%) was the most common presenting symptom, and a palpable abdominal mass (n=36; 64.3%) was the predominant presenting sign. Most patients had disease confined to the liver (n=35; 62.5%). The right lobe of the liver was most commonly affected (n=38; 67.9%), and most patients had a single liver cyst on imaging (n=32; 57.1%). The majority of the patients (n=36; 64.3%) were managed with surgical partial cystectomy, with a perioperative bile leak being the most common complication. CONCLUSION: In our setting, liver CE contributes to a significant local burden of the disease. The disease often has a nonspecific clinical presentation, necessitating imaging for the diagnosis. We observed good short-term outcomes in patients managed with combined partial cystectomy and medical therapy, although there is a risk of perioperative bile leak.

Treatment of uncomplicated hepatic cystic echinococcosis (hydatid disease).

BACKGROUND: Cystic echinococcosis is a parasitic infection mainly impacting people living in low- and middle-income countries. Infection may lead to cyst development within organs, pain, non-specific symptoms or complications including abscesses and cyst rupture. Treatment can be difficult and varies by country. Treatments include oral medication, percutaneous techniques and surgery. One Cochrane review previously assessed the benefits and harms of percutaneous treatment compared with other treatments. However, evidence for oral medication, percutaneous techniques and surgery in specific cyst stages has not been systematically investigated and the optimal choice remains uncertain. OBJECTIVES: To assess the benefits and harms of medication, percutaneous and surgical interventions for treating uncomplicated hepatic cystic echinococcosis. SEARCH METHODS: We searched CENTRAL, MEDLINE, two other databases and two trial registries to 4 May 2023. We searched the reference lists of included studies, and contacted experts and researchers in the field for relevant studies. SELECTION CRITERIA: We included randomized controlled trials (RCTs) in people with a diagnosis of uncomplicated hepatic cystic echinococcosis of World Health Organization (WHO) cyst stage CE1, CE2, CE3a or CE3b comparing either oral medication (albendazole) to albendazole plus percutaneous interventions, or to surgery plus albendazole. Studies comparing praziquantel plus albendazole to albendazole alone prior to or following an invasive intervention (surgery or percutaneous treatment) were eligible for inclusion. DATA COLLECTION AND ANALYSIS: We used standard Cochrane methods. Our primary outcomes were symptom improvement, recurrence, inactive cyst at 12 months and all-cause mortality at 30 days. Our secondary outcomes were development of secondary echinococcosis, complications of treatment and duration of hospital stay. We used GRADE to assess the certainty of evidence. MAIN RESULTS: We included three RCTs with 180 adults and children with hepatic cystic echinococcosis. Two studies enrolled people aged 5 to 72 years, and one study enrolled children aged 6 to 14 years. One study compared standard catheterization plus albendazole with puncture, aspiration, injection and re-aspiration (PAIR) plus albendazole, and two studies compared laparoscopic surgery plus albendazole with open surgery plus albendazole. The three RCTs were published between 2020 and 2022 and conducted in India, Pakistan and Turkey. There were no other comparisons. Standard catheterization plus albendazole versus PAIR plus albendazole The cyst stages were CE1 and CE3a. The evidence is very uncertain about the effect of standard catheterization plus albendazole compared with PAIR plus albendazole on cyst recurrence (risk ratio (RR) 3.67, 95% confidence interval (CI) 0.16 to 84.66; 1 study, 38 participants; very low-certainty evidence). The evidence is very uncertain about the effects of standard catheterization plus albendazole on 30-day all-cause mortality and development of secondary echinococcosis compared to open surgery plus albendazole. There were no cases of mortality at 30 days or secondary echinococcosis (1 study, 38 participants; very low-certainty evidence). Major complications were reported by cyst and not by participant. Standard catheterization plus albendazole may increase major cyst complications compared with PAIR plus albendazole, but the evidence is very uncertain (RR 10.74, 95% CI 1.39 to 82.67; 1 study, 53 cysts; very low-certainty evidence). Standard catheterization plus albendazole may make little to no difference on minor complications compared with PAIR plus albendazole, but the evidence is very uncertain (RR 1.03, 95% CI 0.60 to 1.77; 1 study, 38 participants; very low-certainty evidence). Standard catheterization plus albendazole may increase the median duration of hospital stay compared with PAIR plus albendazole, but the evidence is very uncertain (4 (range 1 to 52) days versus 1 (range 1 to 15) days; 1 study, 38 participants; very low-certainty evidence). Symptom improvement and inactive cysts at 12 months were not reported. Laparoscopic surgery plus albendazole versus open surgery plus albendazole The cyst stages were CE1, CE2, CE3a and CE3b. The evidence is very uncertain about the effect of laparoscopic surgery plus albendazole on cyst recurrence in participants with CE2 and CE3b cysts compared to open surgery plus albendazole (RR 3.00, 95% CI 0.13 to 71.56; 1 study, 82 participants; very low-certainty evidence). The second study involving 60 participants with CE1, CE2 or CE3a cysts reported no recurrence in either group. The evidence is very uncertain about the effect of laparoscopic surgery plus albendazole on 30-day all-cause mortality in participants with CE1, CE2, CE3a or CE3b cysts compared to open surgery plus albendazole. There was no mortality in either group (2 studies, 142 participants; very low-certainty evidence). The evidence is very uncertain about the effect of laparoscopic surgery plus albendazole on major complications in participants with CE1, CE2, CE3a or CE3b cysts compared to open surgery plus albendazole (RR 0.50, 95% CI 0.13 to 1.92; 2 studies, 142 participants; very low-certainty evidence). Laparoscopic surgery plus albendazole may lead to slightly fewer minor complications in participants with CE1, CE2, CE3a or CE3b cysts compared to open surgery plus albendazole (RR 0.13, 95% CI 0.02 to 0.98; 2 studies, 142 participants; low-certainty evidence). Laparoscopic surgery plus albendazole may reduce the duration of hospital stay compared with open surgery plus albendazole (mean difference (MD) -1.90 days, 95% CI -2.99 to -0.82; 2 studies, 142 participants; low-certainty evidence). Symptom improvement, inactive cyst at 12 months and development of secondary echinococcosis were not reported. AUTHORS' CONCLUSIONS: Percutaneous and surgical interventions combined with albendazole can be used to treat uncomplicated hepatic cystic echinococcosis; however, there is a scarcity of randomised evidence directly comparing these interventions. There is very low-certainty evidence to indicate that standard catheterization plus albendazole may lead to fewer cases of recurrence, more major complications and similar complication rates compared to PAIR plus albendazole in adults and children with CE1 and CE3a cysts. There is very low-certainty evidence to indicate that laparoscopic surgery plus albendazole may result in fewer cases of recurrence or fewer major complications compared to open surgery plus albendazole in adults and children with CE1, CE2, CE3a and CE3b cysts. Laparoscopic surgery plus albendazole may lead to slightly fewer minor complications. Firm conclusions cannot be drawn due to the limited number of studies, small sample size and lack of events for some outcomes.

