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Mukthavaram, R.

Publications and source records attributed to Mukthavaram, R..

2 recordsLinked to original sources

Pharmacologic inhibition of OLIG2 by CT-179 disrupts mitotic progression and enhances standard-of-care chemoradiotherapy in glioblastoma

Background: Glioblastoma (GBM) remains uniformly fatal. The basic helix-loop-helix transcription factor OLIG2 is universally expressed in diffuse glioma, sustains the tumor-propagating stem-like fraction, and opposes p53-mediated genotoxic responses, but has historically been considered undruggable. We characterized CT-179, a brain-penetrant small-molecule OLIG2 inhibitor, as a candidate therapeutic for GBM. Methods: Target engagement was assessed by cellular thermal shift assay, differential scanning fluorimetry, crosslinking, and electrophoretic mobility shift assay. Anti-tumor activity was evaluated across eleven patient-derived GBM cell lines, in zebrafish oligodendrocyte precursor cell (OPC)-derived tumors, and orthotopic and subcutaneous GBM models, including combinations with radiotherapy plus temozolomide and with selective Bcl-2 family inhibitors. Results: CT-179 engaged OLIG2 and altered the conformation of dimerized OLIG2 on chromatin without preventing dimerization or fully displacing OLIG2 from DNA. CT-179 reduced OLIG2-positive OPCs and OPC-derived tumors in zebrafish and inhibited proliferation across all eleven GBM lines (IC50 < 360 nM), depleting the stem-like fraction and inducing apoptosis through mitotic catastrophe, mitotic slippage, or a hybrid response. RNA-seq across three patient-derived glioma stem cell lines identified {beta}-tubulin gene isoforms as the most strongly CT-179-down-regulated transcripts at the early time point and showed correlated {beta}-tubulin and OLIG2 expression in TCGA glioblastoma specimens. Combination with the Mcl-1 inhibitor S63845 or the Bcl-2/Bcl-xL/Bcl-w inhibitor ABT-737 produced greater-than-additive cytotoxicity. Oral CT-179 crossed the blood-brain barrier and extended median survival as a single agent across two independent adult orthotopic patient-derived backgrounds (GBM8 and G06); intraperitoneal CT-179 was associated with reduced intratumoral OLIG2 and Ki-67 staining in GBM8 orthotopic xenografts. Combination of CT-179 with radiotherapy and temozolomide eliminated all GBM8 and 7 of 8 GBM4 subcutaneous xenografts without weight loss and significantly extended survival when combined with radiotherapy in MGMT-unmethylated GBM6 orthotopic xenografts (CT-179 administered intraperitoneally). Conclusions: CT-179 is a brain-penetrant inhibitor of an established GBM driver with single-agent and combination activity across several orthotopic models, supporting clinical development for GBM and translational evaluation in other OLIG2-dependent CNS malignancies.

cancer biology↗

Hepatocyte-specific activity of TSC22D4 triggers progressive NAFLD by impairing mitochondrial function

ObjectiveFibrotic organ responses have recently been identified as long-term complication in diabetes. Indeed, insulin resistance and aberrant hepatic lipid accumulation represent driving features of progressive non-alcoholic fatty liver disease (NAFLD), ranging from simple steatosis and non-alcoholic steatohepatitis (NASH) to fibrosis. Effective pharmacological regimens to stop progressive liver disease are still lacking to-date. MethodsBased on our previous discovery of transforming growth factor beta-like stimulated clone (TSC)22D4 as a key driver of insulin resistance and glucose intolerance in obesity and type 2 diabetes, we generated a TSC22D4-hepatocyte specific knockout line (TSC22D4-HepaKO) and exposed mice to control or NASH diet models. Mechanistic insights were generated by metabolic phenotyping and single cell liver sequencing. ResultsHepatic TSC22D4 expression was significantly correlated with markers of liver disease progression and fibrosis in both murine and human livers. Indeed, hepatic TSC22D4 levels were elevated in human NASH patients as well as in several murine NASH models. Specific genetic deletion of TSC22D4 in hepatocytes led to reduced liver lipid accumulation, improvements in steatosis and inflammation scores and decreased apoptosis in mice. Single cell RNA sequencing revealed a distinct gene signature identifying an upregulation of mitochondrial-related processes. An enrichment of genes involved in the TCA cycle, mitochondrial organization, and triglyceride metabolism underscored the hepatocyte-protective phenotype and overall decreased liver damage as seen in mouse models. ConclusionsTogether, our data uncover a new connection between targeted depletion of TSC22D4 and intrinsic metabolic processes in progressive liver disease. Cell-specific reduction of TSC22D4 improves hepatic steatosis, inflammation and promotes hepatocyte survival thus paving the way for further preclinical therapy developments.

physiology↗