今日の臨床サポート

エキノコックス症

著者: 髭修平 JA北海道厚生連札幌厚生病院 第三消化器内科

監修: 金子周一 金沢大学大学院

著者校正済:2021/08/18
現在監修レビュー中
患者向け説明資料

概要・推奨   

  1. 本症の診断のために、エキノコックス抗体検査の実施が強く推奨される(推奨度1)。
  1. エキノコックス症患者の早期発見のために、血清検査と超音波検査の組み合わせによるスクリーニング検査は有用である(推奨度2)。
  1. エキノコックス症の画像診断において、MRIによる小嚢胞性病変の確認が有用である(推奨度2)。
  1. 閲覧にはご契約が必要となります。閲覧にはご契約が必要となります。閲覧にはご契約が必要とな
  1. 閲覧にはご契約が必要となります。閲覧にはご契約が必要となります。閲覧にはご契約が必要となります。閲覧
  1. 閲覧にはご契約が必要となります。閲覧にはご契約が必要となります。閲覧にはご契約が必要となります。
  1. 閲覧にはご契約が必要となります。閲覧にはご契約が必要となります。閲覧にはご契約が必要となります。
薬剤監修について:
オーダー内の薬剤用量は日本医科大学付属病院 薬剤部 部長 伊勢雄也 以下、林太祐、渡邉裕次、井ノ口岳洋、梅田将光による疑義照会のプロセスを実施、疑義照会の対象については著者の方による再確認を実施しております。
※薬剤中分類、用法、同効薬、診療報酬は、エルゼビアが独自に作成した薬剤情報であり、
著者により作成された情報ではありません。
尚、用法は添付文書より、同効薬は、薬剤師監修のもとで作成しております。
※薬剤情報の(適外/適内/⽤量内/⽤量外/㊜)等の表記は、エルゼビアジャパン編集部によって記載日時にレセプトチェックソフトなどで確認し作成しております。ただし、これらの記載は、実際の保険適用の査定において保険適用及び保険適用外と判断されることを保証するものではありません。また、検査薬、輸液、血液製剤、全身麻酔薬、抗癌剤等の薬剤は保険適用の記載の一部を割愛させていただいています。
(詳細はこちらを参照)
著者のCOI(Conflicts of Interest)開示:
髭修平 : 特に申告事項無し[2021年]
監修:金子周一 : 研究費・助成金など(バイエル薬品株式会社,株式会社キュービクス,アボットジャパン合同会社,日東電工株式会社,株式会社スギ薬局,株式会社サイトパスファインダー),奨学(奨励)寄付など(小野薬品工業株式会社,エーザイ株式会社,株式会社ツムラ,アッヴィ合同会社,大日本住友製薬株式会社,ゼリア新薬工業株式会社,塩野義製薬株式会社,大塚製薬株式会社,アステラス製薬株式会社,田辺三菱製薬株式会社,マイランEPD合同会社,EAファーマ株式会社,大鵬薬品工業株式会社,中外製薬株式会社,協和キリン株式会社,持田製薬株式会社,日本ケミファ株式会社,LifeScan Japan株式会社)[2021年]

改訂のポイント:
  1. 定期レビューを行い、疫学情報・治療について加筆修正を行った。

病態・疫学・診察

疾患情報(疫学・病態)  
  1. エキノコックス症とは、多節条虫類であるエキノコックスがヒトに寄生し、主に肝臓に腫瘤性病変を形成する寄生虫疾患である。キツネやイヌなどの終宿主から排泄された虫卵を経口的に摂取後、仔虫が腸管内から肝臓へ移行し、幼虫(包虫)として病変を形成する[1]
  1. わが国のエキノコックス患者発生数は、多包性で年間20~30例程度である。年齢中央値は65歳、男女差はない[2]
  1. 北海道居住者に多いが、北海道以外の発生も報告され、注意を要する。
  1. 潜伏期間は長く、自・他覚症状は乏しい。広汎な肝内進展、他臓器への浸潤、肺・脳などへの転移を来した場合には、予後不良となる。
  1. エキノコックス症は、感染症法の4類感染症に分類され、診断した医師は、ただちに所管の保健所に届け出る必要がある。 
  1. 北海道では患者の早期発見・早期治療を目的として希望者に対して無料のエキノコックス症健康診断を実施している。
 
多包条虫の生活環とヒトへの寄生

終宿主はキツネやイヌ、オオカミなどで、ヒトやネズミが中間宿主となる。成虫(包状虫)は終宿主に、幼虫(包虫)は中間宿主に寄生する。成虫が終宿主の腸管内寄生後に生産して体外に排泄された受精卵を経口摂取した中間宿主の肝臓内で幼虫となり、それを捕食した終宿主内で原頭節を基に成虫となる。

出典

img1:  The global burden of alveolar echinococcosis.
 
