Liver alveolar echinococcosis in China: Clinical aspect with relative basic research

This paper deals with all aspects of liver alveolar echinococcosis (AE) including epidemiology, pathology, clinical manifestations, imaging examinations, diagnosis and differential diagnosis, surgical treatment and chemotherapy.The review is not only based on personal clinical experiences but also in combination with relative basic research such as proliferation and growth of alveococcus, preclinical studies of a novel compound extracted from TCM for treatment of liver AE, and molecular immunology used for specific AE diagnosis, etc.

Compared efficacy of University of Wisconsin and histidine-tryptophan-ketoglutarate solutions in ex-situ liver resection and autotransplantation for end-stage hepatic alveolar echinococcosis patients

Background: The University of Wisconsin (UW) and histidine-tryptophan-ketoglutarate (HTK) solutions are the two most frequently used liver graft preservation fluids. The present study aimed to compare their efficacy in end-stage hepatic alveolar echinococcosis patients who underwent ex-situ liver resection and autotransplantation (ELRA). Methods: A total of 81 patients received ELRA from August 2010 to March 2018. They were allocated into UW ( n = 48) and HTK groups ( n = 33) based on the type of solutions used. Demographic and operational data were retrospectively analyzed. Primary outcomes included 90-day mortality, incidence of early graft loss, primary dysfunction, and postoperative complications. Results: Demographic and operational characteristics were similarly distributed in the two groups. No statistically significant differences were observed with regard to 90-day mortality (12.77% vs. 12.12%) and early graft loss rate (8.51% vs. 9.09%) between the two groups. Patients in the UW and HTK groups showed a primary dysfunction rate of 27.66% and 27.27%, respectively. The UW group exhibited a higher incidence tendency of biliary complications, albeit with no statistical significance. Conclusions: This is the largest cohort study comparing the efficacy of the UW and HTK organ-preserving solutions in end-stage hepatic alveolar echinococcosis patients in ELRA settings. UW and HTK solutions presented similar efficacy and safety. A randomized clinical trial with larger scale is needed for further investigation in future clinical applications.

Associating liver partition and portal vein ligation for staged hepatectomy for extensive alveolar echinococcosis:First case report in the literature

Alveolar echinococcosis(AE) is a zoonotic disease that is caused by Echinococcus multilocularis that affects liver and a variety of organs and tissues. It differs from other echinococcal disease because it shows tumor like behavior in the affected organ and tissues. The treatment of choice is concomitant medical therapy and resection with negative margins. Nevertheless, resection with the intent of negative margins(R0) may lead to serious complications such as liver failure. In the present case report, we used Associating Liver Partition and Portal Vein Ligation for Staged Hepatectomy(ALPPS) procedure, which was defined in 2012 by Schnitzbauer et al, in a 28-year-old male patient to avoid complications of major liver resection in order to treat alveolar echinococcosis. Until now, we have not encountered any study using ALPPS procedure for the treatment of alveolar echinococcosis. In the present case report we aimed to show that ALPPS procedure can be safely performed for marginnegative resection of primary or recurrent AE that shows a tumor like behavior. It is our opinion that this procedure should be performed in centers that have expertise and sufficient technical capacity to perform liver transplantation and advanced liver surgery.

Clinical Outcomes of Ex Vivo Liver Resection and Liver Autotransplantation for Hepatic Alveolar Echinococcosis

The effectiveness of liver autotransplantation for patients with partial hepatic alveolar echinococcosis was analyzed.We retrospectively studied 6 patients with hepatic alveolar echinococcosis who underwent liver autotransplantation in our hospital from 2008 to 2010.We also summarized the surgical indications of liver autotransplantation for hepatic alveolar echinococcosis and our experience in the management of postoperative complications of liver autotransplantation.Of 6 patients,5 achieved good curative results,and one died of multiple organ failure caused by portal vein thrombosis.Main complications included postoperative bleeding,bile leak and small-for-size liver graft syndrome.Liver autotransplantation offers a new approach to cure hepatic alveolar echinococcosis with non-resectable lesions.It could be the most effective method to cure intractable hepatic alveolar echinococcosis if correct handling in operation and proper prevention of complications are performed.But the long-term outcomes are still needed to be confirmed in longer follow-up.

