Lipopolysaccharide and Recombinant Prion Protein Induce Distinct Neurodegenerative Pathologies in FVB/N Mice

Prion diseases are classically attributed to the accumulation of protease-resistant prion protein (PrP<sup>Sc</sup>); however, recent evidence suggests that alternative misfolded prion conformers and systemic inflammatory factors may also contribute to neurodegeneration. This study investigated whether recombinant moPrP<sup>Res</sup>, generated by incubating wild-type mouse PrP<sup>C</sup> with bacterial lipopolysaccharide (LPS), can induce prion-like disease in FVB/N female mice, whether LPS alone causes neurodegeneration, and how LPS modulates disease progression in mice inoculated with the Rocky Mountain Laboratory (RML) strain of prions. Wild-type female FVB/N mice were randomized into six subcutaneous treatment groups: saline, LPS, moPrP<sup>Res</sup>, moPrP<sup>Res</sup> + LPS, RML, and RML + LPS. Animals were monitored longitudinally for survival, body weight, and clinical signs. Brain tissues were analyzed histologically and immunohistochemically for vacuolar degeneration, PrP<sup>Sc</sup> accumulation, reactive astrogliosis, and amyloid-β plaque deposition. Recombinant moPrP<sup>Res</sup> induced a progressive spongiform encephalopathy characterized by widespread vacuolation and astrogliosis, yet with no detectable PrP<sup>Sc</sup> by Western blot or immunohistochemistry. LPS alone triggered a distinct neurodegenerative phenotype, including cerebellar amyloid-β plaque accumulation and terminal-stage spongiosis, with approximately 40% mortality by the end of the study. Co-administration of moPrP<sup>Res</sup> and LPS resulted in variable regional pathology and intermediate survival (50% at 750 days post-inoculation). Interestingly, RML + LPS co-treatment led to earlier clinical onset and mortality compared to RML alone; however, vacuolation levels were not significantly elevated and, in some brain regions, were reduced. These results demonstrate that chronic endotoxemia and non-infectious misfolded PrP conformers can independently or synergistically induce key neuropathological hallmarks of prion disease, even in the absence of classical PrP<sup>Sc</sup>. Targeting inflammatory signaling and toxic prion intermediates may offer novel therapeutic strategies for prion and prion-like disorders.

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Publication Details

Journal
International Journal of Molecular Sciences
Published
2025-06-28
DOI
https://doi.org/10.3390/ijms26136245
Citations
1
Primary Topic
Prion Diseases and Protein Misfolding
Type
article
Field-Weighted Citation Impact
0.81

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article

Lipopolysaccharide and Recombinant Prion Protein Induce Distinct Neurodegenerative Pathologies in FVB/N Mice

David S. Wishart, Grzegorz Zwierzchowski, Burim N. Ametaj, Dagnachew Hailemariam et al.
1 citations
International Journal of Molecular Sciences
Prion Diseases and Protein Misfolding
0.81
article

Lipopolysaccharide and Recombinant Prion Protein Induce Distinct Neurodegenerative Pathologies in FVB/N Mice

David S. Wishart, Grzegorz Zwierzchowski, Burim N. Ametaj, Dagnachew Hailemariam, Seyed Ali Goldansaz, Elda Dervishi, Roman Wójcik
article en
1 citations

Abstract

Prion diseases are classically attributed to the accumulation of protease-resistant prion protein (PrP<sup>Sc</sup>); however, recent evidence suggests that alternative misfolded prion conformers and systemic inflammatory factors may also contribute to neurodegeneration. This study investigated whether recombinant moPrP<sup>Res</sup>, generated by incubating wild-type mouse PrP<sup>C</sup> with bacterial lipopolysaccharide (LPS), can induce prion-like disease in FVB/N female mice, whether LPS alone causes neurodegeneration, and how LPS modulates disease progression in mice inoculated with the Rocky Mountain Laboratory (RML) strain of prions. Wild-type female FVB/N mice were randomized into six subcutaneous treatment groups: saline, LPS, moPrP<sup>Res</sup>, moPrP<sup>Res</sup> + LPS, RML, and RML + LPS. Animals were monitored longitudinally for survival, body weight, and clinical signs. Brain tissues were analyzed histologically and immunohistochemically for vacuolar degeneration, PrP<sup>Sc</sup> accumulation, reactive astrogliosis, and amyloid-β plaque deposition. Recombinant moPrP<sup>Res</sup> induced a progressive spongiform encephalopathy characterized by widespread vacuolation and astrogliosis, yet with no detectable PrP<sup>Sc</sup> by Western blot or immunohistochemistry. LPS alone triggered a distinct neurodegenerative phenotype, including cerebellar amyloid-β plaque accumulation and terminal-stage spongiosis, with approximately 40% mortality by the end of the study. Co-administration of moPrP<sup>Res</sup> and LPS resulted in variable regional pathology and intermediate survival (50% at 750 days post-inoculation). Interestingly, RML + LPS co-treatment led to earlier clinical onset and mortality compared to RML alone; however, vacuolation levels were not significantly elevated and, in some brain regions, were reduced. These results demonstrate that chronic endotoxemia and non-infectious misfolded PrP conformers can independently or synergistically induce key neuropathological hallmarks of prion disease, even in the absence of classical PrP<sup>Sc</sup>. Targeting inflammatory signaling and toxic prion intermediates may offer novel therapeutic strategies for prion and prion-like disorders.

International Journal of Molecular SciencesVol. 26(13)
University of Alberta (CA), Agri Food and Biosciences Institute (GB), University of Warmia and Mazury in Olsztyn (PL)
University of Alberta, Alberta Livestock and Meat Agency
Good health and well-being
Openalex Percentile: Top 26%
Prion Diseases and Protein Misfolding
0.81
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