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Truong, L.

Publications and source records attributed to Truong, L..

10 recordsLinked to original sources

Leveraging Targeted Gene Sets and Neural Networks for Zebrafish Transcriptome Extrapolation in High-Throughput Toxicogenomics

BackgroundZebrafish (Danio rerio) are a powerful vertebrate model for developmental toxicology and chemical safety assessment, yet large-scale transcriptomics in zebrafish remains limited by cost and data heterogeneity. Targeted transcriptomics offers a cost-effective alternative, but gene extrapolation methods tailored to zebrafish have not been systematically developed or evaluated. ObjectivesWhile the S1500+ platform is widely used for toxicogenomics research with rat, mouse, and human cell lines as model systems, its use in zebrafish has been limited due to data scarcity and lack of suitable bioinformatics approaches for analysis of such data. To that end, we sought to (i) curate a large zebrafish transcriptomic training data resource, and (ii) evaluate multiple machine learning strategies for reconstructing unmeasured transcriptome-wide expression profiles for data originating from the zebrafish-specific reduced representation gene set ("Zf S1500+"). MethodsWe assembled 14,924 zebrafish RNA-Seq samples covering 21,930 genes across 1,246 studies. Using the Zf S1500+ gene subset (3,062 genes), we trained and tested three extrapolation approaches: principal components regression (PCR), a locally weighted extension of PCR (PCR+), and a neural network mixture-of-experts model (NN-MoE). Model performance was assessed using mean absolute error (MAE), mean squared regression error (MSRE), and weighted variants of these metrics. ResultsExtrapolation performance using the baseline approach was strongly influenced by tissue and developmental context, with within-tissue models outperforming cross-tissue models. Errors were lowest when training and testing were conducted within the same tissue or between developmentally related tissues. Both PCR+ and NN-MoE improved upon the baseline PCR approach, with NN-MoE reducing average MAE by [~]20% and MSRE by [~]17%. Importantly, extrapolation remained reliable for the majority of genes, even when limiting output to high-confidence predictions using an empirical MAE threshold. ConclusionsWe demonstrate that targeted transcriptomics can be effectively extended to zebrafish, enabling robust transcriptome-wide extrapolation at reduced cost. The NN-MoE method provided the most substantial gains, highlighting the value of non-linear and ensemble modeling in heterogeneous datasets. These results establish a scalable framework for zebrafish toxicogenomics and suggest that accuracy will continue to improve with larger, better-annotated datasets, paving the way for broader application in chemical safety assessments.

bioinformatics↗

OmnibusX: A unified platform for accessible multi-omics analysis

OmnibusX is an integrated, privacy-centric platform that enables code-free multi-omics data analysis by bridging computational methodologies with user-friendly interfaces. Designed to overcome challenges posed by fragmented analytical tools and high computational barriers, OmnibusX consolidates workflows for diverse technologies - including bulk RNA-seq, single-cell RNA-seq, single-cell ATAC-seq, and spatial transcriptomics - into a single, cohesive application. The application integrates established open-source tools such as Scanpy, DESeq2, SciPy, and scikit-learn into transparent, reproducible pipelines, offering users control over analytical parameters. Additionally, OmnibusX features proprietary modules, including a highly accurate cell-type prediction engine and an interactive plotting editor for generating publication-quality visualizations. Available as a standalone desktop application and an enterprise edition for centralized server deployment, OmnibusX ensures all data processing is conducted locally, eliminating external data transfer and usage tracking. By lowering technical barriers and enhancing reproducibility, OmnibusX aims to accelerate biological discovery and foster robust, data-driven collaborations. A fully documented trial version is accessible at: https://omnibusx.com/apps.

bioinformatics↗

TOR and heat shock response pathways regulate peroxisome biogenesis during proteotoxic stress

Peroxisomes are versatile organelles mediating energy homeostasis and redox balance. While peroxisome dysfunction is linked to numerous diseases, the molecular mechanisms and signaling pathways regulating peroxisomes during cellular stress remain elusive. Using yeast, we show that perturbations disrupting protein homeostasis including loss of ER or cytosolic chaperone function, impairments in ER protein translocation, blocking ER N-glycosylation, or reductive stress, cause peroxisome proliferation. This proliferation is driven by increased de novo biogenesis from the ER as well as increased fission of pre-existing peroxisomes, rather than impaired pexophagy. Notably, peroxisome biogenesis is essential for cellular recovery from proteotoxic stress. Through comprehensive testing of major signaling pathways, we determine this response to be mediated by activation of the heat shock response and inhibition of Target of Rapamycin (TOR) signaling. Finally, the effects of proteotoxic stress and TOR inhibition on peroxisomes are also captured in human fibroblasts. Overall, our findings reveal a critical and conserved role of peroxisomes in cellular response to proteotoxic stress.