Approaches to reconstruction of inferior vena cava by ex vivo liver resection and autotransplantation in 114 patients with hepatic alveolar echinococcosis

BACKGROUND Hepatic alveolar echinococcosis(AE)is most commonly found in retrohepatic inferior vena cava(RHIVC).Ex vivo liver resection and autotransplantation(ELRA)can better realize the radical resection of end-stage hepatic AE with severely compromised hepatocaval confluences,and reconstruction of the affected vessels.Currently,there is a scarcity of information regarding RHIVC reconstruction in ELRA.AIM To propose reasonable RHICV reconstruction strategies for ex vivo liver resection and autotransplantation.METHODS We retrospectively summarized the clinical data of 114 patients diagnosed with hepatic AE who treated by ELRA in our department.A total of 114 patients were divided into three groups according to the different reconstruction methods of RHIVC:Group A with original RHIVC being repaired and reconstructed(n=64),group B with RHIVC being replaced(n=43),and group C with RHIVC being resected without reconstruction(n=7).The clinical data of patients,including the operation time,anhepatic phase,intraoperative blood loss,complications and postoperative hospital stay,were analyzed and the patients were routinely followed up.The normally distributed continuous variables were expressed as means±SD,whereas the abnormally distributed ones were expressed as median and analyzed by analysis of variance.Survival curve was plotted by the Kaplan-Meier method.RESULTS All patients were routinely followed up for a median duration of 52(range,12-125)mo.The 30 d mortality rate was 7.0%(8/114)and 7 patients died within 90 d.Among all subjects,the inferior vena cava(IVC)-related complication rates were 17.5%(11/63)in group A and 16.3%(7/43)in group B.IVC stenosis was found in 12 patients(10.5%),whereas thrombus was formed in 6 patients(5.3%).Twenty-two patients had grade III or higher complications,with the complication rates being 17.2%,16.3%,and 57.1%in the three groups.The average postoperative hospital stay in the three groups was 32.3±19.8,26.7±18.2,and 51.3±29.4 d(P=0.03),respectively.CONCLUSION ELRA can be considered a safe and feasible option for end-stage hepatic AE patients with RHIVC infiltration.The RHIVC reconstruction methods should be selected appropriately depending on the defect degree of AE lesions in IVC lumen.The RHIVC resection without any reconstruction method should be considered with caution.

Application of 3D reconstruction for surgical treatment of hepatic alveolar echinococcosis

AIM: To evaluate the reliability and accuracy of threedimensional(3D) reconstruction for liver resection in patients with hepatic alveolar echinococcosis(HAE).METHODS: One-hundred and six consecutive patients with HAE underwent hepatectomy at our hospital between May 2011 and January 2015. Fifty-nine patients underwent preoperative 3D reconstruction and "virtual" 3D liver resection before surgery(Group A). Another 47 patients used conventional imaging methods for preoperative assessment(Group B). Outcomes of hepatectomy were compared between the two groups.RESULTS: There was no significant difference in preoperative data between the two groups. Compared with patients in Group B, those in Group A had a significantly shorter operation time(227.1 ± 51.4 vs 304.6 ± 88.1 min; P < 0.05), less intraoperative blood loss(308.1 ± 135.4 vs 458.1 ± 175.4 m L; P < 0.05), and lower requirement for intraoperative blood transfusion(186.4 ± 169.6 vs 289.4 ± 199.2 m L; P < 0.05). Estimated resection liver volumes in bothgroups had good correlation with actual graft weight(Group A: r = 0.978; Group B: r = 0.960). There was a significant higher serum level of albumin in Group A(26.3 ± 5.9 vs 22.6 ± 4.3 g/L, P < 0.05). Other postoperative laboratory parameters(serum levels of aminotransferase and bilirubin; prothrombin time) and duration of postoperative hospital stay were similar. Sixteen complications occurred in Group A and 19 in Group B. All patients were followed for 3-46(mean, 17.3) mo. There was no recurrence of lesions in Group A, but two recurrences in Group B. There were three deaths: two from cerebrovascular accident, and one from car accident.CONCLUSION: 3D reconstruction provides comprehensive and precise anatomical information for the liver. It also improves the chance of success and reduces the risk of hepatectomy in HAE.

