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Lundström-Stadelmann, B.

Publications and source records attributed to Lundström-Stadelmann, B..

5 recordsLinked to original sources

A new semi-automated, motility-based screening assay for discovery of compounds with activity against the juvenile stage of Fasciola hepatica

Fasciola hepatica is a trematode parasite responsible for fasciolosis, a liver disease that affects humans and livestock worldwide. Together with other food-borne trematode infections, fasciolosis is considered a neglected tropical disease. Further, it imposes substantial agricultural losses due to infections in ruminants. No vaccine is currently available, and control heavily relies on drug treatment, especially with triclabendazole (TCBZ). However, the intensive use of TCBZ over the past four decades has led to increasing rates of treatment failures and the emergence of drug-resistant parasites. Therefore, the identification of new treatment options is an urgent priority. The currently available toolset for drug screening, however, is limited. To address this need, we established a novel, semi-automated, standardized, and objective screening assay based on motility monitoring of newly excysted juveniles using microscopic live imaging. The assay was validated by testing a panel of ten compounds with known anthelmintic properties, amongst them TCBZ (IC50: 1.5 {micro}M) and the new activator of the F. hepatica transient receptor potential melastatin (TRPM) ion channel, benzamidoquinazolinone (IC50: 1.05 {micro}M). In addition to these two compounds with known activity against F. hepatica, three compounds were identified as particularly promising with a fast onset of action and IC50 values in the nanomolar range: the salicylanilides MMV665807 (IC50: 44 nM), niclosamide (IC50: 32 nM), and its ethanolamine salt, niclosamide ethanolamine (IC50: 9 nM). Complementary live/dead staining revealed that only TCBZ displayed parasiticidal activity, while the other compounds, although leading to parasite paralysis, did not lead to parasite death within 72 hours. Scanning electron microscopy of drug treated parasites did not reveal any significant damage at concentrations corresponding to the IC50s, but strong phenotypes were visible at 20 {micro}M. The presented motility assay provides a robust method for the discovery of novel anthelmintic compounds and facilitates the ongoing effort to combat fasciolosis. Author SummaryFasciola hepatica, the common liver fluke, is a parasitic platyhelminth that infects the liver and biliary ducts of humans and livestock, causing fasciolosis, a Neglected Tropical Disease as defined by the World Health Organization. Triclabendazole is the drug of choice to treat humans and animals. However, its intensive use has led to the emergence of drug resistance resulting in treatment failures worldwide. The identification of novel drugs is therefore urgent. Here, we present a semi-automated and objective method to assess the activity of compounds on one of the key life stages of the parasite: the newly excysted juveniles (NEJ). This stage is highly motile and motility assessment can be exploited to screen for bioactive compounds. Using time-lapse imaging, we quantified NEJ movement after drug exposure. From a panel of ten tested reference anthelmintics, two known fasciolicides (triclabendazole and benzamidoquinazolinone) and three additional compounds (MMV665807, niclosamide, and niclosamide ethanolamine) displayed particularly strong activity and were selected for further investigation. This method represents a robust tool for drug screening and facilitates the discovery of new compounds against F. hepatica.

microbiology↗

Repurposing Niclosamide Ethanolamine for Alveolar Echinococcosis Reveals a Disconnect Between In Vitro Efficacy and In Vivo Outcome

