Project ID NS-MH2026_65

ThemeNS-MH

Co Supervisor 1A Dr Jacqueline Mitchell Institute of Psychiatry, Psychology & Neuroscience, School of Neuroscience, Department of Basic & Clinical NeuroscienceEmail

Co Supervisor 1B Dr Sarah Mizielinska Institute of Psychiatry, Psychology & Neuroscience, School of Neuroscience, Department of Basic & Clinical NeuroscienceEmail

Exploring whether differences in nucleocytoplasmic transport may underpin cell vulnerability in motor neuron disease and dementia

Motor neuron disease and frontotemporal disease (MND/FTD) are two overlapping devastating neurodegenerative disease, for which aggregation of TDP-43 is a core feature. However, this pathology is not present throughout the brain, but localised to specific ‘disease vulnerable’ regions. What causes this pathology, and why it is only found in certain neurons, is currently unknown. We have recently found that some of the proteins TDP-43 interacts with differ between disease vulnerable (cortex) and resistant (cerebellum) brain regions. Some of these proteins are known to play roles in the transport of cargoes to and from the cell nucleus – nucleocytoplasmic transport – a pathway already strongly implicated in MND/FTD. This project will explore the interaction of these proteins with TDP-43 in disease vulnerable and resistant brain regions, and investigate whether targeting these proteins may have therapeutic potential for disease.

The aims are:
1. To explore differences in target protein expression and TDP-43 interaction between disease vulnerable and resistant brain regions, and investigate changes in MND/FTD.
2. To assess differences in nucleocytoplasmic transport between disease vulnerable and resistant neurons
3. To investigate whether manipulating target proteins can alter nucleocytoplasmic transport and disease progression

In year 1 the student will work with mouse tissue, carrying out immunohistochemical and western blot analyses, as well as immunoprecipitation and proximity ligation assays, on disease vulnerable and resistant brain regions to address aim 1 using both control and TDP-43 disease model samples. Follow up work will then validate key targets of interest in human post-mortem samples (years 1-2). In year two, the student will use established assays to investigate nucleocytoplasmic transport in cortical, motor and cerebellar neurons derived from induced pluripotent stem cells. In year three, the student will modify selected target proteins in stem cell models and explore the impact on nucleocytoplasmic transport and disease phenotypes.

Representative Publications

Solomon S, Sampathkumar NK, Carre I, Mondal M, Chennell G, Vernon A, Ruepp M-D, Mitchell JC Heterozygous expression of the Alzheimer’s disease protective PLC2 P522R variant enhances A clearance while preserving synapses (2022) Cell. Mol. Life Sci. 79(8) 453 DOI: 10.1007/s00018-022-04473-1 Chen HJ, Mitchell JC Mechanisms of TDP-43 proteinopathy onset and propagation (2021) Int. J. Mol. Sci. 22(11): 6004 DOI: 10.3390/ijms22116004 Mitchell JC, Constable R, So E, Vance C, Scotter EL, Glover L, Hortabagyi T, Arnold ES, Ling S-C, McAlonis M, Da Cruz S, Polymenidou M, Tessarolo L, Cleveland DW, Shaw CE Wild-type human TDP-43 potentiates ALS-linked mutant TDP-43 driven progressive motor and cortical neuron degeneration with pathological features of ALS (2015) Acta Neuropath Comm. 3(36) DOI: 10.1186/s40478-015-0212-4

Casterton C, Hu Y, Martinez-Cotrina A, Barnard J, Anderson R, Janel S, Solomon D, Lafont F, Hirth F, De Nicola GF, Mizielinska S, Fanto M. Karyoptosis mediates cell death and neurodegeneration upon proteotoxic stress, Research Square pre-print (2023) https://doi.org/10.21203/rs.3.rs-3287063/v1 Rothstein JD, Baskerville V, Rapuri S, Mehlhop E, Jafar-Nejad P, Rigo F, Bennett F, Mizielinska S, Isaacs A, Coyne AN. G2C4 targeting antisense oligonucleotides potently mitigate TDP-43 dysfunction in human C9orf72 ALS/FTD induced pluripotent stem cell derived neurons. Acta Neuropathol. 2023 Nov 29;147(1):1. https://doi:10.1007/s00401-023-02652-3 Solomon, D.A., Smikle, R., Reid M.J., Mizielinska, S. (2021). Altered phase separation and cellular impact in C9orf72-linked frontotemporal dementia and amyotrophic lateral sclerosis. Frontiers in Cellular Neuroscience, 15, 121. https://doi.org/10.3389/fncel.2021.664151