 PLoS Negl Trop Dis. 2010 Jun 22;4(6):e72・・・
問診・診察のポイント  
  1. 肝の腫瘤性病変を認めた際に本疾患の存在を想起することが最も重要である。

これより先の閲覧には個人契約のトライアルまたはお申込みが必要です。

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文献 

著者: Hao Wen, Lucine Vuitton, Tuerhongjiang Tuxun, Jun Li, Dominique A Vuitton, Wenbao Zhang, Donald P McManus
雑誌名: Clin Microbiol Rev. 2019 Mar 20;32(2). doi: 10.1128/CMR.00075-18. Epub 2019 Feb 13.
Abstract/Text Echinococcosis is a zoonosis caused by cestodes of the genus Echinococcus (family Taeniidae). This serious and near-cosmopolitan disease continues to be a significant public health issue, with western China being the area of highest endemicity for both the cystic (CE) and alveolar (AE) forms of echinococcosis. Considerable advances have been made in the 21st century on the genetics, genomics, and molecular epidemiology of the causative parasites, on diagnostic tools, and on treatment techniques and control strategies, including the development and deployment of vaccines. In terms of surgery, new procedures have superseded traditional techniques, and total cystectomy in CE, ex vivo resection with autotransplantation in AE, and percutaneous and perendoscopic procedures in both diseases have improved treatment efficacy and the quality of life of patients. In this review, we summarize recent progress on the biology, epidemiology, diagnosis, management, control, and prevention of CE and AE. Currently there is no alternative drug to albendazole to treat echinococcosis, and new compounds are required urgently. Recently acquired genomic and proteomic information can provide a platform for improving diagnosis and for finding new drug and vaccine targets, with direct impact in the future on the control of echinococcosis, which continues to be a global challenge.

Copyright © 2019 American Society for Microbiology.
PMID 30760475  Clin Microbiol Rev. 2019 Mar 20;32(2). doi: 10.1128/CMR・・・
著者: Gitte Bartholomot, Dominique A Vuitton, Said Harraga, Da Zhong Shi, Patrick Giraudoux, Guy Barnish, Yun Hai Wang, Calum N L MacPherson, Philip S Craig
雑誌名: Am J Trop Med Hyg. 2002 Jan;66(1):23-9.
Abstract/Text Alveolar echinococcosis (AE), caused by Echinococcus multilocularis, is a zoonotic helminthic disease that can mimic malignancy. In the 1970s, foci of the disease were found in central China. The aim of the present study was to estimate the prevalence of AE in humans in 2 districts of south Gansu Province, China, by use of ultrasound and Echinococcus serology. After answering an epidemiological questionnaire, 2,482 volunteers from 28 villages underwent ultrasound. Serology via enzyme-linked immunosorbent assay for antibody activity was performed on whole blood collected on filter paper in all subjects; on serum from subjects with an abnormal ultrasound image; and on randomly chosen subjects that either had no lesions or had atypical lesions. At least one (25.3%) abnormal ultrasound image was observed in 630 of the subjects screened. A typical lesion of progressive AE was found in 84 subjects (3.4%). Serologies were positive in 77 (96%) of 80 of patients who had lesions typical of progressive AE. Ultrasound is useful for screening for AE in endemic regions.

