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      Fast-Growing Alveolar Echinococcosis Following Lung Transplantation

      case-report

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          Abstract

          Alveolar echinococcosis is a rare but life-threatening infection caused by the parasite Echinococcus multilocularis. Its natural history is characterized by a slow parasitic growth over several years. Increased incidence and shorter development delay have been reported in immune-compromised patients. We report the reactivation of aborted lesions within 12 months of lung transplantation leading to a fast-growing aggressive hepatic lesion. Timely identification of alveolar echninococcosis allowed prompt albendazole treatment and radical surgery leading to a favorable outcome 42 months after transplantation. However, close clinical, serological and radiological monitoring is required to rule out relapses in the long term. The pre-existence of aborted self-limited lesions of alveolar echinococcosis and the possibility for their atypical rapid growth in patients undergoing profound immunosuppression should be known by healthcare providers, even if working in non-endemic areas.

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          Most cited references22

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          Echinococcosis: Advances in the 21st Century

          SUMMARY 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 In terms of surgery, new procedures have superseded traditional techniques, and total cystectomy in CE, ex vivo resection with autotransplantation in AE, and percutaneous and perendoscopic procedures in both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.
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            Expert consensus for the diagnosis and treatment of cystic and alveolar echinococcosis in humans.

            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.
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              The Echinococcoses: Diagnosis, Clinical Management and Burden of Disease.

              The echinococcoses are chronic, parasitic diseases that are acquired after ingestion of infective taeniid tapeworm eggs from certain species of the genus Echinococcus. Cystic echinococcosis (CE) occurs worldwide, whereas, alveolar echinococcosis (AE) is restricted to the northern hemisphere, and neotropical echinococcosis (NE) has only been identified in Central and South America. Clinical manifestations and disease courses vary profoundly for the different species of Echinococcus. CE presents as small to large cysts, and has commonly been referred to as 'hydatid disease', or 'hydatidosis'. A structured stage-specific approach to CE management, based on the World Health Organization (WHO) ultrasound classification of liver cysts, is now recommended. Management options include percutaneous sterilization techniques, surgery, drug treatment, a 'watch-and-wait' approach or combinations thereof. In contrast, clinical manifestations associated with AE resemble those of a 'malignant', silently-progressing liver disease, with local tissue infiltration and metastases. Structured care is important for AE management and includes WHO staging, drug therapy and long-term follow-up for at least a decade. NE presents as polycystic or unicystic disease. Clinical characteristics resemble those of AE, and management needs to be structured accordingly. However, to date, only a few hundreds of cases have been reported in the literature. The echinococcoses are often expensive and complicated to treat, and prospective clinical studies are needed to better inform case management decisions.
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                Author and article information

                Journal
                Pathogens
                Pathogens
                pathogens
                Pathogens
                MDPI
                2076-0817
                16 September 2020
                September 2020
                : 9
                : 9
                : 756
                Affiliations
                [1 ]Service de Pneumologie, Hôpital Louis Pradel, Hospices Civils de Lyon, 69500 Bron, France; clarisse.dupont@ 123456chu-lyon.fr (C.D.); jean-francois.mornex@ 123456chu-lyon.fr (J.-F.M.)
                [2 ]Unité de Sérologies Parasitaires et Fongiques, Laboratoire de Biologie Médicale, Centre Hospitalier Régional Universitaire, 25000 Besançon, France; fgrenouillet@ 123456chu-besancon.fr
                [3 ]UMR6249 CNRS-UBFC Chrono-Environnement, Université de Bourgogne-Franche-Comté, 25000 Besançon, France
                [4 ]Service de Chirurgie Digestive et de Transplantation Hépatique, Hôpital de la Croix-Rousse, Hospices Civils de Lyon, 69317 Lyon, France; jean-yves.mabrut@ 123456chu-lyon.fr
                [5 ]Inserm, CRCL UMR1052, Université de Lyon, Université Lyon 1, 69373 Lyon, France
                [6 ]Service de Radiologie, Hôpital Edouard Herriot, Hospices Civils de Lyon, 69003 Lyon, France; frederique.gay@ 123456chu-lyon.fr
                [7 ]Institut des Agents Infectieux, Parasitologie Mycologie, Hôpital de la Croix-Rousse, Hospices Civils de Lyon, 69317 Lyon, France; florence.persat@ 123456chu-lyon.fr (F.P.); martine.wallon@ 123456chu-lyon.fr (M.W.)
                [8 ]Physiologie Intégrée du Système D’éveil, Centre de Recherche en Neurosciences de Lyon, INSERM U1028-CNRS UMR 5292, Centre Hospitalier Le Vinatier, Université de Lyon, Université Lyon 1, 69675 Bron, France
                [9 ]INRAE, IVPC, UMR754, Université de Lyon, Université Lyon 1, 69007 Lyon, France
                [10 ]Service de Pneumologie, Hôpital de la Croix Rousse, Hospices Civils de Lyon, 69317 Lyon, France; francois.philit@ 123456chu-lyon.fr
                Author notes
                [* ]Correspondence: damien.dupont@ 123456chu-lyon.fr ; Tel.: +33-4-72-00-15-20; Fax: +33-4-72-07-18-73
                Author information
                https://orcid.org/0000-0001-6001-3135
                https://orcid.org/0000-0002-6992-1073
                Article
                pathogens-09-00756
                10.3390/pathogens9090756
                7559376
                32948027
                10c55163-4f38-4060-b245-0ab8a057006a
                © 2020 by the authors.

                Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license ( http://creativecommons.org/licenses/by/4.0/).

                History
                : 14 August 2020
                : 14 September 2020
                Categories
                Case Report

                echinococcus multilocularis,parasite,liver,lung transplantation,immunosuppression

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