cell biology↗

Clearing the air on pollutant disruptions of the gut-brain axis: Developmental exposure to Benzopyrene disturbs zebrafish behavior and the gut microbiome in adults and subsequent generations

Developmental exposure to benzo[a]pyrene (BaP), a ubiquitous environmental pollutant, has been linked to various toxic effects, including neurodegenerative disorders and, most recently, multigenerational behavioral impairment. While the specific mechanisms driving BaP neurotoxicity are not fully understood, recent work highlights two important determinants of developmental BaP neurotoxicity: (1) the aryl hydrocarbon receptor (AHR), which is responsible for inducing host metabolism of BaP, and (2) the gut microbiome, which may interact with BaP to affect its metabolism, or be perturbed directly by BaP to yield disruptions to the gut-brain axis. To explore the role of AHR, the gut microbiome, and their interaction on BaP-induced neurotoxicity, we utilized the zebrafish model. We sought to determine (1) how exposure to BaP and developmental expression of AHR2, a key gene in the zebrafish AHR pathway, link to adult zebrafish behavior, (2) how these same variables associated with the structure and function of the adult zebrafish gut metagenome, and (3) whether these associations were multigenerational. This finding revealed a reticulated axis of association between BaP exposure, developmental AHR2 expression, the zebrafish gut metagenome, and behavior. Our results also indicate that AHR2 is a key mediator of how BaP elicits neurotoxicity and microbiome dysbiosis. Additionally, this axis of association manifests inter- and transgenerationally, suggesting that exposure to BaP may yield dysbiotic and neurodevelopmental impacts on subsequent generations. These findings demonstrate the power of utilizing the zebrafish model to study pollutant-metagenome interactions and elucidate the role of specific host genes in the neurotoxicity and dysbiosis. ImportanceBenzo[a]pyrene (BaP) is a toxic chemical that is especially ubiquitous in industrialized nations, as it is produced in large quantities by burning coal, oil, and other organic compounds. Early-life and in utero exposure to this chemical can induce adverse behavior changes in model animals, like mice and zebrafish. In humans, developmental exposure is linked to symptoms of ADHD, anxiety, and depression. This study found BaP affects zebrafish behavior and the gut microbiome throughout their life and across subsequent generations. We further discovered that a host regulatory pathway called the aryl hydrocarbon receptor is a key component of how benzo[a]pyrene, the gut microbiome, and behavior interconnect. The aryl hydrocarbon receptor binds many toxicants, as well as microbial metabolites associated with maintaining gut homeostasis, and regulates the gut-brain connection. This receptor presents a new potential mechanism of how gut microbiota relate to BaP exposure and behavior for future studies to investigate.

microbiology↗

Epigenetic control of multiple genes with a single lentiviral vector encoding transcriptional repressors fused to compact zinc finger arrays

Gene silencing without gene editing holds great potential for the development of safe therapeutic applications. Here, we describe a novel strategy to concomitantly repress multiple genes using zinc finger proteins fused to Kruppel-Associated Box repression domains (ZF-Rs). This was achieved via the optimization of a lentiviral system tailored for the delivery of ZF-Rs in hematopoietic cells. We showed that an optimal design of the lentiviral backbone is crucial to multiplex up to three ZF-Rs or two ZF-Rs and a chimeric antigen receptor. ZF-R expression had no impact on the integrity and functionality of transduced cells. Furthermore, gene repression in ZF-R-expressing T cells was highly efficient in vitro and in vivo during the entire monitoring period (up to ten weeks), and it was accompanied by epigenetic remodeling events. Finally, we described an approach to improve ZF-R specificity to illustrate the path towards the generation of ZF-Rs with a safe clinical profile. In conclusion, we successfully developed an epigenetic-based cell engineering approach for concomitant modulation of multiple gene expressions that bypass the risks associated with DNA editing.

bioengineering↗

Identification and structural characterization of small-molecule inhibitors of PINK1

