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Terhune, S. S.

Publications and source records attributed to Terhune, S. S..

11 recordsLinked to original sources

Data-Driven Mechanistic Computational Modeling of Human Cell Cycle Phases for Defining Inhibitor Combinations in Silico

The human cell cycle is a highly regulated process that integrates multiple signaling pathways and checkpoints to ensure faithful genome duplication and cell division. Disruptions in these regulatory networks contribute to a wide range of diseases. Here, we present a novel, updateable computational model of the full human cell cycle that shows sustained oscillations over time and reproduces experimental perturbations. We used a hybrid framework combining mass action and Michaelis-Menten kinetics, incorporating the synthesis, degradation, and regulation of key cell cycle proteins and protein complexes. It consists of 63 distinct biochemical species, interacting through 41 major reactions, and functioning through 63 ODEs. The model is built upon a modular framework, structured around the core regulatory networks of the G1, S, G2, and M phases. Due to its complexity, we determined parameter sets that met strict criteria, namely event timing, comparable concentrations, and continuous cycling. We validated the models behavior by reproducing canonical checkpoint responses, including mitogen dependence and the DNA damage response, both of which produced reversible and robust cell cycle arrests. Importantly, the model was trained and calibrated using in vitro data from human U251-MG glioma cells expressing the FastFUCCI cell cycle reporter. We quantitatively aligned the simulated and experimentally determined phase durations and cell doubling times. Next, we experimentally tested and refined model parameters by using abemaciclib-mediated inhibition of CDK4 and volasertib-mediated inhibition of PLK1. In vitro and in silico data show dose-dependent G1 arrest by abemaciclib and dose-dependent mitotic arrest by volasertib. Finally, we demonstrated that the model predicts changes in cell proliferation over a wide range of drug concentrations and combinations. Overall, our work establishes a robust, data-driven computational model for systems-level analysis of the human cell cycle and its disruption by therapeutic perturbations. AUTHOR SUMMARYKnowledge of the protein-protein interaction networks governing the cell cycle is ever-expanding, yet this information is often fragmented across studies focusing on disconnected subsets of the cycle. For decades, researchers have investigated the underlying mechanisms of cell division, but an integrated, quantitative understanding of the entire process remains elusive. This gap is a major hurdle for predicting how targeted therapies affect cell proliferation, especially when used in combination. Our goal is to develop an in silico simulation of the complete human cell cycle by integrating the key mechanistic relationships across all four phases into a single computational model with enough resolution to approximate outcomes upon perturbation. In achieving this, we have developed a novel, comprehensive computational model that provides an integrated quantitative understanding of how cancer drugs alter the human cell cycle. We have rigorously trained, calibrated, and validated the model by suitably estimating its parameter values to produce accurate cell cycle phase timing, using high-resolution, live-cell imaging data and other cardinal features of the cell cycle in a U251-MG glioblastoma line. This work provides an accessible tool for exploring how normal cell cycle control is disrupted in disease, generating new hypotheses, and identifying potential points of therapeutic intervention.

cancer biology↗

Human cytomegalovirus regulates host DNA repair machinery for viral genome integrity

