A 38-year-old female was admitted due to recurrent right upper quadrant (RUQ) abdominal pain. Three years prior to admission, the patient complained of a sudden onset of RUQ pain radiating to the right flank area. A month after the onset of pain, she consulted a physician, and she was advised to undergo endoscopy, but she did not comply. Three months after the consultation, an abdominal ultrasound was done, revealing a simple hepatic cyst. She then underwent aspiration of the cyst. However there was no resolution of the pain. A contrast-enhanced abdominal computed tomography (CT) scan revealed a recurrence of the hepatic cyst. Subsequently, she underwent laparoscopic fenestration of the cyst in liver segments VI, VII, and VIII. The biopsy results confirmed that the hepatic cyst was a hydatid cyst caused by Echinococcus granulosus. A month after the procedure, the abdominal pain recurred, and a repeat CT scan revealed another recurrence of the cyst. The patient was advised to undergo open surgery, but she did not consent. The patient denied taking anthelmintic drugs in the past. She did not experience any other symptoms—such as jaundice, nausea, vomiting, anorexia, or weight loss—along with the RUQ pain. She previously worked in Lebanon as a domestic helper for seven years and then moved to Taiwan, where she was employed for three years for the same work. She denied any direct exposure to dogs or sheep. On physical examination, she had a non-tender, firm, palpable mass in the RUQ area of the abdomen measuring 4x4 cm. The rest of the physical examination findings were unremarkable. A contrast-enhanced CT scan of the whole abdomen done two months prior to her admission showed multiple, well-defined, hypodense lesions in the right hepatic lobe. These lesions exhibited mildly enhancing walls and internal septations with rosette or honeycomb appearance. The two largest lesions seen in hepatic segments V and VII measured 8.6 x 6.1 x 5.2 cm and 9.5 x 8.5 x 7.4 cm, respectively (Figure 1). Similar hypodense lesions were seen in the right retroperitoneal space. At least two lesions were visible in the right perirenal space, measuring 8.8 x 6.1 x 6.5 cm and 6.1 x 5.9 x 3.8 cm, and at least two other lesions were visible in the right anterior pararenal space, within the region of the distal ascending colon, measuring 6.0 x 5.5 x 5.4 cm and 7.9 x 6.4 x 5.1 cm (Figure 2). At this point, we diagnosed the patient as having a recurrent hydatid cyst in the right hepatic lobe, with intraperitoneal extension in the right perirenal space and right anterior pararenal space, based on the CT scan findings. After securing medical clearance and administering mebendazole prophylactically at a dosage of 40 mg/kg/day, taken 3 times a day for 7 days prior to surgery, we aimed to sterilize the hydatid cysts and prevent surgical contamination.1 We did an exploratory laparotomy through a reverse L (Makuuchi) skin incision on the right upper abdominal quadrant (Figure 3). We subsequently performed a layered dissection down to the peritoneum. Intraoperatively, we noted adhesions at the posterior segments of the liver, right diaphragm and mid transverse colon. We then meticulously performed adhesiolysis. With extreme caution, we ensured to prevent any spillage of the cystic contents into adjacent structures and the abdominal cavity. Then, we covered the abdomen with abdominal packs soaked in 95% ethanol. Intraoperative liver ultrasonography revealed only one cystic lesion involving hepatic segments V, VI, and VII. The cyst was adherent to the right subdiaphragmatic area. We also noted multiple intraperitoneal cysts: one adherent to the right perirenal area measuring 8 x 6 cm, another adherent to the distal ascending colon and the mid transverse colon measuring 7 x 6 cm, and a third one non-adherent and floating in the anterior pararenal area measuring 4 x 4 cm. Before excising the intrahepatic mother cyst, we aspirated approximately 30-40 ml of cyst fluid and infused the cyst with an equivalent amount of 95% ethanol. After 15 minutes, we reaspirated the infused ethanol. We first carefully excised the intraperitoneal extensions of the cyst, (Figure 4) starting with the cyst in the distal ascending colon, then the cyst in the anterior pararenal area, and finally the right perirenal cyst, taking extreme caution to avoid rupturing the cyst. We then proceeded with the dissection of the hepatoduodenal ligament, where the portal triad is located, to provide access for our Pringle Maneuver. This step was followed by adhesiolysis at the right subdiaphragmatic area. Because of dense adhesions, there was a diaphragmatic injury, for which we performed phrenicorrhaphy. We then mobilized the right hepatic lobe through careful dissection of the triangular and coronary ligaments. The margins were marked under ultrasound guidance and the Pringle Maneuver was applied intermittently. We then carried out a parenchymal-sparing resection of segments V, VI, and VII. After securing hemostasis, we placed a Jackson Pratt drain in the subhepatic area. At the end of the surgery, we successfully resected hepatic segments V, VI, and VII, which contained an intrahepatic mother cyst with multiple spherical daughter cysts. Additionally, we excised three secondary cysts from the intraperitoneal area. Grossly, the excised specimen showed the right posterior segment of the liver with a surgically resected yellowish multiseptated cyst attached to it. This cyst has a fibrous rim and it contains several variable-sized daughter cysts (Figure 5). The excised intraperitoneal cyst from the distal ascending colon to mid transverse colon area (Figure 6), has a thick wall with a laminated external layer (Figure 7). The histopathological examination of the hydatid cyst showed E. granulosus protoscolices containing four acetabula and an armed rostellum with 30 to 36 hooks (Figure 8) . Sections of the liver showed liver parenchyma that was infiltrated by mononuclear cells. Adjacent to the liver parenchyma, the hydatid cyst wall contains an outer acellular laminated membrane, a germinal membrane, and remnants of the E. granulosus protoscolices, which resembled grains of sand (Figure 9 and 10). Postoperatively, the patient complained of dyspnea and tachypnea. On chest physical examination, we noted decreased breath sounds on the right. A chest x-ray done revealed a right-sided pneumothorax. Pneumothorax can occur during hepatectomy due to the escape of intraperitoneal carbon dioxide gas into the pleural cavity.2 We performed a chest tube thoracostomy insertion, and removed the tube after 5 days, once we observed full expansion of the patient’s right lung. We discharged the patient on the 9th postoperative day and prescribed oral antibiotics and pain relievers as home medications. Cystic echinococcosis (CE) or hydatid disease is a zoonotic parasitic disease caused by infection with the larval stage of the tapeworm E. granulosus. Dogs and other carnivores are definitive hosts, while sheep, cattle, and goats are intermediate hosts. Humans are accidental, intermediate hosts and become secondarily infected by ingesting food or water contaminated with eggs or gravid proglottids that are excreted in the definitive host’s feces. CE is endemic in North and East Africa, South America, China and Central Asia, and the Mediterranean countries.3 4 5 6 This disease has a low prevalence in Southeast Asia, particularly the Philippines.7 In our case, the patient probably acquired the infection when she worked in Lebanon, where echinococcosis is endemic. The liver and the lungs are the most common sites of the disease in humans. The concomitant occurrence of the cyst in the retroperitoneum or other intra-abdominal spaces is extremely rare,8 9 and it is associated with a high mortality and disability rate.10 Abdominal CT scan is a good imaging technique for visualizing wall calcifications in CE,11 and in our patient, enhancing walls and internal septations, which represent the walls of daughter cysts, were seen. Peritoneal seeding or cyst dissemination can occur due to a previous hepatic hydatid cyst surgery or after a spontaneous or traumatic rupture.12 These secondary cysts are formed from the liberated protoscolices caused by rupture of the daughter cysts.13 Although rare, when a cyst ruptures into the peritoneal cavity, it can lead to reactions ranging from mild hypersensitivity reaction to potentially fatal anaphylactic shock.14 A preoperative course of oral albendazole or mebendazole, as in the case of our patient’s medication, sterilizes the cysts and reduces their tension, thereby facilitating the surgery.15 16 It also kills most of the protoscolices within the hepatic hydatid cyst.17 Postoperatively, these drugs significantly reduce the risk of cyst recurrence.16 The prognosis of CE is generally good, and a complete cure is possible with total surgical excision without spillage.18 For hepatic echinococcosis, it is crucial to prioritize radical surgical resection in the initial surgery and preoperative anthelmintic therapy to minimize morbidity and recurrence often linked with conservative surgery.16 19
Cysts ; Echinococcus granulosus