Complicated Liver Cystic Echinococcosis-A Comprehensive Literature Review and a Tale of Two Extreme Cases.

Cystic echinococcosis is a zoonotic parasitic disease that affects the liver in more than 70% of cases, and there is still an underestimated incidence in endemic areas. With a peculiar clinical presentation that ranges from paucisymptomatic illness to severe and possibly fatal complications, quality imaging and serological studies are required for diagnosis. The mainstay of treatment to date is surgery combined with antiparasitic agents. The surgical armamentarium consists of open and laparoscopic procedures for selected cases with growing confidence in parenchyma-sparing interventions. Endoscopic retrograde cholangiopancreatography (ERCP) is extremely useful for the diagnosis and treatment of biliary fistulas. Recent relevant studies in the literature are reviewed, and two complex cases are presented. The first patient underwent open surgery to treat 11 liver cysts, and during the follow-up, a right pulmonary cyst was diagnosed that was treated by minimally invasive surgery. The second case is represented by the peritoneal rupture of a giant liver cyst in a young woman who underwent laparoscopic surgery. Both patients developed biliary fistulas that were managed by ERCP. Both patients exhibited a non-specific clinical presentation and underwent several surgical procedures combined with antiparasitic agents, highlighting the necessity of customized treatment in order to decrease complications and successfully cure the disease.

Standardized approach to the conservative surgery of hepatic cystic echinococcosis: A prospective study.