The DNA damage response (DDR) encompasses a multitude of interconnected pathways that serve as a cellular defense to protect genome integrity. Dysregulation or failure of these pathways results in cancers and genetic disease. DNA viruses, including the herpesvirus cytomegalovirus (CMV), activate DDR signaling during their replicative program. The mechanisms by which they commandeer these responses for replication of their genome is less well understood. Here, we define a viral protein, UL138, that modulates the activity of host DDR pathways. The loss of UL138 results in structural variants, including inversion, deletions, duplication, with signature of homology-directed repair and other DDR pathways. The actions of UL138 are due, in part, to its modulation of pathways regulated by the cellular deubiquitinating complex that targets proliferating cell nuclear antigen (PCNA) and Fanconi Anemia effectors, FANCD2 and FANCI. However, we also show that UL138 accesses pathways independent of USP1-PCNA/FANCD2/FANCI. Disruption of UL138 or these pathways impacts viral genome replication and had consequences for viral genome integrity. This work provides mechanistic insight into the long-standing questions of how DNA viruses recruit, modulate and use cellular DDR pathways. It also puts forth CMV as a model system for further defining these pathways in human cells. GRAPHICAL ABSTRACT O_FIG O_LINKSMALLFIG WIDTH=200 HEIGHT=108 SRC="FIGDIR/small/683233v1_ufig1.gif" ALT="Figure 1"> View larger version (23K): org.highwire.dtl.DTLVardef@121e55borg.highwire.dtl.DTLVardef@58d8b7org.highwire.dtl.DTLVardef@fc1628org.highwire.dtl.DTLVardef@12b32a_HPS_FORMAT_FIGEXP M_FIG C_FIG Major findingsO_LIUL138 protects HCMV genome integrity through its interaction with USP1-PCNA/FANCD2/FANCI and repeat sequence-mediated pathways C_LIO_LIUL138 protects HCMV genome integrity in the UL region by mechanisms independent of repeat sequences and USP1-PCNA/FANCD2/FANCI C_LIO_LICollectively, US and UL regions of the HCMV genome have fundamentally different pathways acting on them and UL138 modulates DNA damage and repair pathway choice. C_LI

microbiology↗

The effective multiplicity of infection for HCMV depends on activity of the cellular 20S proteasome

Human cytomegalovirus (HCMV) is a betaherpesvirus capable of infecting numerous cell types and persisting throughout an infected individuals life. Disease usually occurs in individuals with compromised or underdeveloped immune systems. Several antivirals exist but have limitations relating to toxicity and resistance. HCMV replication involves upregulation of host proteasomal activities which play important roles in the temporal stages of replication. Here, we defined the impact on replication kinetics of the proteasome inhibitor, bortezomib. We demonstrate that bortezomib significantly reduces levels of viral genomes and infectious virions produced from a population of cells. Inhibition reduced expression of viral proteins that are influenced by genome synthesis. When added prior to 24 hpi, we observe decreases in PCNA and Cdk1 while increases in p21 whose regulations contribute to efficient replication. This response synergized with an antiviral, maribavir. Since some replication occurred, we tested the hypothesis that a subset of infected cells might break through inhibition. Initially, we simulated bortezomib activities using a mechanistic computational model of late-lytic replication. Upon reducing MOI in-silico, we observed near identical simulated results compared to experimental data. Next, we analyzed replication using live-cell imaging. This revealed treated cultures do contain a population of cells with fully developed late-stage cytoplasmic assembly compartments but at significantly lower numbers. We refer to this as the effective MOI. Overall, our studies support a hypothesis in which 20S proteasome inhibition disrupts HCMV replication by reducing the MOI to an effective MOI, defined by a fraction of infected cells capable of progressing to fulminant infection. IMPORTANCEHCMV infection and reactivation continues to contribute to morbidity and mortality around the world. Antiviral compounds are available but have limitations. Here, we have defined the impact of the proteasome inhibitor bortezomib on HCMV replication. Proteasomal activities play a critical role in temporal changes required for replication. We demonstrate that disrupting these activities inhibits viral replication while likely supporting increased antiviral activity of the anti-HCMV agent, maribavir. Using a combination of live cell imaging and computational tools, we discover that a subset of infected cells progresses to fulminant infection which we define as the effective MOI, and this subset would otherwise be missed when analyzing the average of the population.

microbiology↗

Stabilizing microtubules aids neurite structure and disrupts syncytia formation in human cytomegalovirus-infected human forebrain neurons