The larval stages of the cestode parasites belonging to the genus Echinococcus grow within internal organs of humans and a range of animal species. The resulting diseases, collectively termed echinococcoses, include major neglected tropical diseases of humans and livestock. Echinococcus larvae are outwardly protected by the laminated layer (LL), an acellular structure that is unique to this genus. The LL is based on a fibrillar meshwork made up of mucins, which are decorated by galactose-rich O-glycans. In addition, in the species cluster termed E. granulosus sensu lato, the LL features nano-deposits of the calcium salt of myo-inositol hexakisphosphate (Insp6). The main purpose of our article is to update the immunobiology of the LL. Major recent advances in this area are (i) the demonstration of LL "debris" at the infection site and draining lymph nodes, (ii) the characterization of the decoy activity of calcium Insp6 with respect to complement, (iii) the evidence that the LL mucin carbohydrates interact specifically with a lectin receptor expressed in Kupffer cells (Clec4F), and (iv) the characterization of what appear to be receptor-independent effects of LL particles on dendritic cells and macrophages. Much information is missing on the immunology of this intriguing structure: we discuss gaps in knowledge and propose possible avenues for research.
Animals ; Calcium ; Echinococcosis/parasitology* ; Echinococcus/immunology* ; Echinococcus granulosus/immunology* ; Mucins

