Morphological profiling reveals neuroprotection via mitochondrial uncoupling in human dopaminergic neurons

Summary

Parkinson's disease (PD) involves multiple pathological processes in midbrain dopaminergic (mDA) neurons, including protein degradation defects, vesicular trafficking disruption, endolysosomal dysfunction, mitochondrial issues, and oxidative stress. Current PD models often lack complexity and focus on single phenotypes. We used patient-derived SNCA triplication (SNCA-4x) and isogenic control (SNCA-corr) mDA neurons, applying high-content imaging-based morphological profiling to identify and rescue multiple phenotypes. Screening 1,020 compounds, we identified top-scoring compounds that restored healthy profiles in SNCA-4x neurons, increasing Tyrosine hydroxylase (TH) and decreasing α-synuclein (αSyn) levels. Several hits were linked to mitochondrial biology. Tyrphostin A9, a mitochondrial uncoupler, and several of its structural analogues decreased ROS levels, normalized mitochondrial membrane potential, and increased respiration. Western blotting confirmed that Tyrphostin A9 reduces αSyn levels. Our study highlights the neuroprotective potential of mild mitochondrial uncoupling in mDA neurons. © 2025. The Author(s).

Authors Gorgogietas V, Weiss A, Cousin L, Hoffmann D, Schmitt K, Ogier A, Barbuti PA, Santos BFR, Boussaad I, Wittich A, Zaliani A, Pless O, Krüger R, Sommer P, Wilbertz JH
Journal Scientific reports
Publication Date 2025 Aug 12;15(1):29507
PubMed 40796766
PubMed Central PMC12344149
DOI 10.1038/s41598-025-14735-0

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