Mitochondrial DNA Instability Supersedes Parkin Mutations in Driving Mitochondrial Proteomic Alterations and Functional Deficits in Polg Mutator Mice.


Journal

International journal of molecular sciences
ISSN: 1422-0067
Titre abrégé: Int J Mol Sci
Pays: Switzerland
ID NLM: 101092791

Informations de publication

Date de publication:
11 Jun 2024
Historique:
received: 19 04 2024
revised: 30 05 2024
accepted: 07 06 2024
medline: 27 6 2024
pubmed: 27 6 2024
entrez: 27 6 2024
Statut: epublish

Résumé

Mitochondrial quality control is essential in mitochondrial function. To examine the importance of Parkin-dependent mechanisms in mitochondrial quality control, we assessed the impact of modulating Parkin on proteome flux and mitochondrial function in a context of reduced mtDNA fidelity. To accomplish this, we crossed either the Parkin knockout mouse or ParkinW402A knock-in mouse lines to the Polg mitochondrial mutator line to generate homozygous double mutants. In vivo longitudinal isotopic metabolic labeling was followed by isolation of liver mitochondria and synaptic terminals from the brain, which are rich in mitochondria. Mass spectrometry and bioenergetics analysis were assessed. We demonstrate that slower mitochondrial protein turnover is associated with loss of mtDNA fidelity in liver mitochondria but not synaptic terminals, and bioenergetic function in both tissues is impaired. Pathway analysis revealed loss of mtDNA fidelity is associated with disturbances of key metabolic pathways, consistent with its association with metabolic disorders and neurodegeneration. Furthermore, we find that loss of Parkin leads to exacerbation of Polg-driven proteomic consequences, though it may be bioenergetically protective in tissues exhibiting rapid mitochondrial turnover. Finally, we provide evidence that, surprisingly, dis-autoinhibition of Parkin (ParkinW402A) functionally resembles Parkin knockout and fails to rescue deleterious Polg-driven effects. Our study accomplishes three main outcomes: (1) it supports recent studies suggesting that Parkin dependence is low in response to an increased mtDNA mutational load, (2) it provides evidence of a potential protective role of Parkin insufficiency, and (3) it draws into question the therapeutic attractiveness of enhancing Parkin function.

Identifiants

pubmed: 38928146
pii: ijms25126441
doi: 10.3390/ijms25126441
pii:
doi:

Substances chimiques

DNA Polymerase gamma EC 2.7.7.7
Ubiquitin-Protein Ligases EC 2.3.2.27
parkin protein EC 2.3.2.27
DNA, Mitochondrial 0
Polg protein, mouse EC 2.7.7.7
Proteome 0
Mitochondrial Proteins 0

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Subventions

Organisme : Michael J. Fox Foundation
ID : 11845

Auteurs

Andrew J Trease (AJ)

Department of Neurological Sciences, University of Nebraska Medical Center, Omaha, NE 68198, USA.

Steven Totusek (S)

Department of Neurological Sciences, University of Nebraska Medical Center, Omaha, NE 68198, USA.

Eliezer Z Lichter (EZ)

Computational Biomedicine Section, Department of Medicine, Boston University School of Medicine, Boston, MA 02118, USA.

Kelly L Stauch (KL)

Department of Neurological Sciences, University of Nebraska Medical Center, Omaha, NE 68198, USA.

Howard S Fox (HS)

Department of Neurological Sciences, University of Nebraska Medical Center, Omaha, NE 68198, USA.

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Classifications MeSH