The information on mortality from echinococcosis is important not only for a better understanding of the severity of the disease, but also for evaluating the effectiveness of public health interventions. The aim of this research was to study the causes of mortality from echinococcosis. We have collected and analyzed the materials of 1,470 patients in 10 age - groups in the Republic of Armenia (from 2000 to 2016). To find out the causes of mortality from echinococcosis, we have analyzed the medical histories and protocols of postmortem examinations of 19 deaths from echinococcosis and 17 deaths due to other indirect causes not associated with the parasite. The average annual death rate from echinococcosis is 0.007 per 10,000 population, and the mortality is 1.29 (per 100 patients). The highest mortality occurs in people aged 70–79. Mortality from echinococcosis is also recorded among the unoperated children. The rupture of the parasitic cyst and hepatic insufficiency are major among the direct causes of mortality. Sometimes the hydatid cysts unrecognized during the life were first diagnosed at autopsy. Insufficient qualification of doctors in the field of helminthology, as well as the latent course of the disease or manifestation of minor symptoms in echinococcosis over a long period often led to medical errors. Further decline in mortality can be achieved by early diagnosis, timely hospitalization and treatment before the development of severe complications worsening the prognosis and outcomes of surgical intervention.
Armenia ; Autopsy ; Child ; Early Diagnosis ; Echinococcosis ; Echinococcus granulosus ; Echinococcus multilocularis ; Hepatic Insufficiency ; Hospitalization ; Humans ; Medical Errors ; Mortality ; Parasites ; Prognosis ; Public Health ; Rupture

Hydatid disease is Echinococcus granulosus infection in its larva form. It is endemic in many sheep- and cattle-raising countries throughout the world. In Korea seventeen cases have been reported but relapsed case is not. We report relapsed hydatid disease in pelvic cavity 10 years after primary surgery for Echinococcal cysts in the liver and pelvic cavity. The cysts were surgically removed and the patient was managed with postoperative adjuvant treatment with albendazole and followed with serologic test for anti-echinoccocus antibodies by ELISA.
Albendazole ; Antibodies ; Echinococcosis* ; Echinococcus ; Echinococcus granulosus ; Enzyme-Linked Immunosorbent Assay ; Humans ; Korea ; Larva ; Liver ; Serologic Tests

This investigation aimed to evaluate the differential expression of HoxB7 and notch genes in different developmental stages of Echinococcus granulosus sensu stricto. The expression of HoxB7 gene was observed at all developmental stages. Nevertheless, significant fold differences in the expression level was documented in the juvenile worm with 3 or more proglottids, the germinal layer from infected sheep, and the adult worm from an experimentally infected dog. The notch gene was expressed at all developmental stages of E. granulosus; however, the fold difference was significantly increased at the microcysts in monophasic culture medium and the germinal layer of infected sheep in comparison with other stages. The findings demonstrated that the 2 aforementioned genes evaluated in the present study were differentially expressed at different developmental stages of the parasite and may contribute to some important biological processes of E. granulosus.
Adult* ; Animals ; Biological Processes ; Dogs ; Echinococcus granulosus* ; Echinococcus* ; Humans ; Parasites ; Real-Time Polymerase Chain Reaction ; Sheep