BackgroundEchinococcosis is a zoonotic disease caused by cestodes of the genus Echinococcus. Alveolar echinococcosis (AE), caused by E. multilocularis, primarily affects the liver and shows infiltrative, tumor-like growth of the metacestode stage. If untreated, AE is lethal. AE remains a neglected disease with current treatments based on albendazole or mebendazole that are parasitostatic, and not curative, underscoring the need for more effective therapies. Niclosamide is a chlorinated salicylanilide derivative with proven activities against intestinal helminths but is inactive against tissue-dwelling helminths due to poor absorption and limited bioavailability. In this study, we repurposed niclosamide ethanolamine (NEN), a formulation with improved systemic exposure, for the treatment of E. multilocularis infection in vitro and in vivo. Methodology/Principal FindingsWe assessed the in vitro efficacy of niclosamide and NEN against E. multilocularis metacestode vesicles (IC50<0.2 {micro}M) and primary parasite cells (IC50<0.3 {micro}M), with active concentrations largely corresponding to NEN levels reachable in the liver. Metabolic analysis suggested that NEN acts as a mitochondrial uncoupler. Electron microscopy showed that NEN-treatments induced profound structural damage in the metacestode vesicle tissue, but mitochondrial ultrastructure was not notably affected. In mice intraperitoneally infected with E. multilocularis, NEN was orally administered during 9 weeks either alone, or in combination with albendazole. Pharmacokinetic analyses showed that NEN reached blood level concentrations above 1 {micro}M. However, the parasite burden in NEN-treated mice was not significantly reduced. Conclusions/SignificanceAlthough niclosamide and NEN demonstrated potent activity against E. multilocularis in vitro, this efficacy did not translate in the mouse model. The lack of in vivo activity could be attributed to several factors such as infection model, limited drug uptake by the parasite in the animal, or the rapid metabolization of the compound. Future studies should explore novel niclosamide derivatives and formulations to enhance efficacy against AE in vivo. Author SummaryAlveolar echinococcosis (AE) is a severe disease caused by the larval stage of the fox tapeworm Echinococcus multilocularis. The parasite forms tumor-like lesions in the liver and can spread to other organs. The currently licensed drugs for the treatment of AE are not always effective, require long-term use, and can cause side effects that frequently require treatment interruption. Therefore, safer and more efficacious treatment options are urgently needed. Niclosamide is frequently applied for the treatment of adult tapeworm infections in the intestine, but its limited uptake and low biodistribution renders the compound unsuitable for systemic treatment. In this study, we tested a non-toxic salt formulation, niclosamide ethanolamine (NEN), exhibiting improved absorption. In vitro, NEN was highly effective against E. multilocularis metacestode vesicles. It induced profound structural alterations in metacestode vesicles and impaired the mitochondrial membrane potential, and thus interfering in energy production. However, NEN was not effective against AE in experimentally infected mice. Our results suggest that NEN treatment appears promising in vitro, but to translate to the in vivo situation, new formulations and delivery strategies should be developed to increase absorption, bioavailability and metabolic stability of the compound for an effective treatment for AE.

pharmacology and toxicology↗

Triclabendazole inhibits succinate dehydrogenase in vitro

Succinate dehydrogenase (SDH, complex II), is essential for mitochondrial respiration. This study shows that triclabendazole and its primary metabolites inhibit SDH activity in mitochondria of the cestode Echinococcus multilocularis, while other tested benzimidazoles do not. Further analyses revealed that triclabendazole and metabolites also inhibit SDH in mitochondria of additional helminths (trematodes, nematodes) and mammalian cells. These findings indicate a shared mode of action and highlight potential safety risks associated with long-term triclabendazole treatment in mammals.

pharmacology and toxicology↗

Establishment and application of unbiased in vitro drug screening assays for the identification of compounds against Echinococcus granulosus s.s.

Echinococcus multilocularis and E. granulosus s.l. are the causative agents of alveolar and cystic echinococcosis, respectively. Drug treatment options for these severe and neglected diseases are limited to benzimidazoles, which are not always efficacious, and adverse side effects are reported. Thus, novel and improved treatments are needed. In this study, the previously established platform for E. multilocularis in vitro drug assessment was adapted to E. granulosus s.s.. In a first step, in vitro culture protocols for E. granulosus s.s. were established. This resulted in the generation of large amounts of E. granulosus s.s. metacestode vesicles as well as germinal layer (GL) cells. In vitro culture of these cells formed metacestode vesicles displaying structural characteristics of metacestode vesicles generated in vivo. Next, drug susceptibilities of E. multilocularis and E. granulosus s.s. protoscoleces, metacestode vesicles and GL cells were comparatively assessed employing established assays including (i) metacestode vesicle damage marker release assay, (ii) metacestode vesicle viability assay, (iii) GL cell viability assay, and (iv) protoscolex motility assay. The standard drugs albendazole, buparvaquone, mefloquine, MMV665807, monepantel, niclosamide and nitazoxanide were included. MMV665807, niclosamide and nitazoxanide were active against the parasite in all four assays against both species. MMV665807 and monepantel were significantly more active against E. multilocularis metacestode vesicles, while albendazole and nitazoxanide were significantly more active against E. multilocularis GL cells. Albendazole displayed activity against E. multilocularis GL cells, but no effects were seen in albendazole-treated E. granulosus s.s. GL cells within five days. Treatment of protoscoleces with albendazole and monepantel had no impact on motility. Similar results were observed for both species with praziquantel and its enantiomers against protoscoleces. In conclusion, in vitro culture techniques and drug screening methods previously established for E. multilocularis were successfully implemented for E. granulosus s.s., allowing comparisons of drug efficacy between the two species.