PMID 12135263  Am J Trop Med Hyg. 2002 Jan;66(1):23-9.
著者: Akira Ito, Yasuhito Sako, Hiroshi Yamasaki, Wulamu Mamuti, Kazuhiro Nakaya, Minoru Nakao, Yuji Ishikawa
雑誌名: Acta Trop. 2003 Feb;85(2):173-82.
Abstract/Text Extensive experience has documented that Em2(plus)-ELISA, Em10-ELISA and Em18-immunoblot and Em18-ELISA are reliable serologic methods for detection of alveolar echinococcosis (AE) caused by the metacestodes of Echinococcus multilocularis. Among these, tests based on detection of antibodies to the specific Em18 antigen, either immunoblot or ELISA, appears to be the most specific for AE. Between 90 and 97% of AE cases with characteristic hepatic lesions detectable by image analysis have been positive in Em18-serology. In contrast Antigen B (8 kDa)-immunoblot is the most sensitive for all forms of echinococcosis, although it can not differentiate AE from cystic echinococcosis (CE). Primary serologic screening for echinococcosis, especially for CE using hydatid cyst fluid of Echinococcus granulosus appears to be highly sensitive in endemic areas. Glycoproteins (GPs) purified from cyst fluid of Taenia solium are highly specific for diagnosis of T. solium neuorcysticercosis (NCC). Using currently available antigens it is not difficult to differentiate these three larval cestodiases serologically. We recommend that (1) primary screening of CE in endemic areas should be carried out using hydatid cyst fluid of E. granulosus prepared from cysts in either sheep, human or mouse for immunoblot and from sheep or mouse for ELISA, (2) both primary screening and confirmation of AE in endemic areas should be carried out using Em18-ELISA, Em18-immunoblot or Em2(plus)-ELISA. Serodiagnosis in areas where both AE and CE are endemic, such as in China, should be carried out as a combination of (1) and (2), and (3) serology of NCC should be carried out using GP-ELISA or GP-immunoblot. All samples showing antibody to Em18 are exclusively from echinococcosis cases. There have been no false positive test reactions with sera from other diseases. Strongest Em18 responders are all from patients with AE but some weaker responses may be found in sera of persons with advanced complex lesions of CE. These highly reliable serodiagnostic methods using native, recombinant and synthetic antigens are briefly summarized and experiences with these methods in Japan is reviewed. We believe that use of these specific antigens in screening and confirmation programs for AE in Japan will improve specificity and reduce the confusion, anxiety and expense in persons whose sera give false positive reactions with crude echinococcal antigens.

PMID 12606094  Acta Trop. 2003 Feb;85(2):173-82.
著者: Johannes Eckert, Peter Deplazes
雑誌名: Clin Microbiol Rev. 2004 Jan;17(1):107-35.
Abstract/Text Echinococcosis in humans is a zoonotic infection caused by larval stages (metacestodes) of cestode species of the genus Echinococcus. Cystic echinococcosis (CE) is caused by Echinococcus granulosus, alveolar echinococcosis (AE) is caused by E. multilocularis, and polycystic forms are caused by either E. vogeli or E. oligarthrus. In untreated cases, AE has a high mortality rate. Although control is essentially feasible, CE remains a considerable health problem in many regions of the northern and southern hemispheres. AE is restricted to the northern hemisphere regions of North America and Eurasia. Recent studies have shown that E. multilocularis, the causative agent of AE, is more widely distributed than previously thought. There are also some hints of an increasing significance of polycystic forms of the disease, which are restricted to Central and South America. Various aspects of human echinococcosis are discussed in this review, including data on the infectivity of genetic variants of E. granulosus to humans, the increasing invasion of cities in Europe and Japan by red foxes, the main definitive hosts of E. multilocularis, and the first demonstration of urban cycles of the parasite. Examples of emergence or reemergence of CE are presented, and the question of potential spreading of E. multilocularis is critically assessed. Furthermore, information is presented on new and improved tools for diagnosing the infection in final hosts (dogs, foxes, and cats) by coproantigen or DNA detection and the application of molecular techniques to epidemiological studies. In the clinical field, the available methods for diagnosing human CE and AE are described and the treatment options are summarized. The development of new chemotherapeutic options for all forms of human echinococcosis remains an urgent requirement. A new option for the control of E. granulosus in the intermediate host population (mainly sheep and cattle) is vaccination. Attempts are made to reduce the prevalence of E. multilocualaris in fox populations by regular baiting with an anthelmintic (praziquantel). Recent data have shown that this control option may be used in restricted areas, for example in cities, with the aim of reducing the infection risk for humans.

PMID 14726458  Clin Microbiol Rev. 2004 Jan;17(1):107-35.
著者: B Gottstein, P Jacquier, S Bresson-Hadni, J Eckert
雑誌名: J Clin Microbiol. 1993 Feb;31(2):373-6.
Abstract/Text Alveolar echinococcosis (AE) in humans is generally a fatal disease when not diagnosed early enough to provide curative treatment such as radical surgery. Immunodiagnosis for early detection of AE was improved by the isolation of an affinity-purified metacestode Em2 antigen and by the synthesis of recombinant Echinococcus multilocularis antigen II/3-10. Both antigens were individually assessed by enzyme-linked immunosorbent assay (ELISA) and demonstrated high specificities and diagnostic sensitivities, although both missed approximately 4 to 11% of diagnostic cases of AE. To provide an optimal serodiagnostic test, we investigated the two purified antigens by using a test employing a mixture of both purified antigens (designated Em2plus antigen) in one assay. For comparative purposes, crude E. multilocularis and Echinococcus granulosus metacestode antigens were investigated as well. The Em2plus ELISA proved to be the optimal diagnostic test with the highest diagnostic sensitivity, 97%, in serum samples from 140 patients with AE and an overall specificity of 99% for infections due to other Echinococcus and non-Echinococcus parasites. The new test combination (Em2plus ELISA) is suggested for the serodiagnosis of AE in patients and for seroepidemiological surveys.