Human hydatid disease (cystic echinococcosis, CE) is a chronic parasitic infection caused by the larval stage of the cestode Echinococcus granulosus. As the disease mainly affects the liver, approximately 70% of all identified CE cases are detected in this organ. Optical molecular imaging (OMI), a noninvasive imaging technique, has never been used in vivo with the specific molecular markers of CE. Thus, we aimed to construct an in vivo fluorescent imaging mouse model of CE to locate and quantify the presence of the parasites within the liver noninvasively. Drug-treated protoscolices were monitored after marking by JC-1 dye in in vitro and in vivo studies. This work describes for the first time the successful construction of an in vivo model of E. granulosus in a small living experimental animal to achieve dynamic monitoring and observation of multiple time points of the infection course. Using this model, we quantified and analyzed labeled protoscolices based on the intensities of their red and green fluorescence. Interestingly, the ratio of red to green fluorescence intensity not only revealed the location of protoscolices but also determined the viability of the parasites in vivo and in vivo tests. The noninvasive imaging model proposed in this work will be further studied for long-term detection and observation and may potentially be widely utilized in susceptibility testing and therapeutic effect evaluation.
Animals ; Cestoda ; Echinococcosis ; Echinococcus granulosus* ; Echinococcus* ; Fluorescence ; Humans ; In Vitro Techniques ; Liver ; Mice* ; Molecular Imaging ; Parasites

Echinococcosis is mainly prevalent in the agricultural and pastoral areas in the northwest of China, but it is relatively rare in Hunan Province. Here, we reported the clinical data of a case of echinococcosis in Hunan Province. The patient was an 11-year-old male, who sought treatment at the Second Xiangya Hospital of Central South University due to abdominal mass. According to the symptoms, signs, and laboratory examinations, he was initially diagnosed as "intra-abdominal mass" and "spleen cyst". Subsequently, he underwent abdominal massive occupying resection and splenectomy. Postoperative pathological examination revealed the cuticle and germinal layer of hydatid and protoscolex, which was consistent with characteristics of echinococcosis. In addition, the serological examination showed that the specific anti-hydatid IgG antibody was positive. Combined with the patient's condition, he was given praziquantel treatment. After a month of follow-up, the patient was asymptomatic.
Animals ; Child ; China ; Echinococcosis/surgery* ; Echinococcus granulosus ; Humans ; Male ; Splenectomy

Echinococcus granulosus sensu lato (s.l.) is a causative agent of cystic echinococcosis or cystic hydatid disease in humans and domestic and wild animals. The disease is a serious health problem in countries associated with poverty and poor hygiene practices, particularly in livestock raising. We introduced a practical algorism for genotyping the parasite, which may be useful to many developing countries. To evaluate the efficiency of the algorism, we genotyped 3 unknown strains isolated from human patients. We found that unknowns 1 and 3 were included in G1, G2, and G3 genotypes group and unknown 2 was included in G4 genotype (Echinococcus equinus) according to the algorisms. We confirmed these results by sequencing the 3 unknown isolates cox1 and nad1 PCR products. In conclusion, these new algorisms are very fast genotype identification tools that are suitable for evaluating E. granulosus s.l. isolated from livestock or livestock holders, particularly in developing countries.
Animals ; Animals, Wild ; Developing Countries ; Echinococcosis ; Echinococcus granulosus* ; Echinococcus* ; Genotype ; Humans ; Hygiene ; Livestock ; Parasites ; Polymerase Chain Reaction ; Poverty

Hydatid disease is a parasitic infection that is caused by the larval stage of Echinococcus granulosus. This disease is widely distributed in a temperate and subtropical countries, and it is especially prevalent in sheep and cattle raising countries. The most common infection site is the liver, and the second most common site is the lung. We report here on our experience with Echinococcal hydatid cysts that were found in the bladder of a 38 year old worker from Uzbekistan. The cysts were surgically removed and the patient was managed postoperatively with albendazole.
Adult ; Albendazole ; Animals ; Cattle ; Echinococcosis* ; Echinococcus ; Echinococcus granulosus ; Humans ; Liver ; Lung ; Sheep ; Urinary Bladder Neoplasms* ; Urinary Bladder* ; Uzbekistan

Hydatid disease is a parasitic infection that is caused by the larval stage of Echinococcus granulosus. This disease is widely distributed in a temperate and subtropical countries, and it is especially prevalent in sheep and cattle raising countries. The most common infection site is the liver, and the second most common site is the lung. We report here on our experience with Echinococcal hydatid cysts that were found in the bladder of a 38 year old worker from Uzbekistan. The cysts were surgically removed and the patient was managed postoperatively with albendazole.
Adult ; Albendazole ; Animals ; Cattle ; Echinococcosis* ; Echinococcus ; Echinococcus granulosus ; Humans ; Liver ; Lung ; Sheep ; Urinary Bladder Neoplasms* ; Urinary Bladder* ; Uzbekistan