Alveolar echinococcosis-spreading disease challenging clinicians: A case report and literature review

Human alveolar echinococcosis (AE) is a potentially deadly disease; recent studies have shown that the endemic area of Echinococcus multilocularis , its causative agent, is larger than previously known. This disease has low prevalence and remains underreported in Europe. Emerging clinical data show that diagnostic difficulties are still common. We report on a 76-year old patient suffering from AE lesions restricted to the left lobe of the liver who underwent a curative extended left hemihepatectomy. Prior to the resection a liver biopsy under the suspicion of an atypical malignancy was performed. After the intervention he developed a pseudoaneurysm of the hepatic artery that was successfully coiled. Surprisingly, during surgery, the macroscopic appearance of the tumour revealed a growth pattern that was rather typical for cystic echinococcosis (CE), i.e. , a gross tumour composed of multiple large vesicles with several centimeters in diameter. In addition, there were neither extensive adhesions nor infiltrations of the neighboring pancreas and diaphragm as was expected from previous imaging results. The unexpected diagnosis of AE was confirmed by definite histopathology, specific polymerase chain reaction and serology results. This is a rare case of unusual macroscopic presentation of AE that posed immense diagnostic challenges and had an eventful course. To our knowledge this is the first case of an autochthonous infection in this particular geographic area of Germany, the federal state of Saxony. This report may provide new hints for an expanding area of risk for AE and emphasizes the risk of complications in the scope of diagnostic procedures and the limitations of modern radiological imaging.

Successful robotic radical resection of hepatic echinococcosis located in posterosuperior liver segments

BACKGROUND Radical resection is an important treatment method for hepatic echinococcosis.The posterosuperior segments of the liver remain the most challenging region for laparoscopic or robotic hepatectomy.AIM To demonstrate the safety and preliminary experience of robotic radical resection of cystic and alveolar echinococcosis in posterosuperior liver segments.METHODS A retrospective analysis was conducted on the clinical data of 5 patients with a median age of 37 years(21-56 years)with cystic and alveolar echinococcosis in difficult liver lesions admitted to two centers from September to December 2019.The surgical methods included total pericystectomy,segmental hepatectomy,or hemihepatectomy.RESULTS Among the 5 patients,4 presented with cystic echinococcosis and 1 presented with alveolar echinococcosis,all of whom underwent robotic radical operation successfully without conversion to laparotomy.Total caudate lobectomy was performed in 2 cases,hepatectomy of segment Ⅶ in 1 case,total pericystectomy of segment Ⅷ in 1 case,and right hemihepatectomy in 1 case.Operation time was 225 min(175-300 min);blood loss was 100 mL(50-600 mL);and postoperative hospital stay duration was 10 d(5-19 d).The Clavien-Dindo complication grade was Ⅰ in 4 cases and Ⅱ in 1 case.No recurrence of echinococcosis was found in any patient at the 3 mo of follow-up.CONCLUSION Robotic radical surgery for cystic and selected alveolar echinococcosis in posterosuperior liver segments is safe and feasible.

Late biliary complications in human alveolar echinococcosis are associated with high mortality

AIM: To evaluate the incidence of late biliary complications in non-resectable alveolar echinococcosis (AE) under long-term chemotherapy with benzimidazoles.

Preoperative albumin level is a marker of alveolar echinococcosis recurrence after hepatectomy

AIM To identify a preoperative blood marker predictive of alveolar echinococcosis(AE) recurrence after hepatectomy.METHODS All consecutive patients who underwent operation for liver AE at the Lausanne University Hospital(CHUV) between January 1992 and December 2015 were included in this retrospective study. Preoperative laboratory values of leukocytes, mean corpuscular volume(MCV), red blood cell distribution width(RDW), thrombocytes, C-reactive protein(CRP) and albumin were collected and analyzed. Univariate and multivariate Cox regression analyses were performed to determine the risk factors for AE recurrence after liver resection. A receiver operating characteristic(ROC) curve was used to define the best discrimination threshold of the blood marker. Moreover, recurrencefree survival curves were calculated using the KaplanMeier method.RESULTS The cohort included 68 adult patients(37 females) with median age of 61 years [interquartile range(IQR): 46-71]. Eight of the patients(12%) presented a recurrence over a median follow-up time of 76 mo(IQR: 34-128). Median time to recurrence was 10 mo(IQR: 6-11). Median preoperative leukocyte, MCV, RDW,thrombocyte and CRP levels were similar between recurrent and non-recurrent cases. Median preoperative albumin level was 43 g/L(IQR: 41-45) for nonrecurrent cases and 36 g/L(IQR: 33-42) for recurrent cases(P = 0.005). The area under the ROC curve for preoperative albumin level to predict recurrence was 0.840(95%CI: 0.642-1, P = 0.002). The cutoff albumin level value was 37.5 g/L for sensitivity of 94.5% and specificity of 75%. In multivariate analysis, preoperative albumin and surgical resection margins were independent predictors of AE recurrence(HR = 0.099, P = 0.007 and HR = 0.182, P = 0.045 respectively).CONCLUSION Low preoperative albumin level was associated with AE recurrence in the present cohort. Thus, preoperative albumin may be a useful biomarker to guide follow-up.