PMID 8432825  J Clin Microbiol. 1993 Feb;31(2):373-6.
著者: Yoshihisa Kodama, Nobuyuki Fujita, Tadashi Shimizu, Hideho Endo, Toshikazu Nambu, Naoki Sato, Satoru Todo, Kazuo Miyasaka
雑誌名: Radiology. 2003 Jul;228(1):172-7. doi: 10.1148/radiol.2281020323. Epub 2003 May 15.
Abstract/Text PURPOSE: To clarify the magnetic resonance (MR) imaging findings of alveolar echinococcosis in the liver.
MATERIALS AND METHODS: Thirty-five patients with 50 lesions histologically proven to be alveolar echinococcosis were evaluated with MR imaging. Lesions were assessed with regard to the distribution pattern of solid and cystic components and pattern of contrast material enhancement.
RESULTS: Cystic components exhibited two patterns at T2-weighted MR imaging: small round cysts and large and/or irregular cysts. Forty-eight lesions (96%) contained small round cysts. Twenty-six lesions (52%) had large and/or irregular cysts. Forty-five lesions (90%) were associated with a solid component. MR imaging characteristics were categorized into five types: multiple small round cysts without a solid component (two lesions [4%], type 1), multiple small round cysts with a solid component (20 lesions [40%], type 2), a solid component surrounding large and/or irregular cysts with multiple small rounds cysts (23 lesions [46%], type 3), a solid component without cysts (two lesions [4%], type 4), and a large cyst without a solid component (three lesions [6%], type 5). In most cases (97%), contrast enhancement was weak.
CONCLUSION: The MR findings of alveolar echinococcosis in the liver are multiple small round cysts with a weakly enhanced solid component. The cystic component can be a large and/or irregular lesion, and such lesions are depicted clearly at T2-weighted MR imaging.

PMID 12750459  Radiology. 2003 Jul;228(1):172-7. doi: 10.1148/radiol.2・・・
著者: S Reuter, K Nüssle, O Kolokythas, U Haug, A Rieber, P Kern, W Kratzer
雑誌名: Infection. 2001 May-Jun;29(3):119-25.
Abstract/Text BACKGROUND: We compared the imaging findings in patients with alveolar liver echinococcosis using ultrasound (US), computerized tomography (CT) and magnetic resonance imaging (MRI) in a prospective study.
PATIENTS AND METHODS: 30 patients with alveolar echinococcosis (AE) were examined with the above imaging techniques.
RESULTS: 30 lesions were detected with all three methods and most lesions (n = 55) were detected with CT. Calcifications were seen in 15 lesions with US, in 21 with CT and in 16 with MRI. MRI best detected necrotic areas and multivesicuLar structures.
CONCLUSION: US is the screening method of choice and should primarily be complemented by CT due to its ability to detect the greatest number of lesions and clear demarcation of the characteristic calcifications. MRI may facilitate the diagnosis in uncertain cases with noncalcified or partially calcified lesions by showing the characteristic multivesicular structure, necrotic areas and proximity to vascular structures.

PMID 11440381  Infection. 2001 May-Jun;29(3):119-25.
著者: Peter Kern, Hao Wen, Naoki Sato, Dominique A Vuitton, Beate Gruener, Yinmei Shao, Eric Delabrousse, Wolfgang Kratzer, Solange Bresson-Hadni
雑誌名: Parasitol Int. 2006;55 Suppl:S283-7. doi: 10.1016/j.parint.2005.11.041. Epub 2005 Dec 15.
Abstract/Text Alveolar echinococcosis is caused by the larval stage of the fox tapeworm (Echinococcus multilocularis) and is frequently diagnosed as a space occupying lesion in the liver. The growth pattern resembles that of a malignant tumor with infiltration throughout the liver, spreading into neighbouring organs and metastases formation in distant organs. Thus, one of the prevailing differential diagnoses is liver cancer. Guided by the Tumor-Node-Metastasis (TNM) system of liver cancer, the European Network for Concerted Surveillance of Alveolar Echinococcosis and the WHO Informal Working Group on Echinococcosis proposed a clinical classification for alveolar echinococcosis. It was designated as PNM system (P = parasitic mass in the liver, N = involvement of neighbouring organs, and M = metastasis). As for TNM in oncology, single PNM categories were combined into four stages, I to IV. The system was developed by a retrospective analysis of 97 patients' records from two treatment centers (Besançon/France and Ulm/Germany). Recently, this WHO classification was applied to 222 patients in 4 clinical centers around the world (Besançon/France, n = 26; Urumqi/China, n = 46; Sapporo/Japan, n = 58; and Ulm/Germany, n = 92). All patients could be classified who had been diagnosed in the period from January 1998 to June 2005. The stage grouping indicated center differences, but appeared to segregate patients according to various treatment regimens. The WHO classification not only serves as a tool for the international standardization of disease manifestation but also aids to evaluate the outcome of a chosen diagnostic and treatment procedure in different treatment centers in Europe and Asia.

