Stem Cell Reports · 2019 · 94 citations · 31 references
Life SciencesNeurobiologyNeuroscienceCellular NeuroscienceMitochondrial BiologyMolecular NeuroscienceMitochondrial Function In NeuronsNeural Stem Cell BiologyNeural Stem CellsNeurodegenerative DiseasesNeurodegenerative DisordersParkinson DiseaseDopaminergic NeurodegenerationMitochondrial Structure In NeuronsNeurodegenerative Mitochondrial Dysfunction
Emerging evidence suggests that Parkinson's disease (PD), besides being an age-associated disorder, might also have a neurodevelopment component. Disruption of mitochondrial homeostasis has been highlighted as a crucial cofactor in its etiology. Here, we show that PD patient-specific human neuroepithelial stem cells (NESCs), carrying the LRRK2-G2019S mutation, recapitulate key mitochondrial defects previously described only in differentiated dopaminergic neurons. By combining high-content imaging approaches, 3D image analysis, and functional mitochondrial readouts we show that LRRK2-G2019S mutation causes aberrations in mitochondrial morphology and functionality compared with isogenic controls. LRRK2-G2019S NESCs display an increased number of mitochondria compared with isogenic control lines. However, these mitochondria are more fragmented and exhibit decreased membrane potential. Functional alterations in LRRK2-G2019S cultures are also accompanied by a reduced mitophagic clearance via lysosomes. These findings support the hypothesis that preceding mitochondrial developmental defects contribute to the manifestation of the PD pathology later in life.
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