Percutaneous management in hepatic alveolar echinococcosis:A sum of single center experiences and a brief overview of the literature

BACKGROUND Hepatic alveolar echinococcosis(HAE)is a serious zoonotic infection that affects humans.It may have a tumor-like appearance at times.Percutaneous treatment of HAE patients is extremely relaxing for them.HAE is a significant human zoonotic infection caused by the fox tapeworm Echinococcus Multilocularis larvae.It possesses the characteristics of an invasive tumor-like lesion due to its infiltrative growth pattern and protracted incubation period.The disease is endemic over central Europe,Asia,and North America.AIM To characterize HAE patients who were treated percutaneously,their outcomes,and the major technical features of percutaneous treatment in HAE.METHODS Patients who were treated with percutaneous cyst drainage and/or percutaneous biliary drainage were included in the study.Uncorrected abnormal coagulation values and solid or non-infected HAE with minor necrotic change were excluded.RESULTS Thirty-two patients underwent percutaneous cyst drainage,two patients underwent percutaneous biliary drainage,and four patients underwent percutaneous biliary drainage alone.Interventional radiology is utilized to drain echinococcal necrosis and abscesses within/without the liver,as well as diseased and clogged bile ducts.CONCLUSION Percutaneous drainage of cyst contents and/or biliary channels using a minimally invasive technique is a very beneficial.Percutaneous cyst drainage with albendazole therapy improves quality of life in patients who are unable to undergo surgery,even when the mass resolves with long-term treatment.

Diagnostic imaging and interventional procedures in a growing problem: Hepatic alveolar echinococcosis

Alveolar echinococcosis(AE) of the liver is caused by the metacestode of the fox tapeworm Echinococcus multilocularis(E. multilocularis), which is endemic in many parts of the world. AE is a very aggressive and potentially fatal infestation which always affects the liver primarily and metastasizes to any part of the body. Without timely diagnosis and therapy, the prognosis is dismal, with death the eventual outcome in most cases. Diagnosis is usually based on findings at radiological imaging and in serological analyses. The alveolar cysts grow by exogenous proliferation and behave like a malignant neoplasm. Since AE lesions can occur almost anywhere in the body, familiarity with the spectrum of cross-sectional imaging appearances is advantageous. Therefore, AE lesions can cause physicians to generate a long list of differential diagnoses, including malignant tumors. Disseminated parasitic lesions in unusual locations with atypical imaging appearances may make it difficult to narrow the differential diagnosis. For diagnosis, ultrasonography(US) remains the first line examination. For a more accurate disease evaluation, aiming to guide the surgical strategy, computed tomography(CT), magnetic resonance imaging(MRI), including magnetic resonance cholangiography(MRC) imaging, are of importance, providing useful complementary information. However, making the correct diagnosis is possible if imaging findings are correlated with appropriate clinical findings. We present an overview of the radiological patterns produced by E. multilocularis lesions as seen on US, CT and MRI and discuss the interventional procedures in hepatic AE lesions.

肝泡型包虫病的诊治进展

肝包虫病为慢性寄生虫病,由多房棘球蚴的幼虫引发,此病具有相当高的致残率和死亡风险,又被称作“虫癌”。临床上将肝包虫病分为肝泡型包虫病和肝囊型包虫病,肝包虫病在世界各地广泛流行,我国主要在西部农牧区居民群体中发生,对该地区居民的生存质量构成了巨大的威胁。目前临床上治疗肝包虫病以手术治疗方式为主,伴随着外科诊治技术的迅速发展和对肝包虫病研究不断深入,其诊治方案也不断完善。本文将主要综述肝泡型包虫病的诊断和治疗进展,旨在为临床医务人员提供参考,以尽早发现和治疗,降低疾病对患者产生的不良影响,改善患者预后。

Complex inferior vena cava reconstruction during ex vivo liver resection and autotransplantation:A case report

