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Biology subjects

Klein Geltink, R.

Publications and source records attributed to Klein Geltink, R..

3 recordsLinked to original sources

Molecular and metabolomic characterization of hiPSC-derived cardiac fibroblasts transitioning to myofibroblasts

1.Mechanical stress and pathological signaling trigger the activation of fibroblasts to myofibroblasts, which impacts extracellular matrixcomposition, disrupts normal wound healing,andcan generate deleterious fibrosis (Bohl et al., 2008; Sutton and Sharpe, 2000). Myocardial fibrosis independently promotes cardiac arrhythmias, sudden cardiac arrest, and contributes to the severity of heart failure (Frangogiannis, 2021). Fibrosis can also alter cell-to-cell communication and increase myocardial stiffness which eventually may lead to lusitropic and inotropic cardiac dysfunction (PMID: 33135058). Human induced pluripotent stem cell derived cardiac fibroblasts (hiPSC-CFs) have the potential to enhance clinical relevance in precision disease modeling by facilitating the study of patient-specific phenotypes. However, it is unclear whether hiPSC-CFs can be activated to become myofibroblasts akin to primary cells, and the key signaling mechanisms in this process remain unidentified. We hypothesize that the passaging of hiPSC-CFs, like primary cardiac fibroblasts, induces specific genes required for myofibroblast activation and increased mitochondrial metabolism. Passaging of hiPSC-CFs from passage 0 to 3 (P0 to P3) and treatment of P0 with TGF{beta}1 was associated with a gradual induction of genes to initiate the activation of these cells to myofibroblasts, including collagen, periostin, fibronectin, and collagen fiber processing enzymes with concomitant downregulation of cellular proliferation markers. Most importantly, canonical TGF{beta}1 and Hippo signaling component genes including TAZ were influenced by passaging hiPSC-CFs. Seahorse assay revealed that passaging and TGF{beta}1 treatment increased mitochondrial respiration, consistent with fibroblast activation requiring increased energy production, whereas treatment with the glutaminolysis inhibitor BPTES completely attenuated this process. Based on these data, the hiPSC-CF passaging enhanced fibroblast activation, activated fibrotic signaling pathways, and enhanced mitochondrial metabolism approximating what has been reported in primary cardiac fibroblasts. Thus, hiPSC-CFs may provide an accurate in vitro preclinical model for the cardiac fibrotic condition, which may facilitate the identification of putative anti-fibrotic therapies, including patient-specific approaches. HighlightsO_LIPassaging promotes the activation of fibroblasts to myofibroblasts. C_LIO_LITGF{beta}1 treatment activates the fibroblasts, but their expression profile was uniquely different from myofibroblasts. C_LIO_LIHigh energy requiring fibroblast activation is dependent on glutaminase-based mitochondrial metabolism. C_LIO_LIPassaging induces TGF{beta}1 and Hippo signaling pathways in activated fibroblasts and myofibroblasts. C_LI Graphical Abstract CaptionProbing the activation of fibroblasts to myofibroblasts is key in ECM remodeling processes to avoid fibrosis-related adverse complications, and to better understand disease pathology. Here we report that passaging of hiPSC-derived cardiac fibroblasts promotes fibroblast activation along with a gradual shift in gene expression and metabolic changes towards myofibroblasts. TGF{beta}1 treatment activates non-passaged fibroblasts, but they are dissimilar to myofibroblasts. The energy-intensive fibroblast to myofibroblast activation process is dependent on glutaminase-mediated mitochondrial metabolism and is prevented by treatment with GLS-1 inhibitor BPTES. Our work demonstrates that hiPSC-CFs can offer a preclinical model analogous to primary cardiac fibroblasts that is comparable with passage-mediated myofibroblast activation and increased mitochondrial metabolism. hiPSC-CFs may also facilitate patient-specific novel anti-fibrosis drug screening and disease management. O_FIG O_LINKSMALLFIG WIDTH=200 HEIGHT=154 SRC="FIGDIR/small/561455v2_ufig1.gif" ALT="Figure 1"> View larger version (33K): org.highwire.dtl.DTLVardef@139f3a9org.highwire.dtl.DTLVardef@1abea35org.highwire.dtl.DTLVardef@19d7bfborg.highwire.dtl.DTLVardef@369e9e_HPS_FORMAT_FIGEXP M_FIG C_FIG

cell biology↗

Loss of FBXO11 function establishes a stem cell program in acute myeloid leukemia through dysregulation of the mitochondrial protease LONP1

Acute myeloid leukemia (AML) is an aggressive cancer with very poor outcomes. To identify additional drivers of leukemogenesis, we analyzed sequence data from 1,727 unique individual AML patients, which revealed mutations in ubiquitin ligase family genes in 11.2% of adult AML samples with mutual exclusivity. The Skp1/Cul1/Fbox (SCF) E3 ubiquitin ligase complex gene FBXO11 was the most significantly downregulated gene of the SCF complex in AML. FBXO11 catalyzes K63-linked ubiquitination of a novel target, LONP1, which promotes entry into mitochondria, thereby enhancing mitochondrial respiration. Reduced mitochondrial respiration secondary to FBXO11 depletion imparts myeloid-biased stem cell properties in primary CD34+ hematopoietic stem progenitor cells (HSPC). In a human xenograft model, depletion of FBXO11 cooperated with AML1-ETO and mutant KRASG12D to generate serially transplantable AML enriched for primitive cells. Our findings suggest that reduced FBXO11 primes HSPC for myeloid-biased self-renewal through attenuation of LONP1-mediated regulation of mitochondrial respiration.

cancer biology↗

In macrophages fatty acid oxidation spares glutamate for use in diverse metabolic pathways required for alternative activation

Fatty acid oxidation (FAO) is upregulated in IL-4-stimulated (alternatively activated) macrophages (M(IL-4)). We examined the effect of loss of function of the enzyme Cpt1a, which facilitates the entry of long chain fatty acids (FA) into mitochondria for FAO, on alternative activation. Expression of M(IL-4) markers ARG1, CD301 and RELM, was impaired in tamoxifen-treated ERT2Cre x Cpt1afl/fl macrophages and in macrophages expressing shRNA targeting Cpt1a (Cpt1a-shRNA). In contrast, VaviCre x Cpt1afl/fl and LysmCre x Cpt1afl/fl M(IL-4) responded normally to IL-4. Reduced alternative activation due to Cpt1a loss of function was linked to decreased cellular pools of -ketoglutarate, glutamate, and glutathione, diminished commitment of glucose carbon to serine/glycine synthesis, and decreased expression of genes in the Nrf2-oxidative stress response pathway. Consistent with this, reactive oxygen species were increased. Restoration of glutathione pools with N-acetyl cysteine normalized oxidative stress and allowed alternative activation in the face of Cpt1a-deficiency, pointing to a role for FAO in the control of ROS and as being important for alternative activation. In VaviCre x Cpt1afl/fl M(IL-4), glutamine uptake was increased, compensating for the loss of FAO to meet necessary metabolic demands, to allow alternative activation. The data indicate that macrophages are able to regulate glutamine metabolism to compensate for chronic disruption of FAO to meet metabolic needs.

immunology↗