PMID 16343985  Parasitol Int. 2006;55 Suppl:S283-7. doi: 10.1016/j.par・・・
著者: Norio Kawamura, Toshiya Kamiyama, Naoki Sato, Kazuaki Nakanishi, Hideki Yokoo, Hirofumi Kamachi, Munenori Tahara, Shoji Yamaga, Michiaki Matsushita, Satoru Todo
雑誌名: J Am Coll Surg. 2011 May;212(5):804-12. doi: 10.1016/j.jamcollsurg.2011.02.007. Epub 2011 Mar 12.
Abstract/Text BACKGROUND: Hepatectomy is the first-line treatment for alveolar echinococcosis (AE) if complete resection is feasible. However, a strategy for the treatment of patients with AE in whom the tumor cannot be resected completely remains to be defined.
STUDY DESIGN: Data were retrospectively collected from 188 consecutive patients between 1984 and 2009. Overall survival (OS), progression-free survival (PFS), and risk factors were analyzed in patients classified into 3 groups (group A: complete resection, group B: reduction surgery, and group C: drainage or exploratory laparotomy).
RESULTS: In group A (n = 119), the 10-, 15-, and 20-year OS was 98.9%. In group B (n = 63), the 10-, 15-, and 20-year OS was 97.1%, 92.8%, and 61.9%. In group C (n = 6), the 10- and 15-year OS was 50.0% and 33.3%. Patients in groups A and B had better prognoses than those in group C (p < 0.001). In group A, the 10-, 15-, and 20-year PFS was 96.5%, 94.4%, and 94.4%. In group B, the 10-, 15-, and 20-year PFS was 87.1%, 71.6%, and 61.4%. In group C, the 10- and 15-year PFS was 50.0% and 33.3%. Patients in group A had better PFS than those in groups B and C (p < 0.001). Curability was the only independent factor for both OS and PFS by multivariate analysis.
CONCLUSIONS: Although the most effective therapy for AE is complete resection, a better prognosis can be achieved by reduction surgery and/or adjuvant albendazole therapy for patients with AE that cannot be completely resected.

Copyright © 2011 American College of Surgeons. Published by Elsevier Inc. All rights reserved.
PMID 21398158  J Am Coll Surg. 2011 May;212(5):804-12. doi: 10.1016/j.・・・
著者: Paul R Torgerson, Alexander Schweiger, Peter Deplazes, Maja Pohar, Jürg Reichen, Rudolf W Ammann, Philip E Tarr, Nerman Halkik, Beat Müllhaupt
雑誌名: J Hepatol. 2008 Jul;49(1):72-7. doi: 10.1016/j.jhep.2008.03.023. Epub 2008 Apr 28.
Abstract/Text BACKGROUND/AIMS: Alveolar echinococcosis (AE) is a serious liver disease. The aim of this study was to explore the long-term prognosis of AE patients, the burden of this disease in Switzerland and the cost-effectiveness of treatment.
METHODS: Relative survival analysis was undertaken using a national database with 329 patient records. 155 representative cases had sufficient details regarding treatment costs and patient outcome to estimate the financial implications and treatment costs of AE.
RESULTS: For an average 54-year-old patient diagnosed with AE in 1970 the life expectancy was estimated to be reduced by 18.2 and 21.3 years for men and women, respectively. By 2005 this was reduced to approximately 3.5 and 2.6 years, respectively. Patients undergoing radical surgery had a better outcome, whereas the older patients had a poorer prognosis than the younger patients. Costs amount to approximately Euro108,762 per patient. Assuming the improved life expectancy of AE patients is due to modern treatment the cost per disability-adjusted life years (DALY) saved is approximately Euro6,032.
CONCLUSIONS: Current treatments have substantially improved the prognosis of AE patients compared to the 1970s. The cost per DALY saved is low compared to the average national annual income. Hence, AE treatment is highly cost-effective in Switzerland.