Human cytomegalovirus (HCMV) is a prolific human herpesvirus that infects most individuals by adulthood. While typically asymptomatic in adults, congenital infection can induce serious neurological symptoms including hearing loss, visual deficits, cognitive impairment, and microcephaly in 10-15% of cases. HCMV has been shown to infect most neural cells with our group recently demonstrating this capacity in stem cell-derived forebrain neurons. Infection of neurons induces deleterious effects on calcium dynamics and electrophysiological function paired with gross restructuring of neuronal morphology. Here, we utilize an iPSC-derived model of the human forebrain to demonstrate how HCMV infection induces syncytia, drives neurite retraction, and remodels microtubule networks to promote viral production and release. We establish that HCMV downregulates microtubule associated proteins at 14 days postinfection while simultaneously sparing other cytoskeletal elements, and this includes HCMV-driven alterations to microtubule stability. Further, we pharmacologically modulate microtubule dynamics using paclitaxel (stabilize) and colchicine (destabilize) to examine the effects on neurite structure, syncytial morphology, assembly compartment formation, and viral release. With paclitaxel, we found improvement of neurite outgrowth with a corresponding disruption to HCMV-induced syncytia formation and Golgi network disruptions but with limited impact on viral titers. Together, these data suggest that HCMV infection-induced disruption of microtubules in human cortical neurons can be partially mitigated with microtubule stabilization, suggesting a potential avenue for future neuroprotective therapeutic exploration. IMPORTANCEInfection by human cytomegalovirus (HCMV) continues to cause significant damage to human health. In the absence of a vaccine, vertical transmission from mother to fetus can result in profound neurological damage impacting quality of life. These studies focus on understanding the impact of HCMV infection on forebrain cortical neurons derived from iPSCs. We show that infection results in loss of neurite extension accompanied by cell-to-cell fusion. These pathogenic changes involve HCMV infection-mediated disruption to the microtubule network. Upon addition of the microtubule stabilization agent paclitaxel, the structural damage was limited, but infection still progressed to produce infectious particles. This work is part of our continued efforts to define putative strategies to limit HCMV-induced neurological damage.

neuroscience↗

Replication efficiencies of human cytomegalovirus-infected epithelial cells are dependent on source of virus production

Human cytomegalovirus (HCMV) is a prevalent betaherpesvirus, and infection can lead to a range of symptomatology from mononucleosis to sepsis in immunocompromised individuals. HCMV is also the leading viral cause of congenital birth defects. Lytic replication is supported by many cell types with different kinetics and efficiencies leading to a plethora of pathologies. The goal of these studies was to elucidate HCMV replication efficiencies for viruses produced on different cell types upon infection of epithelial cells by combining experimental approaches with data-driven computational modeling. HCMV was generated from a common genetic background of TB40-BAC4, propagated on fibroblasts (TB40Fb) or epithelial cells (TB40Epi), and used to infect epithelial cells. We quantified cell-associated viral genomes (vDNA), protein levels (pUL44, pp28), and cell-free titers over time for each virus at different multiplicities of infection. We combined experimental quantification with data-driven simulations and determined that parameters describing vDNA synthesis were similar between sources. We found that pUL44 accumulation was higher in TB40Fb than TB40Epi. In contrast, pp28 accumulation was higher in TB40Epi which coincided with a significant increase in titer for TB40Epi over TB40Fb. These differences were most evident during live-cell imaging, which revealed syncytia-like formation during infection by TB40Epi. Simulations of the late lytic replication cycle yielded a larger synthesis constant for pp28 in TB40Epi along with increase in virus output despite similar rates of genome synthesis. By combining experimental and computational modeling approaches, our studies demonstrate that the cellular source of propagated virus impacts viral replication efficiency in target cell types. IMPORTANCEHuman cytomegalovirus (HCMV) is a ubiquitous pathogen that can cause serious disease under conditions of immunodeficiency and upon congenital infection. HCMV replicates in diverse cell types throughout the human body with tropism influenced by the source of the virus. Here, we investigated the contribution of viral sources to the kinetics of HCMV replication in epithelial cells using both experimental and mechanistic computational modeling approaches. These studies reveal that HCMV produced from epithelial cells exhibits a higher efficiency of replication despite similar viral DNA synthesis kinetics between viral sources. These differences likely involve a propensity of epithelial-derived virus to induce syncytia versus fibroblast-derived virus, and an accompanying higher synthesis rate of a late virion protein ultimately resulting in production of more extracellular infectious virus.

