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Observational Study
. 2015 Sep 21;9(9):e0003964.
doi: 10.1371/journal.pntd.0003964. eCollection 2015.

Outcome after Discontinuing Long-Term Benzimidazole Treatment in 11 Patients with Non-resectable Alveolar Echinococcosis with Negative FDG-PET/CT and Anti-EmII/3-10 Serology

Rudolf W Ammann  1 , Katrin D M Stumpe  2 , Felix Grimm  3 , Peter Deplazes  3 , Sabine Huber  1 , Kaja Bertogg  1 , Dorothee R Fischer  4 , Beat Müllhaupt  5
Affiliations
Observational Study

Outcome after Discontinuing Long-Term Benzimidazole Treatment in 11 Patients with Non-resectable Alveolar Echinococcosis with Negative FDG-PET/CT and Anti-EmII/3-10 Serology

Rudolf W Ammann et al. PLoS Negl Trop Dis. .

Abstract

Background/aims: Benzimidazoles are efficacious for treating non-resectable alveolar echinococcosis (AE), but their long-term parasitocidal (curative) effect is disputed. In this study, we prospectively analyzed the potential parasitocidal effect of benzimidazoles and whether normalization of FDG-PET/CT scans and anti-Emll/3-10-antibody levels could act as reliable "in vivo" parameters of AE-inactivation permitting to abrogate chemotherapy with a low risk for AE-recurrence.

Method: This prospective study included 34 patients with non-resectable AE subdivided into group A (n = 11), followed-up after diagnosis and begin of chemotherapy at months 6, 12 and 24, and group B (n = 23) with a medium duration of chemotherapy of 10 (range 2-25) years. All patients were assessed by FDG-PET/CT examinations and anti-EmII/3-10 serology. Chemotherapy was abrogated in patients with normalization of FDG-PET/CT and serum anti-EmII/3-10 levels. These patients were closely followed-up for AE recurrence. Endpoint (parasitocidal efficacy) was defined by the absence of AE-recurrence >24 months after stopping treatment.

Results: Normalization of FDG-PET/CT scan and anti-EmII/3-10 levels occurred in 11 of 34 patients (32%). After abrogation of chemotherapy in these 11 patients, there was no evidence of AE-recurrence within a median of 70.5 (range 16-82) months. However, the patients' immunocompetence appears pivotal for the described long-term parasitocidal effect of benzimidazoles.

Conclusions: The combination of negative FDG-PET/CT-scans and anti-EmII/3-10 antibody levels seem to be reliable parameters for assessing in vivo AE-larval inactivity after long-term benzimidazole chemotherapy.

Trial registration: clinicaltrials.gov: NCT00658294.

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Conflict of interest statement

The authors have declared that no competing interests exist.

Figures

Fig 1
Fig 1. Disposition of the study population.
Fig 2
Fig 2. (A9): Baseline and follow-up FDG-PET/CT scans without i.v. contrast.
a) Large and small AE-lesions (arrow) at the time of diagnosis (2003) with very strongly increased FDG uptake. b) The lesions became FDG-negative within two years of albendazole treatment. c) Progressive calcifications of AE lesions over the next 6 years after abrogation of chemotherapy (2011). No signs of FDG uptake are present.
Fig 3
Fig 3. (A5): Long-term follow-up of a large AE recurrence (Sept 2001), 3 years following presumed radical surgery).
Albendazole was administered from Sept. 2001 until February 2006. Oligosymptomatic choledocholithiasis was treated by ERCP with papillotomy and stone extraction in April 2008. The procedure was complicated by cholangitis, which was treated by long-term antibiotic therapy. No AE-recurrence was noted during 70 months of follow-up after stopping albendazole treatment.
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