BACKGROUND Ex vivo liver resection and autotransplantation(ELRA)is an essential approach for treating patients with end-stage hepatic alveolar echinococcosis(AE),and its surgical indications involve severe invasion of important hepatic vessels,which makes in vivo resection impossible.Revascularization is a major step in the process of ELRA,which is extremely challenging when the invaded vessels have huge defects.CASE SUMMARY Herein,we have reported the case of a 26-year-old patient with hepatic AE in an autologous liver graft who underwent complex inferior vena cava(IVC)reconstruction using disease-free IVC,autologous portal vein fragments,and umbilical vein within the ligamentum teres hepatis.The patient showed good surgical recovery without vascular-related complications during the long-term follow-up.CONCLUSION We reviewed three studies that have reported complex revascularization of the IVC.This case report and systematic review showed that the use of autologous perihepatic vessels prevents donor-area trauma,immune rejection,and other adverse reactions.When the blood vessel is severely invaded and a single vascular material cannot repair and reconstruct the defect,ELRA may provide a safe and feasible surgical approach,which has good prospects for clinical application.

终末期肝泡型包虫病的肝移植治疗

肝泡型包虫病(HAE)是我国西部地区常见的人畜共患地方性寄生虫病,早期缺乏典型临床表现,症状明显时常已进入终末期,具有极高的致死率。在终末期HAE(es-HAE)治疗中,因残余肝体积不足、无法控制的出血、脉管在体重建困难等,同种异体原位肝移植几乎成为唯一根治的方式。但因供肝短缺、术后需长期使用免疫抑制药等因素限制了其应用,离体肝切除联合自体肝移植术(ELRA)的出现打破了这一窘境,极大拓宽了es-HAE手术指征。此外,我国多中心对ELRA进行了诸多优化与改良,进一步完善es-HAE的治疗体系。目前,es-HAE的肝移植(包括ELRA)治疗仍是临床医师研究的热点。本文对原位肝移植、ELRA、辅助性ELRA等多种手段在es-HAE的外科治疗进行综述,旨在进一步提升es-HAE的诊治水平,改善患者临床预后。

Ex vivo liver resection followed by autotransplantation for end-stage hepatic alveolar echinococcosis

Background For patients with end-stage hepatic alveolar echinococcosis （AE）, in vivo resection of the involved parts of the liver is usually very difficult, therefore, allogenic liver transplantation is indicated. However, we hypothesize that for selected patents, ex vivo liver resection for thorough elimination of the involved tissues and liver autotransplantation may offer a chance for clinical cure. Methods We presented a 24-year-old women with a giant hepatic AE lesion who was treated with hepatectomy, ex vivo resection of the involved tissue and hepatic autotransplantation. The patient had moderate jaundice and advanced hepatic AE lesion which involved segments I, IV, V, VI, VII, VIII and retrohepatic inferior vena cava. The lateral segments （II and III） of the left liver remained normal with over 1000 ml in its volume. No extrahepatic metastases （such as to the lung or brain） could be found. As the first step of treatment, X-ray guided percutaneous transhepatic cholangiodrainage （PTCD） was performed twice for bile drainage in segment III and II separately until her serum total bilirubin decreased gradually from 236 to 88 umol/L. Total liver resection was then performed, followed by extended right hepatic trisegmentectomy and the entire retrohepatic vena cava was surgically removed en bloc while her hemodynamics parameters were stable. Neither veino-veinous bypass nor temporary intracorporeal cavo-caval or porto-caval shunt was used during the 5.7-hour anhepatic phase. The remained AE-free lateral segments of the-left liver were re-implanted in situ. The left hepatic vein was directly anastomosed end-to-end to the suprahepatic inferior vena cava due to the lack of the retrohepatic inferior vena cava with AE total infiltration. Because compensatory retroperitoneal porto-caval collateral circulation developed, we enclosed remained infrahepatic inferior vena cava at renal vein level without any haemodynamics problems. Results During a 60-day following-up after operation, the patient had a good recovery except for a mildly elevated serum total bilirubin. Conclusions As a radical approach, ex vivo liver resection and liver autotransptantation in a case has shown a optimal potential for treatment of the end-stage hepatic AE. Strict compliance with its indications, evaluation of vessels of patients pre-operatively, and precise surgical techniques are the keys to improve the prognosis of patients.

