Slota Jessy A, Lamoureux Lise, Myskiw Jennifer, Frost Kathy L, Medina Sarah J, Kielich Dominic M S, Leonhardt Melanie, Thapar Gunjan, Bailey-Elkin Ben A, Booth Stephanie A
Mycobacteriology, Vector-Borne and Prion Diseases Division, National Microbiology Laboratory, Public Health Agency of Canada, Winnipeg, MB, Canada.
Department of Medical Microbiology and Infectious Diseases, Faculty of Health Sciences, University of Manitoba, Winnipeg, MB, Canada.
Acta Neuropathol Commun. 2025 Apr 30;13(1):86. doi: 10.1186/s40478-025-01999-w.
Cultured brain slices rapidly replicate murine prions, exhibit prion pathology, and are amenable towards drug discovery, but have not been infected with human prions. As deer mice (Peromyscus maniculatus) are susceptible to human prions in vivo, here we investigated deer mouse organotypic brain slice cultures as a potential model of human prion disease. Deer mouse brain slices supported replication of rodent-adapted strains of scrapie and Creutzfeldt-Jakob disease (CJD), but they resisted infection with primary human prion inoculum. To better understand this discrepancy, we quantified prion replication rates, characterized cellular and molecular changes, and estimated inoculum clearance within wildtype CD1 and deer mouse brain slice cultures. Prion replication rates varied by species, strain, and brain region, independently of PrP sequence homology. Scrapie-infected CD1 cerebellar slice cultures exhibited the fastest prion replication rate, closely matching in vivo bioassay kinetics and showing neuronal and synaptic degeneration at similar timepoints. However, deer mouse slice cultures replicated deer mouse-adapted sCJD MM1 prions less efficiently than in vivo inoculation. These findings clarify both the utility and constraints of brain slice cultures in modeling prion disease and imply that the slice culture molecular environment may be suboptimal for human prion replication.
培养的脑片能快速复制鼠朊病毒,呈现朊病毒病理特征,且适用于药物研发,但尚未感染过人朊病毒。由于鹿鼠(白足鼠)在体内对人朊病毒易感,我们在此研究了鹿鼠器官型脑片培养物作为人朊病毒疾病潜在模型的情况。鹿鼠脑片支持啮齿动物适应株的羊瘙痒病和克雅氏病(CJD)的复制,但它们抵抗原发性人朊病毒接种物的感染。为了更好地理解这种差异,我们量化了朊病毒复制率,表征了细胞和分子变化,并估计了野生型CD1和鹿鼠脑片培养物中的接种物清除情况。朊病毒复制率因物种、毒株和脑区而异,与朊蛋白(PrP)序列同源性无关。感染羊瘙痒病的CD1小脑片培养物表现出最快的朊病毒复制率,与体内生物测定动力学密切匹配,并在相似时间点出现神经元和突触退化。然而,鹿鼠片培养物复制适应鹿鼠的散发性克雅氏病MM1朊病毒的效率低于体内接种。这些发现阐明了脑片培养物在朊病毒疾病建模中的效用和局限性,并表明片培养的分子环境可能对人朊病毒复制并非最优。