PMID 18485517  J Hepatol. 2008 Jul;49(1):72-7. doi: 10.1016/j.jhep.200・・・
著者: Z Kadry, E C Renner, L M Bachmann, N Attigah, E L Renner, R W Ammann, P-A Clavien
雑誌名: Br J Surg. 2005 Sep;92(9):1110-6. doi: 10.1002/bjs.4998.
Abstract/Text BACKGROUND: Alveolar echinococcosis is a rare disorder, which makes a comparison of different treatment modalities within a clinical trial difficult to perform. Data prospectively recorded over a period of 25 years were used to evaluate three therapeutic strategies: benzimidazole therapy alone, complete 'curative' resection followed by 2 years of adjuvant benzimidazole treatment, and partial debulking resection followed by continuous administration of a benzimidazole.
METHODS: Details of 113 patients with hepatic alveolar echinococcosis treated between 1976 and 2003 were analysed. Kaplan-Meier survival curves were constructed and, using a Cox regression model, patient age, year of initial treatment and PNM stage were entered as co-variates in the analysis.
RESULTS: Kaplan-Meier overall survival curves stratified for treatment strategy indicated an improved long-term survival in patients undergoing the debulking procedure (P = 0.061) or curative resection (P = 0.002) compared with benzimidazole therapy alone. However, when PNM stage, patient age and year of initial treatment were introduced into the analysis, there was a trend for survival advantage only with curative resection (P = 0.07 versus benzimidazole alone). Debulking resulted in a higher rate of progression of hepatic echinococcosis than curative surgery (P = 0.008). The incidence of parasite-related complications was similar for debulking resection and benzimidazole therapy alone (P = 0.706).
CONCLUSION: Debulking hepatic resections do not appear to offer any advantage in the treatment of patients with alveolar echinococcosis.

Copyright 2005 British Journal of Surgery Society Ltd.
PMID 16044412  Br J Surg. 2005 Sep;92(9):1110-6. doi: 10.1002/bjs.4998・・・
著者: Klaus Buttenschoen, Daniela Carli Buttenschoen, Beate Gruener, Peter Kern, Hans G Beger, Doris Henne-Bruns, Stefan Reuter
雑誌名: Langenbecks Arch Surg. 2009 Jul;394(4):689-98. doi: 10.1007/s00423-008-0392-5. Epub 2008 Jul 24.
Abstract/Text INTRODUCTION: Alveolar echinococcosis (AE) is life-threatening and reports on surgical procedures and results are rare, but essential.
MATERIALS AND METHODS: Longitudinal surveillance and long-term follow-up of patients surgically treated for AE during the periods 1982-1999 (group A) and 2000-2006 (group B).
SETTING: University hospital within an endemic area.
RESULTS: The median (min-max) follow-up period was 141 (5-417) months. Forty-eight surgical procedures were performed in 36 patients with AE: 63% were partial resections of the liver (additional extrahepatic resection in ten of them), 17% just extrahepatic resections, 10% biliodigestive anastomosis, and 10% exploratory laparotomies. Seventy-five percent of the operations were first-time procedures, 25% done due to a relapse. Forty-two percent of the operations were estimated to be curative (R0), whereas 58% were palliative (R1, R2). All patients had additional medical treatment and periodical follow-up. Two out of 18 (11%) patients, estimated to have had curative surgery, developed a relapse 42 and 54 months later. R0-resection rates depended on the primary, neighboring, metastasis stage of AE (S1, 100%; S2, 100%; S3a, 33%; S3b, 27%; S4, 11%). During the period 2000-2006 elective radical surgery for AE was done only if a safe distance of at least 2 cm was attainable. This concept was associated with an increased R0-resection rate of 87% for group B compared to 24% for group A. Operative procedures done to control complicated courses of AE (jaundice, cholangitis, vascular compression, bacterial superinfection) have not been curative (R2) in 82% because the disease had spread into irresectable structures. Morbidity was 19%. All patients with curative resections are alive. Fifty-six percent of the patients with palliative treatment are alive as long as 14-237 months, 28% died from AE 164-338 months after diagnosis (late lethality), and 17% died due to others diseases 96-417 months after diagnosis of AE. One out of seven (14%) patients suffering from suppurative parasitic necrosis died because it was impossible to control systemic sepsis (3% hospital lethality).
CONCLUSION: Curative surgery for AE is feasible if the parasitic mass is removable entirely. The earlier the stage, the more frequent is R0 resectability. The observance of a minimal safe distance increases the rate of R0 resections. The benefit of palliative surgery is uncertain due to favorable long-term results of medical treatment alone. However, necrotic tissue is at risk of bacterial superinfection, which can cause life-threatening sepsis. Palliative surgery is an option to treat complications, which could not be managed otherwise.

