RRC ID 51527
著者 Cooper JF, Machiela E, Dues DJ, Spielbauer KK, Senchuk MM, Van Raamsdonk JM.
タイトル Activation of the mitochondrial unfolded protein response promotes longevity and dopamine neuron survival in Parkinson's disease models.
ジャーナル Sci Rep
Abstract While the pathogenesis of Parkinson's disease (PD) is incompletely understood, mitochondrial dysfunction is thought to play a crucial role in disease pathogenesis. Here, we examined the relationship between mitochondrial function and dopamine neuron dysfunction and death using C. elegans mutants for three mitochondria-related genes implicated in monogenic PD (pdr-1/PRKN, pink-1/PINK1 and djr-1.1/DJ-1). We found that pdr-1 and pink-1 mutants exhibit deficits in dopamine-dependent behaviors, but no loss of dopamine neurons, while djr-1.1 mutants showed an increased sensitivity to oxidative stress. In examining mitochondrial morphology and function, we found that djr-1.1 mutants exhibit increased mitochondrial fragmentation leading to decreased rate of oxidative phosphorylation and ATP levels. pdr-1 and pink-1 mutants show an accumulation of dysfunctional mitochondria with age, which leads to activation of the mitochondrial unfolded protein response (mitoUPR). Preventing the upregulation of the mitoUPR with a deletion in atfs-1 results in decreased lifespan and dopamine neuronal loss in pdr-1 and pink-1 mutants but not in wild-type worms. Overall, our results suggest that mutations in pdr-1 and pink-1 cause the accumulation of dysfunctional mitochondria, which activates the mitoUPR to mitigate the detrimental effect of these mutations on dopamine neuron survival.
巻・号 7(1)
ページ 16441
公開日 2017-11-27
DOI 10.1038/s41598-017-16637-2
PII 10.1038/s41598-017-16637-2
PMID 29180793
PMC PMC5703891
MeSH Animals Caenorhabditis elegans / metabolism* Caenorhabditis elegans Proteins / genetics Caenorhabditis elegans Proteins / metabolism Cell Death / drug effects Cell Survival / drug effects Disease Models, Animal Dopamine / pharmacology Dopaminergic Neurons / drug effects Dopaminergic Neurons / metabolism Dopaminergic Neurons / pathology* Green Fluorescent Proteins / metabolism Longevity* / drug effects Mitochondria / drug effects Mitochondria / metabolism* Mutation / genetics Nerve Degeneration / metabolism Nerve Degeneration / pathology Oxidative Stress / drug effects Parkinson Disease / pathology* Unfolded Protein Response* / drug effects
IF 3.998
引用数 22
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リソース情報
線虫 tm1346 tm918