OBJECTIVE: Surgery is the mainstay of hepatic cystic echinococcosis (HCE). The conservative surgery of HCE carries a non-negligible risk of recurrence and significant morbidity, dominated by Deep Surgical Site Infections (DSSI). To address these issues, we have improved and standardized this technique, which could reduce complications and achieve better postoperative outcomes. PATIENTS AND METHODS: We conducted a prospective study from June 2017 to June 2022 involving of patient operated using a standardized open technique for uncomplicated HCE at Habib Bourguiba University Hospital, Sfax, Tunisia. The aim was to obtain results at least similar to radical management in terms of DSSI. Patients with large cystobiliary fistulas or patients with complicated cysts were excluded. RESULTS: Fifty patients with 106 cysts were operated using the standardized technique comprising of liver mobilization, intraoperative ultrasound, systematic methylene blue injection to detect cystobiliary fistulas and omentoplasty. The median age of the patients was 44(semi-interquartile range: 16) years. The main symptom described by the patient was pain in 43 cases (86%). An abnormal liver test was found in 20 cases (40%). On imaging studies, the cyst had a median size of 7.4(3.0) cm. Cyst of the hepatic dome accounted for 38 cases (35.8%) with most cysts being situated in the right hemi-liver. Visual inspection of the cavity and Methylene blue testing allowed for the discovery of 57 cysts (53.7%) that had cystobiliary fistulas that were sutured. Omentoplasty was performed in 77 cysts (72.6%). Postoperatively, only 2 cases (1.9%) developed a DSSI in the form of an external bile leak with resolved with conservative management. No case of recurrence was found after a median follow-up of 24 months. CONCLUSION: The standardized conservative surgical technique, in selected patients, shows promise in reducing DSSI rates and overall morbidity, and achieve as equally good result as radical management.

The role of endoscopic retrograde cholangiopancreatography in the treatment of hepatic cystic Echinococcus in a high HIV prevalence population: a retrospective cohort study.

BACKGROUND: Endoscopic retrograde cholangiopancreatography (ERCP) is a useful, minimally invasive intervention in managing complicated hepatic cystic echinococcosis (HCE). This study aims to assess the use of ERCP in a South African HCE cohort with and without human immunodeficiency virus (HIV) co-infection. METHODS: An analysis was performed of patients with HCE who were assessed for surgery and underwent ERCP at a tertiary hospital in South Africa between 2011 and 2023. Demographics, clinical data, imaging characteristics, operative management, and postoperative complications were compared between HIV-negative (HIV-) and HIV-positive (HIV+) cohorts. RESULTS: Of the 91 patients assessed, 45 (mean age 34.6 years, 73.3% females, 23 HIV+) required ERCP. HIV status did not significantly affect cyst characteristics or surgical outcomes. HIV+ patients had a higher incidence of intraoperative bile leaks (p = 0.025). There were 18 patients who underwent preoperative ERCPs, mainly for biliary-cyst complications primarily causing obstructive jaundice. A total of 40 patients required postoperative ERCPs, mainly for bile leaks. There were no ERCP-related mortalities and only one case of pancreatitis. ERCP success rates were comparable in both cohorts, with an overall success rate of 86.7%. CONCLUSION: HIV co-infection did not significantly impact the clinical course or outcomes of cystic echinococcosis (CE) patients undergoing ERCP. Perioperative ERCP proved effective in managing biliary complications of HCE as well as postoperative complications, regardless of HIV status. This study underscores the importance of endoscopic interventions in the comprehensive management of CE.

Does HIV co-infection promote Echinococcus dissemination?

SUMMARY: The influence of human immunodeficiency virus (HIV) on the severity of hepatic cystic echinococcosis (CE) is uncertain. HIV-modulated immune suppression may increase the risk of contracting CE with less self-limiting disease, more rapid progression, and a higher likelihood of complications. A 30-year-old male with concurrent, untreated HIV underwent surgery for two large, complicated hepatic CE cysts, which were replacing the right hemiliver, and innumerable peritoneal daughter cysts. At operation, 30 kg of cystic material was removed from the liver and peritoneal cavity. Despite postoperative complications, including cardiac arrest, respiratory failure, and a bile leak, the patient made a full recovery.

