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Open Access Case report

30-yr course and favorable outcome of alveolar echinococcosis despite multiple metastatic organ involvement in a non-immune suppressed patient

Karine Bardonnet12*, Dominique A Vuitton13, Frédéric Grenouillet145, Georges A Mantion16, Eric Delabrousse17, Oleg Blagosklonov18, Jean-Philippe Miguet19 and Solange Bresson-Hadni14569

Author Affiliations

1 WHO Collaborating Centre for Prevention and Treatment of Human Echinococcosis, University Hospital, University of Franche-Comté, Besançon 25030, France

2 Department of Biochemistry, University Hospital, Besançon 25030, France

3 EA 3181 “Epithelial carcinogenesis: prognosis and prediction factors”, University Hospital and CNRS, University of Franche-Comté, Besançon 25030, France

4 Dept of Parasitology, University Hospital, Besançon 25030, France

5 UMR 6249 Chrono-Environment Joint Research Unit, University Hospital and CNRS-INRA, University of Franche-Comté, Besançon 25030, France

6 Deparment of Digestive Surgery, University Hospital, Besançon, 25030, France

7 Deparment of Radiology, University Hospital, Besançon, 25030, France

8 Department of Nuclear Medicine, University Hospital, Besançon, 25030, France

9 Department of Hepatology, University Hospital, Besançon, 25030, France

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Annals of Clinical Microbiology and Antimicrobials 2013, 12:1  doi:10.1186/1476-0711-12-1

Published: 2 January 2013

Abstract

We report the 30-yr history of a well-documented human case of alveolar echinococcosis, with a lung lesion at presentation followed by the discovery of a liver lesion, both removed by surgery. Subsequently, within the 13 years following diagnosis, metastases were disclosed in eye, brain and skull, as well as additional lung lesions. This patient had no immune suppression, and did not have the genetic background known to predispose to severe alveolar echinococcosis; it may thus be hypothesized that iterative multi-organ involvement was mostly due to the poor adherence to benzimidazole treatment for the first decade after diagnosis. Conversely, after a new alveolar echinococcosis recurrence was found in the right lung in 1994, the patient accepted to take albendazole continuously at the right dosage. After serology became negative and a fluoro-deoxy-glucose-Positron Emission Tomography performed in 2005 showed a total regression of the lesions in all organs, albendazole treatment could be definitively withdrawn. In 2011, the fluoro-deoxy-glucose-Positron Emission Tomography showed a total absence of parasitic metabolic activity and the patient had no clinical symptoms related to alveolar echinococcosis.

The history of this patient suggests that multi-organ involvement and alveolar echinococcosis recurrence over time may occur in non-immune suppressed patients despite an apparently “radical” surgery. Metastatic dissemination might be favored by a poor adherence to chemotherapy. Combined surgery and continuous administration of albendazole at high dosage may allow alveolar echinococcosis patients to survive more than 30 years after diagnosis despite multi-organ involvement.