Mutations in PTEN-induced putative kinase 1 (PINK1) cause early-onset autosomal recessive Parkinsons Disease (PD). PINK1 is a Ser/Thr protein kinase which functions as a mitochondrial damage sensor and initiates mitochondrial quality control by accumulating on the damaged organelle. There, it will autophosphorylate and then phosphorylate ubiquitin chains, which in turn will recruit and activate Parkin, and E3 ubiquitin ligase also implicated in PD. Ubiquitylation of mitochondrial proteins leads to the autophagic degradation of the damaged organelle. Pharmacological modulation of PINK1 constitutes an appealing avenue to study its physiological function and develop PD therapeutics. In this study, we used a thermal shift assay to identify small-molecule inhibitors of PINK1. In vitro kinase activity assays demonstrate that these molecules are ATP competitive inhibitors that block ubiquitin phosphorylation. PRT062607 (a SYK inhibitor) is the most potent inhibitor of PINK1 in our screen and has an IC50 of 2 M against insect PINK1 and 1 M in HeLa cells expressing human PINK1. The crystal structures of PINK1 in complex with PRT062607 or CYC116 reveal how the compounds interact with the ATP-binding pocket. PRT062607 notably engages with the catalytic aspartate (type-1 inhibition) and causes a destabilization of insert-2 at the autophosphorylation dimer interface. Our findings provide a scaffold for the development of more selective and potent inhibitors of PINK1 that can be used as chemical probes.

biochemistry↗

Deep autoencoder-based behavioral pattern recognition outperforms standard statistical methods in high-dimensional zebrafish studies

Zebrafish have become an essential tool in screening for developmental neurotoxic chemicals and their molecular targets. The success of zebrafish as a screening model is partially due to their physical characteristics including their relatively simple nervous system, rapid development, experimental tractability, and genetic diversity combined with technical advantages that allow for the generation of large amounts of high-dimensional behavioral data. These data are complex and require advanced machine learning and statistical techniques to comprehensively analyze and capture spatiotemporal responses. To accomplish this goal, we have trained semi-supervised deep autoencoders using behavior data from unexposed larval zebrafish to extract quintessential "normal" behavior. Following training, our network was evaluated using data from larvae shown to have significant changes in behavior (using a traditional statistical framework) following exposure to toxicants that include nanomaterials, aromatics, per- and polyfluoroalkyl substances (PFAS), and other environmental contaminants. Further, our model identified new chemicals (Perfluoro-n-octadecanoic acid, 8-Chloroperfluorooctylphosphonic acid, and Nonafluoropentanamide) as capable of inducing abnormal behavior at multiple chemical-concentrations pairs not captured using distance moved alone. Leveraging this deep learning model will allow for better characterization of the different exposure-induced behavioral phenotypes, facilitate improved genetic and neurobehavioral analysis in mechanistic determination studies and provide a robust framework for analyzing complex behaviors found in higher-order model systems. Author SummaryWe demonstrate that a deep autoencoder using raw behavioral tracking data from zebrafish toxicity screens outperforms conventional statistical methods, resulting in a comprehensive evaluation of behavioral data. Our models can accurately distinguish between normal and abnormal behavior with near-complete overlap with existing statistical approaches, with many chemicals detectable at lower concentrations than with conventional statistical tests; this is a crucial finding for the protection of public health. Our deep learning models enable the identification of new substances capable of inducing aberrant behavior, and we generated new data to demonstrate the reproducibility of these results. Thus, neurodevelopmentally active chemicals identified by our deep autoencoder models may represent previously undetectable signals of subtle individual response differences. Our method elegantly accounts for the high degree of behavioral variability associated with the genetic diversity found in a highly outbred population, as is typical for zebrafish research, thereby making it applicable to multiple laboratories. Utilizing the vast quantities of control data generated during high-throughput screening is one of the most innovative aspects of this study and to our knowledge is the first study to explicitly develop a deep autoencoder model for anomaly detection in large-scale toxicological behavior studies.

pharmacology and toxicology↗

Single-nucleus resolution mapping of the adult C. elegans and its application to elucidate inter- and trans-generational response to alcohol.

Single-cell RNA transcriptomic platforms have significantly contributed to our understanding of tissue heterogeneity as well as of developmental and cellular differentiation trajectories. They also provide an opportunity to map an organisms response to environmental cues with high resolution and unbiasedly identify the cell types that are the most transcriptionally sensitive to exposures. Here, we applied single nucleus RNA-seq experimental and computational approaches to C. elegans to establish the transcriptome of the adult nematode and comprehensively characterize the transcriptional impact of ethanol as a model environmental exposure on the entire organism at cell type-resolution over several generations. Clustering, tissue and phenotype enrichment, and gene ontology analyses identified 31 clusters representing a diverse number of adult cell types, including those from syncytial and multi-nucleated tissues which are difficult to assess by single cell RNA-seq, such as the mitotic and meiotic germline, hypodermal cells, and the intestine. We applied this method to identify the impact of inter- and trans-generational exposure to two human-relevant doses of alcohol. Cell type proportions were not significantly altered by ethanol. However, Euclidean distance analysis identified several germline, striated muscle, and neuronal clusters as being major transcriptional targets of ethanol at both the F1 and F3 generations although the relative order of clusters changed between generations. The impact on germline clusters was further confirmed by phenotypic enrichment analysis as well as functional validation, namely a remarkable inter- and trans-generational increase in germline apoptosis, aneuploidy, and embryonic lethality. Together, snRNA-seq of the adult C. elegans represents a powerful approach for the detailed examination of an adult organisms response to environmental cues.