Whole-transcriptome sequencing and bioinformatics analysis of calcified outer-layer tissue from cystic echinococcosis.

Echinococcosis is a common zoonotic disease in livestock; the type with the highest incidence is cystic echinococcosis (CE). In clinical management, patients with CE of the liver in which the cyst wall is calcified have been found to have better prognoses than those without calcification. In this study, we collected calcified and uncalcified cyst wall tissue from patients with hepatic CE and observed significant changes in the expression of 2336 messenger ribonucleic acids (mRNAs), 178 long noncoding RNAs (lncRNAs), 210 microRNAs (miRNAs), and 33 circular RNAs (circRNAs) using high-throughput sequencing (HTS). Gene ontology (GO) and Kyoto Encyclopedia of Genes and Genomes (KEGG) enrichment analyses of differentially expressed RNAs (DERNAs: DEmRNAs, DElncRNAs, DEmiRNAs, and DEcircRNAs) were performed to explore these RNAs' potential biological functions and signaling pathways. Ultimately, the results of hematoxylin and eosin (H&E) and terminal deoxynucleotidyl transferase deoxyuridine triphosphate (dUTP) nick end labeling (TUNEL) staining confirmed the correlation between calcification and apoptosis of the cyst wall. In summary, this study was an initial exploration of the molecular-biological mechanism underlying spontaneous calcification of the hydatid cyst wall, and it provides a theoretical basis for exploring new targets for drug treatment in CE.

Comparative analysis of host immune responses to Hydatid cyst in human and ovine hepatic cystic Echinococcosis.

BACKGROUND: Hydatid disease is caused by the larval stages of the canine tapeworm Echinococcus granulosus. It is one of the most critical helminthic diseases, representing worldwide public health and socio-economic concern. AIM: This study aimed to investigate the expression of apoptosis and immune response within hepatic tissues of humans and sheep infected with the Hydatid cyst. METHODS: Paraffin-embedded tissue was prepared from each tissue sample and used for histopathological examination by Haematoxylin- Eosin. Also, toluidine blue staining was used for mast cell detection, while an immunohistochemical study was performed to assess CD3 T lymphocytes, CD4 helper T lymphocytes, CD8 cytotoxic T lymphocytes, CD20 memory B lymphocytes, CD68 macrophage, and caspase-3 antibodies. RESULTS: The histological examination revealed significant changes, including the infiltration of inflammatory cells, predominantly lymphocytes with scattered giant cells, necrotic hepatic tissue, and fibrosis. Toluidine blue stain revealed a higher number of mast cells (5 cells/field) in humans compared to sheep (3.6 cells/field). The immunohistochemical analysis confirmed that the CD3 were the most predominant inflammatory cell in the hepatic tissue of humans (intensive 70%), and sheep (moderate 38.47%). Caspase-3 was observed in all samples in different grades and mostly in human liver tissue. CONCLUSION: This data could aid in recognizing immunological markers for differentiating disease progression, as well as enhance the understanding of local immune responses to cystic Echinococcosis (CE). The findings could provide preliminary data for future studies on immune responses associated with Hydatid cysts.

Disasters that develop in the liver due to hydatid cyst: a case report.

Hilar cavernous transformation is the formation of venous structures rich in collateral around the portal vein. Portal vein thrombosis is a rare entity. Although there are many reasons for its etiology, few cases have been reported secondary to hydatid cysts in the liver. Here, we present a 24-year-old patient with complaints of abdominal pain and swelling. Her CT and MRI scans show cholelithiasis with portal vein thrombosis and hilar cavernous transformation due to giant hydatid cyst compression in the lateral liver sector.

La transformación cavernosa hiliar es la formación de estructuras venosas ricas en colaterales alrededor de la vena porta. La trombosis de la vena porta es una afección poco frecuente. Aunque existen muchas razones en su etiología, se han descrito pocos casos secundarios a quiste hidatídico en el hígado. Aquí se presenta el caso de una paciente de 24 años con quejas de dolor abdominal e hinchazón. La tomografía computarizada y la resonancia magnética mostraron colelitiasis con trombosis de la vena porta y transformación cavernosa hiliar por compresión del quiste hidatídico gigante en el sector lateral del hígado.

