Neurology GeneticsPathologyPeer-reviewed journal article
Immune cell mitochondrial phenotypes are largely preserved in mitochondrial diseases and do not reflect disease severity
Cynthia C. LiuiD, Mangesh KuradeiD, Anna Sophia MonzeliD, Catherine A. KellyiD, Robert‐Paul JusteriD, Caroline TrumpffiD, Michio HiranoiD, Martin A. PicardiD
Abstract
ObjectiveTo profile immune cell mitochondrial phenotypes in mitochondrial diseases (MitoD) and evaluate how those phenotypes relate to disease manifestations or biomarkers. MethodsWe profiled mitochondrial content and OxPhos enzymatic activities in isolated monocytes, lymphocytes, neutrophils, platelets, and mixed peripheral blood mononuclear cells (PBMCs) from 37 individuals with MitoD (m.3243A>G, n=23; single, large-scale mitochondrial DNA (mtDNA) deletions, n=14) and 68 healthy women and men from the MiSBIE study. ResultsWe first confirm and quantify robust cell type differences in mitochondrial content, activities of OxPhos complexes I, II, and IV, and the mitochondrial respiratory capacity (MRC) index. In relation to MitoD, neither mitochondrial content nor OxPhos capacity were consistently affected, other than a mild monocyte-specific reduction in complex I (partially mtDNA encoded) relative to complex II (entirely nDNA encoded), consistent with the mtDNA defects examined. Relative to the large differences in cell type-specific mitochondrial phenotypes, differences in MitoD relative to controls were generally small (<25%) across mitochondrial measures. The MitoD biomarkers GDF15 and FGF21, as well as clinical disease severity measures, were most strongly related to mitochondrial abnormalities in platelets, and most weakly related to mitochondrial OxPhos capacity in lymphocytes, which are known to eliminate mtDNA defects. Finally, comparing PBMCs collected in the morning/fasted state to the afternoon/fed state following a stressful experience, we report significant time-dependent changes in mitochondrial biology over the time scale of hours. ConclusionsOverall, these results demonstrate that the dynamic and cell-type specific mitochondrial phenotypes are preserved in mitochondrial diseases and are generally unrelated to the severity of MitoD symptoms.
- Citations
- 5
- FWCI
- 8.37
- Field percentile
- top 3%
Reproducibility ledger
Nobody has reported trying to reproduce this paper yet. Did you replicate an experiment or run the code? Your report saves the next lab a week.
Sign in to file a reproducibility report.
Related papers
- Mitochondrial phenotypes in purified human immune cell subtypes and cell mixtures
S. Rausser, C. Trumpff, … M. Picard·bioRxiv·Oct 16, 2020
- Leukocyte cytokine responses in adult patients with mitochondrial DNA defects
K. R. Karan, C. Trumpff, … M. Picard·bioRxiv·Dec 13, 2021
- A stagewise response to mitochondrial dysfunction in mitochondrial DNA maintenance disorders
A. E. Vincent, C. Chen, … C. Lawless·Biochimica et Biophysica Acta (BBA) - Molecular Basis of DiseasePeer-reviewed·Jun 1, 2024
- OxPhos Dysfunction Causes Hypermetabolism and Reduces Lifespan in Cells and in Patients with Mitochondrial Diseases
G. Sturm, K. R. Karan, … M. Picard·bioRxiv·Nov 29, 2021
- Mitochondrial leakage and mtDNA damage trigger early immune response in Inclusion Body Myositis.
Felix Kleefeld, Emily Cross, … Rita Horváth·bioRxiv·Aug 5, 2024
- Multilevel impairment of mitochondrial respiration with sex-specific signatures in inclusion body myositis
Ibrahim Shammas, Hazem Iaali, … Elie Naddaf·bioRxiv·Feb 20, 2026