PMID 18651165  Langenbecks Arch Surg. 2009 Jul;394(4):689-98. doi: 10.・・・
著者: Enrico Brunetti, Peter Kern, Dominique Angèle Vuitton, Writing Panel for the WHO-IWGE
雑誌名: Acta Trop. 2010 Apr;114(1):1-16. doi: 10.1016/j.actatropica.2009.11.001. Epub 2009 Nov 30.
Abstract/Text The earlier recommendations of the WHO-Informal Working Group on Echinococcosis (WHO-IWGE) for the treatment of human echinococcosis have had considerable impact in different settings worldwide, but the last major revision was published more than 10 years ago. Advances in classification and treatment of echinococcosis prompted experts from different continents to review the current literature, discuss recent achievements and provide a consensus on diagnosis, treatment and follow-up. Among the recognized species, two are of medical importance -Echinococcus granulosus and Echinococcus multilocularis - causing cystic echinococcosis (CE) and alveolar echinococcosis (AE), respectively. For CE, consensus has been obtained on an image-based, stage-specific approach, which is helpful for choosing one of the following options: (1) percutaneous treatment, (2) surgery, (3) anti-infective drug treatment or (4) watch and wait. Clinical decision-making depends also on setting-specific aspects. The usage of an imaging-based classification system is highly recommended. For AE, early diagnosis and radical (tumour-like) surgery followed by anti-infective prophylaxis with albendazole remains one of the key elements. However, most patients with AE are diagnosed at a later stage, when radical surgery (distance of larval to liver tissue of >2cm) cannot be achieved. The backbone of AE treatment remains the continuous medical treatment with albendazole, and if necessary, individualized interventional measures. With this approach, the prognosis can be improved for the majority of patients with AE. The consensus of experts under the aegis of the WHO-IWGE will help promote studies that provide missing evidence to be included in the next update.

Copyright 2009 Elsevier B.V. All rights reserved.
PMID 19931502  Acta Trop. 2010 Apr;114(1):1-16. doi: 10.1016/j.actatro・・・
著者: L Ma, A Ito, Y H Liu, X G Wang, Y Q Yao, D G Yu, Y T Chen
雑誌名: Trans R Soc Trop Med Hyg. 1997 Jul-Aug;91(4):476-8.
Abstract/Text Eleven cases of alveolar echinococcosis (Echinococcus multilocularis infection) with non-resectable lesions but treated with albendazole for 17 to 69 months were followed-up clinically and serologically for 4.5-11.5 years. Based on the clinical outcome and computerized tomography (CT) scanning, they were divided into 4 groups of 2 cured cases, 5 stabilized cases, 3 cases with recurrences, and one treatment failure. Forty-seven sequentially collected sera from the 11 cases were analysed by sequential enzyme-linked immunosorbent assay (ELISA) using Em2plus antigen (Em2plus-ELISA) and Western blotting to detect antibody response against Em18 (Em18-Western blots). The antibody levels in one of the cured and 2 of the stabilized cases fell below the cut-off level in the Em2plus-ELISA 4.5-6 years after effective treatment, whereas all other cases, including 2 of those with recurrences, showed large reductions initially but increased again during the follow-up period. Em18-Western blots of the 2 cured cases and 2 of the stabilized cases became negative. IgG subclasses with responses against Em18 which fell to zero included IgG1 (2), IgG3 (one) and IgG4 (one). All other cases showed no decrease in antibody response against Em18. There were, in general, reasonably reliable correlations between the success or failure of chemotherapy and antibody responses by Em2plus-ELISA and Em18-Western blots. These results suggest that both Em2plus-ELISA and Em18-Western blot are potentially useful in evaluating and predicting the efficacy of chemotherapy.