Clinical Application of Artificial Intelligence in the Ultrasound Classification of Hepatic Cystic Echinococcosis.

Hepatic cystic echinococcosis (HCE) is a zoonotic disease that occurs when the larvae of Echinococcus granulosus parasitize the livers of humans and mammals. HCE has five subtypes, and accurate subtype classification is critical for choosing a treatment strategy. To evaluate the clinical utility of artificial intelligence (AI) based on convolutional neural networks (CNNs) in the classification of HCE subtypes via ultrasound imaging, we collected ultrasound images from 4,012 HCE patients at the First Affiliated Hospital of Xinjiang Medical University between 2008 and 2020. Specifically, 1,820 HCE images from 967 patients were used as the training and validation sets for the construction of the AI model, and the remaining 6,808 images from 3,045 patients were used as the test set to evaluate the performance of the AI models. The 6,808 images were randomly divided into six groups, and each group contained equal proportions of the five subtypes. The data of each group were analyzed by a resident physician. The accuracy of HCE subtype classification by the AI model and by manual inspection was compared. The AI HCE classification model showed good performance in the diagnosis of subtypes CE1, CE2, CE4, and CE5. The overall accuracy of the AI classification (90.4%) was significantly greater than that of manual classification by physicians (86.1%; P <0.05). The CNN can better identify the five subtypes of HCE on ultrasound images and should help doctors with little experience in more accurately diagnosing HCE.

Liver resection with two-step vascular exclusion, in situ hypothermic portal perfusion for the treatment of end-stage hepatic alveolar echinococcosis.

PURPOSE: To evaluate the safety and efficacy of two-step vascular exclusion and in situ hypothermic portal perfusion in patients with end-stage hepatic hydatidosis. METHODS: This study involved patients with advanced hepatic hydatid disease undergoing surgical treatment between 2022 and 2023, which included resection and reconstruction of the hepatic veins, inferior vena cava (IVC), and portal vein (PV). We described the technical details of liver resection and vascular reconstruction, as well as the use of two-step vascular exclusion and in situ hypothermic portal perfusion techniques during the vascular reconstruction process. RESULT: We included 7 patients with advanced hepatic hydatid disease who underwent surgical resection using two-step vascular exclusion and in situ hypothermic portal perfusion. The mean duration of surgery was 12.5 h (range, 7.5-15.0 h). The average hepatic ischemia time was 45 min (range, 25-77 min), while the occlusion time of the IVC was 87 min (range, 72-105 min). The total blood loss was 1000 milliliters (range, 500-1250 milliliters). Postoperatively, patients exhibited good recovery of liver and renal function. The mean ICU stay was 2 days (range, 1-3 days), and the mean postoperative hospital stay was 13 days (range, 9-16 days), with no Grade III or above complications observed during a mean follow-up period of 15 months (range, 9-24 months), CONCLUSION: two-step vascular exclusion and in situ hypothermic portal perfusion for surgical resection of end-stage hepatic hydatid disease is safe and effective. This significantly reduces the anhepatic time.

[Atypical alveolar echinococcosis with systemic involvement in a patient treated with dupilumab]. / Une échinococcose alvéolaire atypique d'évolution systémique chez une patiente traitée par dupilumab.

INTRODUCTION: Alveolar echinococcosis is an endemic parasitic disease prevalent in certain cold regions of the Northern Hemisphere, including Eastern France, Switzerland, Germany, Canada, and the United States. Widely underdiagnosed, it is associated with infection by Echinococcus multilocularis, a small tapeworm belonging to the cestode class, capable of causing multi-systemic involvement, particularly in elderly or immunocompromised patients. CASE REPORT: We present the case of an 82-year-old patient, immunocompromised due to prolonged corticosteroid therapy and treatment with dupilumab. She was referred to our department for a diagnostic assessment of atypical hepatic and pulmonary lesions, initially suspected of tuberculosis or an IgG4-related disease. The hypothesis of alveolar echinococcosis caused by E. multilocularis was eventually considered based on a set of arguments, further confirmed by molecular diagnosis. We discuss the role of dupilumab in the systemic evolution and atypical presentation of the disease, through the induction of a specific immunosuppression. CONCLUSION: Alveolar echinococcosis should be systematically considered in case of systemic disease with prominent hepatic and pulmonary involvement, especially in immunocompromised patients.

