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Modulation of SLP-2 expression protects against alpha-synuclein neuropathology by mitigating mitochondrial dysfunction

Bolduc, C.; Castelo Rueda, M. P.; Lorente Picon, M.; Gentile, G.; Kalvakuri, S.; Soto Linan, V.; Zanon, A.; Laouafa, S.; Lang, M.; Lavdas, A. A.; Gilmozzi, V.; Bernardo, G.; Coulombe, V.; Gora, C.; Peralta, M.; Rioux, V.; Honisch, C.; Pramstaller, P. P.; Ruzza, P.; Ziviani, E.; Bodmer, R.; Hicks, A. A.; Parent, M.; Soliz, J.; Joseph, V.; Pichler, I.; Levesque, M.

2025-06-15 neuroscience
10.1101/2025.06.13.659577 bioRxiv
Show abstract

Parkinsons Disease (PD) is a progressive neurodegenerative disorder characterized by dopaminergic neuron loss and the accumulation of alpha-synuclein (Syn)-rich aggregates known as Lewy bodies. Mitochondrial dysfunction is a key contributor to PD pathology, and mitochondrial defects are part of the pathogenic mechanisms induced by Syn. Stomatin-Like protein 2 (SLP-2) is a mitochondrial scaffold protein that regulates mitochondrial integrity and function. Here, we investigated whether SLP-2 induction can counteract Syn-induced mitochondrial dysfunction and neurodegeneration. We found that SLP-2 levels were reduced in human PD brains and an A53T Syn mouse model. Mild overexpression of SLP-2 improved mitochondrial function, reduced oxidative stress, and prevented Syn-mitochondria interactions in human iPSC-derived neurons. In vivo, SLP-2 overexpression protected dopaminergic neurons and motor function, while its depletion exacerbated degeneration and motor deficits in both mouse and Drosophila models. These findings suggest SLP-2 as a key regulator of mitochondrial resilience and a potential therapeutic target for PD and alpha-synucleinopathies.

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