microbiology↗

Human Cytomegalovirus Induces Significant Structural and Functional Changes in Terminally Differentiated Human Cortical Neurons

Human cytomegalovirus (HCMV) is a highly prevalent viral pathogen that typically presents asymptomatically in healthy individuals despite lifelong latency. However, in 10-15% of congenital cases, this beta-herpesvirus demonstrates direct effects on the central nervous system, including microcephaly, cognitive/learning delays, and hearing deficits. HCMV has been widely shown to infect neural progenitor cells, but the permissiveness of fully differentiated neurons to HCMV is controversial and chronically understudied, despite potential associations between HCMV infection with neurodegenerative conditions. Using a model system representative of the human forebrain, we demonstrate that induced pluripotent stem cell (iPSC)-derived, excitatory glutamatergic and inhibitory GABAergic neurons are fully permissive to HCMV, demonstrating complete viral replication, competent virion production, and spread within the culture. Interestingly, while cell proliferation was not induced in these post-mitotic neurons, HCMV did increase expression of proliferative markers Ki67 and PCNA suggesting alterations in cell cycle machinery. These finding are consistent with previous HCMV-mediated changes in various cell types and implicate the virus ability to alter proliferative pathways to promote virion production. HCMV also induces significant structural changes in forebrain neurons, such as the formation of syncytia and retraction of neurites. Finally, we demonstrate that HCMV disrupts calcium signaling and decreases neurotransmission, with action potential generation effectively silenced after 15 days post infection. Taken together, our data highlight the potential for forebrain neurons to be permissive to HCMV infection in the CNS, which could have significant implications on overall brain health and function.

neuroscience↗

Neutralizing Antibodies with Neurotropic Factor Treatment Maintain Neurodevelopmental Gene Expression Upon Exposure to Human Cytomegalovirus

Human cytomegalovirus (HCMV) is a beta herpesvirus that causes severe congenital birth defects including microcephaly, vision loss, and hearing loss. Infection of cerebral organoids with HCMV causes significant downregulation of genes involved in critical neurodevelopmental pathways. The precise features of the infection causing this dysregulation remain unknown. Entry of HCMV into human cells is determined by the composition of glycoproteins in viral particles, which is influenced by the source of the virus. This includes a trimer complex and a pentamer complex with the latter enriched from replication in epithelial cells. To begin dissecting which features contribute to neuronal pathogenesis, we evaluated infection using virus from different sources along with the distribution of cellular entry receptors on cells in cerebral organoids. We observed significant increases in the number of viral genomes, viral spread and penetrance, and multinucleated syncytia in neural tissues infected with HCMV propagated in epithelial cells compared to fibroblasts. To determine if this was related to entry receptor distribution, we measured expressions of cellular entry receptors and observed similar distributions of all receptors on cells obtained from organoids indicating that source of virus is likely the key determinant. Next, we asked whether we could limit pathogenesis using neutralization antibodies. We found that pre-treatment with antibodies against viral glycoprotein B (gB) and gH successfully decreased viral genome levels, viral gene expression, and virus-induced syncytia. In contrast, targeting specific cellular entry receptors failed to limit infection. Using an antibody against gB, we also observed partial protection of developmental gene expression that was further improved by the addition of brain derived neurotropic factor (BDNF). These studies indicate that source of HCMV is a key determinant of neuronal pathogenesis that can be limited by neutralization antibodies and neurotropic factors.