genetics↗

Leveraging High-Throughput Screening Data and Conditional Generative Adversarial Networks to Advance Predictive Toxicology

There are currently 85,000 chemicals registered with the Environmental Protection Agency (EPA) under the Toxic Substances Control Act, but only a small fraction have measured toxicological data. To address this gap, high-throughput screening (HTS) methods are vital. As part of one such HTS effort, embryonic zebrafish were used to examine a suite of morphological and mortality endpoints at six concentrations from over 1,000 unique chemicals found in the ToxCast library (phase 1 and 2). We hypothesized that by using a conditional Generative Adversarial Network (cGAN) and leveraging this large set of toxicity data, plus chemical structure information, we could efficiently predict toxic outcomes of untested chemicals. CAS numbers for each chemical were used to generate textual files containing three-dimensional structural information for each chemical. Utilizing a novel method in this space, we converted the 3D structural information into a weighted set of points while retaining all information about the structure. In vivo toxicity and chemical data were used to train two neural network generators. The first used regression (Go-ZT) while the second utilized cGAN architecture (GAN-ZT) to train a generator to produce toxicity data. Our results showed that both Go-ZT and GAN-ZT models produce similar results, but the cGAN achieved a higher sensitivity (SE) value of 85.7% vs 71.4%. Conversely, Go-ZT attained higher specificity (SP), positive predictive value (PPV), and Kappa results of 67.3%, 23.4%, and 0.21 compared to 24.5%, 14.0%, and 0.03 for the cGAN, respectively. By combining both Go-ZT and GAN-ZT, our consensus model improved the SP, PPV, and Kappa, to 75.5%, 25.0%, and 0.211, respectively, resulting in an area under the receiver operating characteristic (AUROC) of 0.663. Considering their potential use as prescreening tools, these models could provide in vivo toxicity predictions and insight into untested areas of the chemical space to prioritize compounds for HT testing. SummaryA conditional Generative Adversarial Network (cGAN) can leverage a large chemical set of experimental toxicity data plus chemical structure information to predict the toxicity of untested compounds.

bioinformatics↗

Nitrate and nitrite exposure increases anxiety-like behavior and alters brain metabolomic profile in zebrafish

IntroductionDietary nitrate lowers blood pressure and improves athletic performance in humans, yet data supporting observations that it may increase cerebral blood flow and improve cognitive performance are mixed. Here we tested the hypothesis that nitrate and nitrite treatment would improve indicators of learning and cognitive performance in a zebrafish (Danio rerio) model. We also explored the extent to which nitrate and nitrite treatment affected the brain metabolome in order to understand how nitrate and nitrite supplementation may affect indices of cognitive function. MethodsFish were exposed to sodium nitrate (606.9 mg/L), sodium nitrite (19.5 mg/L), or control water for 2-4 weeks and free swim, startle response, innate predator avoidance, social cohesion, and shuttle box assays were performed. ResultsNitrate and nitrite treatment did not change fish weight, length, predator avoidance, or distance and velocity traveled in an unstressed environment. Nitrate- and nitrite-treated fish initially experienced more negative reinforcement and increased time to decision in the shuttle box assay, which is consistent with a decrease in associative learning or executive function however, over multiple trials, all treatment groups demonstrated behaviors associated with learning. Nitrate and nitrite treatment significantly increased anxiety-like behavior but did not alter epinephrine, norepinephrine or dopamine levels. Targeted LC-MS/MS analysis revealed no significant increase in brain nitrate or nitrite concentrations with treatment. An untargeted metabolomics analysis found 47 metabolites whose abundance was significantly altered in the brain with nitrate and nitrite treatment including an 18-19% reduction in the neurotransmitter {gamma}-aminobutyric acid (GABA), and 17-22% reduction in its precursor, glutamine, which may contribute to the increased anxiety-like behavior. ConclusionNitrate and nitrite treatment did not adversely affect multiple parameters of zebrafish health but was associated with mild anxiety-like behavior, changes in the brain metabolome, and caused a short-term decrease in executive function or associative learning.

animal behavior and cognition↗