pharmacology and toxicology↗

Albendazole reduces endoplasmic reticulum stress induced by Echinococcus multilocularis in mice

BackgroundEchinococcus multilocularis causes alveolar echinococcosis (AE), a rising zoonotic disease in the northern hemisphere. Treatment of this fatal disease is limited to chemotherapy using benzimidazoles and surgical intervention, with relatively frequent disease recurrence in cases without radical surgery. Elucidating the molecular mechanisms underlying E. multilocularis infections and host-parasite interactions aids developing novel therapeutic options. This study explored an involvement of unfolded protein response (UPR) and endoplasmic reticulum-stress (ERS) during E. multilocularis infection in mice. MethodsE. multilocularis- and mock-infected C57BL/6 mice were subdivided six weeks after infection into vehicle and albendazole (ABZ) treated groups. Eight weeks later, liver tissue was collected to examine mRNA, microRNA (miR) and protein expression of UPR- and ERS-related genes. ResultsE. multilocularis infection upregulated UPR- and ERS-related proteins, including ATF6, CHOP, GRP78, ERP72, H6PD and calreticulin, whilst PERK and its target eIF2 were not affected, and IRE1 and ATF4 were downregulated. ABZ treatment in E. multilocularis infected mice reversed the increased ATF6 and calreticulin protein expression, tended to reverse increased CHOP, GRP78, ERP72 and H6PD expression, and decreased ATF4 and IRE1 expression to levels seen in mock-infected mice. The expression of miR-146a-5p (downregulated by IRE1) and miR-1839-5p (exhibiting a unique target site in the IRE1 3UTR) were significantly increased in E. multilocularis infected mice, an effect reversed by ABZ treatment. Other miRs analyzed were not altered in E. multilocularis infected mice. Conclusions and SignificanceAE causes UPR activation and ERS in mice. The E. multilocularis-induced ERS was ameliorated by ABZ treatment, indicating its effectiveness to inhibit parasite proliferation and downregulate its activity status. ABZ itself did not affect UPR in control mice. Identified miR-146a-5p and miR-1839-5p might represent biomarkers of E. multilocularis infection. Modulation of UPR and ERS, in addition to ABZ administration, could be exploited to treat E. multilocularis infection. Author summaryAlveolar echinococcosis is a zoonotic disease caused by the fox tapeworm Echinococcus multilocularis. Treatment of this fatal disease is limited to surgical intervention, preferably radical curative surgery if possible, and the use of parasitostatic benzimidazoles. It is not yet fully understood how the parasite can remain in the hosts tissue for prolonged periods, complicating the development of therapeutic applications. This work investigated an involvement of the unfolded protein response (UPR) and endoplasmic reticulum-stress (ERS) during E. multilocularis infection and upon treatment with albendazole (ABZ) in mice. The results revealed increased expression levels of the ERS sensor ATF6 and of downstream target genes in liver tissue of E. multilocularis- compared to mock-infected mice. Additionally, H6PD, generating NADPH within the endoplasmic reticulum, and the lectin-chaperone calreticulin were increased in E. multilocularis infected liver tissue while the expression of the ERS associated genes ATF4 and IRE1 were decreased. The miR-1839-5p and miR-146-p, linked to IRE1, were elevated upon E. multilocularis infection, offering potential as novel biomarkers of alveolar echinococcosis. The observed gene expression changes were at least partially reversed by ABZ treatment. Whether modulation of UPR and ERS targets can improve the therapy of alveolar echinococcosis remains to be investigated.

pathology↗