neuroscience↗

Human Cytomegalovirus UL138 Interaction with USP1 Activates STAT1 in infection

Innate immune responses are crucial for limiting virus infection. However, viruses often hijack our best defenses for viral objectives. Human Cytomegalovirus (HCMV) is a beta herpesvirus which establishes a life-long latent infection. Defining the virus-host interactions controlling latency and reactivation is vital to the control of viral disease risk posed by virus reactivation. We defined an interaction between UL138, a pro-latency HCMV gene, and the host deubiquintase complex, UAF1-USP1. UAF1 is a scaffold protein pivotal for the activity of ubiquitin specific peptidases (USP), including USP1. UAF1-USP1 sustains an innate immune response through the phosphorylation and activation of signal transducer and activator of transcription-1 (pSTAT1), as well as regulates the DNA damage response. After the onset of viral DNA synthesis, pSTAT1 levels are elevated and this depends upon UL138 and USP1. pSTAT1 localizes to viral centers of replication, binds to the viral genome, and influences UL138 expression. Inhibition of USP1 results in a failure to establish latency, marked by increased viral genome replication and production of viral progeny. Inhibition of Jak-STAT signaling also results in increased viral genome synthesis in hematopoietic cells, consistent with a role for USP1-mediated regulation of STAT1 signaling in the establishment of latency. These findings demonstrate the importance of the UL138-UAF1-USP1 virus-host interaction in regulating HCMV latency establishment through the control of innate immune signaling. It will be important going forward to distinguish roles of UAF1-USP1 in regulating pSTAT1 relative to its role in the DNA damage response in HCMV infection. ImportanceHuman cytomegalovirus (HCMV) is one of nine herpesviruses that infect humans. Following a primary infection, HCMV establishes a life-long latent infection that is marked by sporadic, and likely frequent reactivation events. While these reactivation events are asymptomatic in the immune competent host, they pose important disease risks for the immune compromised, including solid organ or stem cell transplant recipients. Its complex interactions with host biology and deep coding capacity make it an excellent model for defining mechanisms important for viral latency and reactivation. Here we define an interaction with host proteins that commandeer typically antiviral innate immune signaling for the establishment of latency.

microbiology↗

Nitric Oxide Attenuates Human Cytomegalovirus Infection yet Disrupts Neural Cell Differentiation and Tissue Organization

Human cytomegalovirus (HCMV) is a prevalent betaherpesvirus that is asymptomatic in healthy individuals but can cause serious disease in immunocompromised patients. HCMV is also the leading cause of viral-mediated birth defects. Many of these defects manifest within the central nervous system and include microcephaly, sensorineural hearing loss, and cognitive developmental delays. Nitric oxide is a critical effector molecule produced as a component of the innate immune response during infection. Using a 3-dimensional cortical organoid model, we demonstrate that nitric oxide inhibits HCMV spread and simultaneously disrupts neural rosette structures resulting in tissue disorganization. Nitric oxide also attenuates HCMV replication in 2-dimensional cultures of neural progenitor cells (NPCs), a prominent cell type in cortical organoids that differentiate into neurons and glial cells. The multipotency factor SOX2 was decreased during nitric oxide exposure, suggesting early neural differentiation is affected. Maximal mitochondrial respiration was also reduced in both uninfected and infected NPCs. We determined this reduction likely influences neural differentiation as neurons (Tuj1+GFAP-Nestin-) and glial populations (Tuj1-GFAP+Nestin-) were reduced following differentiation. We also observed changes in calcium signaling during exposure to nitric oxide with increased cellular response to ATP (purinergic receptors) and KCl (voltage gated calcium channels). Importantly, nitric oxide could not rescue HCMV-mediated defects in calcium response. Our studies indicate a prominent, immunopathogenic role of nitric oxide in promoting developmental defects within the brain despite its antiviral activity during congenital HCMV infection. Author summaryHuman cytomegalovirus (HCMV) infection can result in serious disease to immunocompromised individuals. HCMV is also the leading cause of viral-mediated congenital birth defects. Congenitally-infected infants can have a variety of symptoms, including microcephaly, sensorineural hearing loss, and developmental delays. The use of 3-dimensional (3-D) cortical organoids to model infection of the fetal brain has advanced the current understanding of developmental defects and allowed a broader investigation of the mechanisms behind disease. Here, we investigate the effect of nitric oxide, a critical effector molecule, on cortical development and HCMV infection. We demonstrate that nitric oxide plays an antiviral role during infection yet results in significant disorganization to cortical tissue. Despite inhibiting viral replication in neural progenitor cells, nitric oxide contributes to differentiation defects of these cells and does not rescue functional consequences of HCMV infection on calcium signaling. Our results indicate that immunopathogenic consequences of nitric oxide during congenital infection promote developmental defects that undermine its antiviral activity.