Intrabiliary Rupture of a Hepatic Hydatid Cyst: A Case Report.

BACKGROUND Hydatid disease is a common parasitic infection in many areas of Asia, South America, and Africa. It can affect any organ, most commonly the liver. The hydatid is often asymptomatic and the diagnosis is made when complications arise. The most common complication of this disease is opening in the bile ducts, which is a life-threatening condition causing serious acute cholangitis. We report a case of acute cholangitis caused by hydatid cyst rupture into the right bile duct. CASE REPORT A 33-year-old woman, with no medical or surgical history, presented to our Emergency Department with abdominal pain, jaundice, and fever for 3 days prior to admission. The patient was hemodynamically stable. In the examination, we noticed right upper-quadrant tenderness with guarding, icterus sclera, and negative Murphy sign. A CT scan showed a liver hydatid cyst of the 4th and 8th of segments, with intrahepatic and extrahepatic biliary duct dilation. The cyst communicated with the right hepatic bile duct via a large fistula. A diagnosis of acute cholangitis was made and she underwent conservative treatment with external drainage of the pericystic cavity through the biliary duct. The postoperative course was uncomplicated and she was discharged 15 days later. CONCLUSIONS The surgical approach to hepatic hydatid must be customized based on the specific characteristics of the cyst and associated complications. Acute hydatid cholangitis is a rare but serious complication of a hydatid cyst, which requires early diagnosis and adequate surgical management.

Ultrasonography screening of hepatic cystic echinococcosis in sheep flocks used for evaluating control progress in a remote mountain area of Hejing County, Xinjiang.

BACKGROUND: Although ultrasonography (US) has been widely used in the diagnosis of human diseases to monitor the progress of cystic echinococcosis (CE) control, the screening method for hepatic CE in sheep flocks requires adjustment. In this study, we used a US scanner to screen sheep flocks and evaluated the efficacy of dosing dogs once a year with praziquantel for 7 years from 2014 to 2021. METHODS: All sheep in the three flocks were screened using an ultrasound scanner in 2014 and compared with the prevalence of infection in 2021 in Bayinbuluke, Xinjiang, China. Sheep age was determined using incisor teeth. Cyst activity and calcification were determined using US images. The dogs were dewormed with praziquantel once a year to control echinococcosis in the community. RESULTS: Three flocks had 968 sheep in 2014, with 13.22%, 22.62%, 18.7%, 27.27%, 11.88%, and 6.3% of sheep aged 1, 2, 3, 4, 5, and ≥ 6 years old, respectively. US scanning revealed that the overall CE prevalence was 38.43% (372/968), with active cysts and calcified cysts present in 9.40% (91/968) and 29.02% (281/968) of the sheep, respectively. For the young sheep aged 1 and 2 years, the prevalence of active and calcified cysts was: 1.56% and 0.91%, and 10.94% and 18.72%, respectively. Approximately 15.15% and 16.52% of the 4- and 5-year-old sheep, respectively, harbored active cysts. There was no significant difference in the infection rates of sheep between 2014 and 2021 (P > 0.05). CONCLUSIONS: US is a practical tool for the field screening of CE in sheep flocks. One-third of the sheep population in the flocks was 1-2 years old, and these sheep played a very limited role in CE transmission, as most of the cysts were calcified. Old sheep, especially culled aged sheep, play a key role in the transmission of CE. Dosing dogs once a year did not affect echinococcosis control.

Comparative proteomics analysis of samples from hepatic cystic echinococcosis patients using data-independent acquisition approach.