PMID 9373660  Trans R Soc Trop Med Hyg. 1997 Jul-Aug;91(4):476-8.
著者: Yoshinori Fujimoto, Akira Ito, Yuji Ishikawa, Mitsutaka Inoue, Yasuaki Suzuki, Masumi Ohhira, Takaaki Ohtake, Yutaka Kohgo
雑誌名: J Gastroenterol. 2005 Apr;40(4):426-31. doi: 10.1007/s00535-004-1559-7.
Abstract/Text Alveolar echinococcosis (AE) is a rare parasitic disease caused by Echinococcus multicularis and most commonly involves the liver. Early diagnosis is essential to improve the prognosis of patients with AE of the liver. Em18, an 18-kD diagnostic antigen from Echinococcus multilocularis, is highly specific and sensitive to detect AE. We previously reported that an enzyme-linked immunosorbent assay (ELISA) system using a recombinant Em18 antigen (RecEm18) was highly useful in the differential serodiagnosis of AE. In this report, we present seven AE patients who showed dynamic changes in RecEm18-ELISA values in the course of long-term follow up of albendazole (ABZ) chemotherapy, and/or resections of the liver or bone metastasis. All seven AE patients revealed positive values, over the cutoff level, of the RecEm18-ELISA before the treatments. The values in six patients fell below the cutoff level after the treatments, but the value in a patient with recurrence never fell below the cutoff level, and increased again. From these results, it seems that the RecEm18-ELISA is useful to evaluate the efficacy of treatment and predict recurrence in patients with AE. RecEm18-ELISA may be an important examination for: (a) the mass screening of AE in Japan, (b) the confirmative diagnosis of AE prior to surgical and/or chemotherapeutic treatments, (c) the follow up of AE patients after treatments, and (d) for deciding on the discontinuation of chemotherapy in patients with an appropriate response.

PMID 15870979  J Gastroenterol. 2005 Apr;40(4):426-31. doi: 10.1007/s0・・・
著者:
雑誌名: Bull World Health Organ. 1996;74(3):231-42.
Abstract/Text Summarized in this article are recent experiences in the treatment of human cystic echinococcosis (CE) and alveolar echinococcosis (AE) of the liver caused by the metacestode stages of Echinococcus granulosus and E. multilocularis, respectively. For CE, surgery remains the first choice for treatment with the potential to remove totally the parasite and completely cure the patient. However, chemotherapy with benzimidazole compounds (albendazole or mebendazole) and the recently developed PAIR procedure (puncture-aspiration-injection-re-aspiration) with concomitant chemotherapy offer further options for treatment of CE cases. Chemotherapy is not yet satisfactory: cure can be expected in about 30% of patients and improvement in 30-50%, after 12 months' follow-up. AE is generally a severe disease, with over 90% mortality in untreated patients. Radical surgery is recommended in all operable cases but has to be followed by chemotherapy for at least 2 years. Inoperable cases and patients who have undergone nonradical resection or liver transplantation require continuous chemotherapy for many years. Long-term chemotherapy may significantly prolong survival, even for inoperable patients with severe AE. Liver transplantation may be indicated as a life-saving measure for patients with severe liver dysfunction, but is associated with a relatively high risk of proliferation of intraoperatively undetected parasite remnants. Details of indications, contraindications, treatment schedules and other aspects are discussed.

PMID 8789923  Bull World Health Organ. 1996;74(3):231-42.
著者: H Ishizu, J Uchino, N Sato, S Aoki, K Suzuki, H Kuribayashi
雑誌名: Hepatology. 1997 Mar;25(3):528-31. doi: 10.1002/hep.510250305.
Abstract/Text Patients with alveolar echinococcosis of the liver (AEL) can be cured by complete excision of the lesions; however, it is not always completely resectable in advanced cases. Recently, benzimidazole-type drugs have been reported to be effective in nonresectable AEL. One hundred fifty-two patients with AEL have been surgically treated in our institution since 1937. Our clinical trial with albendazole, one of the benzimidazole carbamates, has included 26 cases of AE since September 1988, each of whom had undergone an operation. Complete resection was performed in only six cases. Evaluation of response to the treatment was possible in 20 cases. A favorable response to albendazole, such as decreases in the size of the lesions, changes in cyst morphology, and amelioration in clinical symptoms or signs, was achieved in 11 (55%) cases. These favorable responses were also seen in cases of noncurative resection and palliative operation. The cumulative survival rate of the patients was 87%, 15 years after the operation. A complete response was achieved in one case; the residual lesion in the liver completely disappeared on the computed tomography image 3.5 years after noncurative surgery. Palliative or mass reduction surgery combined with albendazole therapy may be a strategy for advanced disease, especially when complete resection might result in significant morbidity or mortality.

PMID 9049192  Hepatology. 1997 Mar;25(3):528-31. doi: 10.1002/hep.510・・・

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