microbiology↗

Downregulation of Neurodevelopmental Gene Expression in iPSC-Derived Cerebral Organoids Upon Infection by Human Cytomegalovirus

Human cytomegalovirus (HCMV) is a beta herpesvirus that, upon congenital infection, can cause severe birth defects including vision and hearing loss, microcephaly, and seizures. Currently, no approved treatment options exist for in utero infections. We previously demonstrated that HCMV infection decreases calcium signaling responses and alters neuronal differentiation in induced pluripotent stem cell (iPSC) derived neural progenitor cells (NPCs). Here we aimed to determine the impact of infection on the transcriptome in developing human neurons using iPSC-derived 3-dimensional cerebral organoids. We infected iPSC-derived cerebral organoids with HCMV encoding eGFP and sorted cell populations based on GFP signal strength. Significant transcriptional downregulation was observed including in key neurodevelopmental gene pathways in both the GFP (+) and intermediate groups. Interestingly, the GFP (-) group also showed downregulation of the same targets indicating a mismatch between GFP expression and viral infection. Using a modified HCMV virus destabilizing IE 1 and 2 proteins, we still observed significant downregulation of neurodevelopmental gene expression in infected neural progenitor cells. Together, these data indicate that IE viral proteins are not the main drivers of neurodevelopmental gene dysregulation in HCMV infected neural tissues suggesting therapeutically targeting IE gene expression is insufficient to restore neural differentiation and function.

cell biology↗

TNF-α induces reactivation of human cytomegalovirus independently of myeloid cell differentiation following post-transcriptional establishment of latency

We used the Kasumi-3 model to study HCMV latency and reactivation in myeloid progenitor cells. Kasumi-3 cells were infected with HCMV strain TB40/Ewt-GFP, flow sorted for GFP+ cells, and cultured for various times to monitor establishment of latency, as judged by repression of viral gene expression (RNA/DNA ratio) and loss of virus production. We found that, in the vast majority of cells, latency was established post-transcriptionally in the GFP+ infected cells: transcription was initially turned on, and then turned off. We also found that some of the GFP-cells were infected, suggesting that latency might be established in these cells at the outset of infection. We were not able to test this hypothesis because some GFP-cells expressed lytic genes, and thus, it was not possible to separate them from GFP-quiescent cells. In addition, we found that the pattern of expression of lytic genes that have been associated with latency, including UL138, US28, and RNA2.7, was the same as that of other lytic genes, indicating that there was no preferential expression of these genes once latency is established. We confirmed previous studies showing that TNF- induced reactivation of infectious virus, and by analyzing expression of the progenitor cell marker CD34 as well as myeloid cell differentiation markers in IE+ cells after treatment with TNF-, we showed that TNF- induced transcriptional reactivation of IE gene expression independently of differentiation. TNF--mediated reactivation in Kasumi-3 cells was correlated with activation of NF-{kappa}B, KAP-1 and ATM.\n\nIMPORTANCEHCMV is an important human pathogen that establishes lifelong latent infection in myeloid progenitor cells, and reactivates frequently to cause significant disease in immunocompromised people. Our observation that viral gene expression is first turned on, and then turned off to establish latency suggests that there is a host defense, which may be myeloid-specific, responsible for transcriptional silencing of viral gene expression. Our observation that TNF- induces reactivation independently of differentiation provides insight into molecular mechanisms that control reactivation.

microbiology↗