Cystic echinococcosis is a zoonotic disease resulting from infection caused by the larval stage of Echinococcus granulosus. This study aimed to assess the specific proteins that are potential candidates for the development of a vaccine against E. granulosus. The data-independent acquisition approach was employed to identify differentially expressed proteins (DEPs) in E. granulosus samples. The Gene Ontology (GO) and Kyoto Encyclopedia of Genes and Genomes (KEGG) pathway enrichment analysis was employed to identify several noteworthy proteins. Results: The DEPs in E. granulosus samples were identified (245 pericystic wall vs. parasite-free yellowish granuloma (PYG, 1725 PY vs. PYG, 2274 PN vs. PYG). Further examination of these distinct proteins revealed their predominant enrichment in metabolic pathways, amyotrophic lateral sclerosis, and neurodegeneration-associated pathways. Notably, among these DEPs, SH3BGRL, MST1, TAGLN2, FABP5, UBE2V2, and RARRES2 exhibited significantly higher expression levels in the PYG group compared with the PY group (P < 0.05). The findings may contribute to the understanding of the pathological mechanisms underlying echinococcosis, providing valuable insights into the development of more effective diagnostic tools, treatment modalities, and preventive strategies. SIGNIFICANCE: CE is a major public health hazard in the western regions of China, Central Asia, South America, the Mediterranean countries, and eastern Africa. Echinococcus granulosus is responsible for zoonotic disease through infection Our analysis focuses on the proteins in various samples by data-dependent acquisition (DIA) for proteomic analysis. The importance of this research is to develop new strategies and targets to protect against E. granulosus infections in humans.

Applying LASSO logistic regression for the prediction of biliary complications after ex vivo liver resection and autotransplantation in patients with end-stage hepatic alveolar echinococcosis.

BACKGROUND: The purpose of this study was to explore the relevant risk factors associated with biliary complications (BCs) in patients with end-stage hepatic alveolar echinococcosis (HAE) following ex vivo liver resection and autotransplantation (ELRA) and to establish and visualize a nomogram model. METHODS: This study retrospectively analysed patients with end-stage HAE who received ELRA treatment at the First Affiliated Hospital of Xinjiang Medical University between August 1, 2010 and May 10, 2023. The least absolute shrinkage and selection operator (LASSO) regression model was applied to optimize the feature variables for predicting the incidence of BCs following ELRA. Multivariate logistic regression analysis was used to develop a prognostic model by incorporating the selected feature variables from the LASSO regression model. The predictive ability, discrimination, consistency with the actual risk, and clinical utility of the candidate prediction model were evaluated using receiver operating characteristic (ROC) curves, calibration plots, and decision curve analysis (DCA). Internal validation was performed by the bootstrapping method. RESULTS: The candidate prediction nomogram included predictors such as age, hepatic bile duct dilation, portal hypertension, and regular resection based on hepatic segments. The model demonstrated good discrimination ability and a satisfactory calibration curve, with an area under the ROC curve (AUC) of 0.818 (95% CI 0.7417-0.8958). According to DCA, this prediction model can predict the risk of BCs occurrence within a probability threshold range of 9% to 85% to achieve clinical net benefit. CONCLUSIONS: A prognostic nomogram with good discriminative ability and high accuracy was developed and validated to predict BCs after ELRA in patients with end-stage HAE.

A Rare Autochthonous Case of Hepatic Hydatid Cyst in the Non-Endemic Region of Taiwan.

BACKGROUND Hepatic hydatid cyst disease, caused by the parasite Echinococcus granulosus, is endemic in certain rural areas of the world, but not in most of East Asia outside Mainland China. In Taiwan, only one autochthonous case has been reported over the past 40 years. We present the case of an urban 91-year-old female patient without international travel history for more than 40 years. CASE REPORT The 91-year-old woman who used a wheelchair came to the Emergency Department reporting melena for 2 days and 1 episode of coffee-grounds vomitus. Epigastric tenderness was present. An incidental finding of elevated liver enzymes along with the clinical picture prompted further survey. Computed tomography revealed a 14×10×12 cm homogeneous cystic lesion in the right hepatic lobe with a partially calcified wall. On sonograms, a similar lesion was found, and the pathognomic "water-lily" sign was visualized along with the isoechoic-to-hypoechoic internal septa, fulfilling the diagnosis despite the patient's refusal to undergo magnetic resonance imaging studies and invasive definite diagnostic procedures. Although anthelmintic chemotherapy and invasive therapeutic measures were also refused, her symptoms improved and was not recurrent under supportive measures. However, the cyst was still present 12 months after discharge. CONCLUSIONS The case highlights that in areas with few cases of hepatic hydatid disease, the accurate diagnosis could be missed in patients without a significant epidemiological history. However, once imaging findings, especially those that are pathognomic, are appropriately interpreted on at least 2 imaging modalities, such cases could be diagnosed without further definitive studies.