Mfn2、miRNA-101调控转化生长因子-β1参与肝泡型包虫病肝纤维化的研究

目的 探讨Mfn2、miRNA-101通过TGF-β1调控肝泡型包虫病肝纤维化的潜在作用。方法 收集2020年7月-2020年10月青海省人民医院确诊的20例肝泡型包虫病患者术后切除的肝组织,采集距肝脏病灶边缘2 cm以外正常肝组织、距肝脏病灶边缘2 cm以内肝组织。行HE、Masson染色,将不同标本纤维化分级,通过免疫组化、RT-qPCR检测Mfn2、TGF-β1、α-SMA、miRNA-101在不同级别纤维化组织中的表达。结果 Mfn2、TGF-β1、α-SMA、miRNA-101在不同级别纤维化组织中的差异表达均具有统计学意义(P<0.05)。Mfn2与TGF-β1、α-SMA表达量均呈负相关(P<0.05),miRNA-101与TGF-β1、α-SMA表达量均呈负相关(P<0.05),TGF-β1与α-SMA表达量呈正相关(P<0.05),Mfn2与miRNA-101表达量无显著相关性(P>0.05)。结论 Mfn2、miRNA-101可能抑制肝泡型包虫病肝纤维化,并且可能通过抑制TGF-β1发挥抗纤维化作用。

泡球蚴感染对小鼠肝脏TRIF/IRF3通路和IFITM3的影响

目的探索泡球蚴感染后不同时间对小鼠肝脏β干扰素TIR结构域衔接蛋白(TRIF)/干扰素调节因子3(IRF3)通路及干扰素诱导的跨膜蛋白3(IFITM3)的影响。方法对泡型包虫病患者和其他非感染性肝病(肝血管瘤)手术患者的肝脏组织进行HE及免疫组织化学染色,观察肝组织病理变化及IFITM3和叉头框蛋白P3(Foxp3)的表达。48只C57BL/6J雌性小鼠随机分为感染组(腹腔感染泡球蚴后8,30,60,90,180 d)和对照组。Masson染色观察小鼠肝脏纤维化病变,Western blot和实时荧光定量PCR检测小鼠肝脏TRIF/IRF3通路、IFITM3和Foxp3变化。结果泡型包虫病患者肝脏病灶周围肝组织排列紊乱,有大量炎性细胞浸润;肝血管瘤患者肝脏组织完整。免疫组化检测结果,泡型包虫病患者IFITM3和Foxp3的表达量高于肝血管瘤患者。Masson染色显示泡球蚴感染导致小鼠肝脏纤维化病变。实时荧光定量PCR结果显示,感染组小鼠肝脏TRIF、IRF3、IFNB1和IFITM3 mRNA表达在感染60 d和90 d时均高于对照组(P<0.05),但感染180 d时TRIF、IFNB1和IFITM3 mRNA表达与对照组比较差异无统计学意义(P>0.05);Foxp 3 mRNA的表达随着泡球蚴感染时间呈先上升后下降再上升的趋势,在感染180 d上升到峰值(P<0.05)。Western blot结果显示,泡球蚴感染小鼠90 d,肝脏TRIF、IRF3、IFITM3蛋白表达量均高于对照组(P<0.05),但感染180 d时与对照组比较差异无统计学意义(P>0.05);在感染90 d和180 d时,Foxp3蛋白表达高于对照组,并在180 d上升到峰值(P<0.05)。结论晚期泡球蚴感染时,小鼠肝脏的TRIF/IRF3通路及IFITM3表达下调,影响了天然免疫的抗病原体功能,抑制宿主发挥抗病原体的作用。

Present epidemic situation of liver alveolar echinococcosis in Gansu Province, China

Echinococcosis is a common parasitic zoonosis in the west of China. Twotypes, cystic echinococcosis (CE) and alveolar echinococcosis (AE) are respectively caused by thelarval stage of Echinococcus granulosus and Echinococcus multilocularis. As early as the 1960s, AE,simply named alveococcosis, was successively reported in Qinghai, Xinjiang and Gansu, and then inNingxia, Sichuan, Tibet, Inner Mongolia and Heilongjiang. At the same time, the epidemiologicalsurveys and clinical or experimental studies were also conducted. Until now, the total number of AEcases reported in China. has exceeded one thousand. Since the first report of AE in Gansu in 1971, atotal of 105 cases have been diagnosed. The regional distribution and epidemic factors of AE inGansu are reviewed here.