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Biology subjects

van Beuningen, S. F. B.

Publications and source records attributed to van Beuningen, S. F. B..

7 recordsLinked to original sources

NuclearIDTracker resolves intestinal cell identity and lineage dynamics through nuclear phenotypic signatures

Organoid models have transformed our understanding of intestinal renewal. Fluorescent imaging has been extensively used to identify key cell types and their differentiation pathways, but immunofluorescence provides only static readouts, whereas live imaging requires fluorescent-reporter engineering and is constrained by limited multiplexing and spectral overlap. Here, we introduce NuclearIDTracker, an explainable machine-learning framework that infers cell identity directly from 3D nuclear segmentations. Using a single nuclear marker, NuclearIDTracker accurately classifies intestinal cell types and integrates with single-cell tracking to resolve lineages and reconstruct dynamic state transitions during organoid development. We show that TA-like cells, rather than stem cells, drive early crypt formation and generate enterocyte and Paneth lineages, as well as the stem-cell population, which emerges only later and subsequently replenishes the TA-like compartment. Following stem-cell ablation, crypt regeneration was not driven by a single discrete cell type. Instead, multiple epithelial populations converged on a proliferative regenerative state with a nuclear phenotypic signature that resembled, but remained distinct from, that of homeostatic TA-like cells, and a YAP/TAZ-associated fetal-like transcriptional signature. Thus, nuclear phenotypic signatures resolve cell identity and reveal coordinated epithelial plasticity during crypt regeneration. NuclearIDTracker establishes a non-perturbative tool to quantify cell identity and state dynamics at single-cell resolution, revealing previously inaccessible biological dynamics and expanding the toolkit for studying epithelial homeostasis, regeneration, and disease.

cell biology↗

UDIST: unsupervised disentanglement of shape and texture for multi-scale phenotypic profiling in 2D microscopy

Microscopy-based phenotypic profiling relies increasingly on autonomous, unsupervised feature extraction, yet no existing method explicitly separates shape from texture into dedicated and independent latent subspaces by architectural design. Therefore texture, encoding critical biological information such as protein distribution and intracellular organisation, remains inaccessible as an independent feature domain in standard unsupervised approaches. This represents a fundamental limitation that prevents unbiased phenotypic analysis across biological scales. Here we introduce UDIST (Unsupervised Disentanglement of Shape and Texture), a sequential dual variational autoencoder (VAE) framework that tackles this fundamental limitation by explicitly decoupling shape from texture into independent, non-overlapping latent subspaces at the single-object level. By training two VICReg-regularised VAEs on principal-axis-aligned objects, UDIST separates binary shape from continuous texture information into rotation-invariant feature spaces, enabling separate downstream analysis of both domains. We validated UDIST across biological scales, from nuclei and single cells to patient-derived intestinal organoids, using both fluorescence and brightfield imaging, revealing phenotypic differences previously hidden by morphological variation and enabling the independent analysis of shape and texture in downstream analyses including clustering and similarity measurements. UDIST provides a versatile, label-free, and unsupervised tool for multi-scale phenotypic profiling in high-content microscopy and screening.

bioinformatics↗

LiFT: Live foci tracking for quantitative analysis of DNA damage dynamics

Quantitative analysis of radiation induced DNA double strand breaks (DSBs) and their repair is essential for understanding and eventually contributing to improving radiation-based cancer therapies. Using live-cell microscopy, the formation and resolution of DSBs over time can be followed in individual cells through tracking of foci formed by accumulation of DSB repair proteins. However, manual analysis of such time-lapse datasets is a tedious time-consuming task that is prone to operator bias, affecting the reproducibility. Here, we present LiFT, an automated image analysis pipeline, specifically designed for robust quantification of DSB kinetics in live-cell imaging experiments. To quantify DSB kinetics, our pipeline first segments and tracks cell nuclei without requiring a nuclear stain. After correcting for inter-frame motion through image registration, automatic detection and tracking of foci within these nuclei enables direct quantification of the dynamics of individual repair events. Multiple algorithmic options were implemented for each step of the pipeline, ensuring more general applicability to potentially different imaging setups and applications. We evaluated the pipeline using PLC/PRF/5 cells and demonstrated its generalizability on U2OS-SSTR2 cells. Our results show that LiFT enables reproducible and scalable quantification of DSB dynamics, providing a broadly applicable framework to analyse live-cell imaging data in cancer research. To improve the adoption of LiFT, we made it available as an open-source Python package and provided a graphical user interface to select different methods and adjust method related parameters.

bioinformatics↗

Personalized multi-assay profiling of respiratory motile ciliopathies and mRNA therapy

IntroductionImpaired motile cilia function contributes to many respiratory disorders, but therapies targeting this cellular defect are currently lacking. Personalized airway epithelial models combined with quantitative, complementary ciliary assays can pave the way for the development of such therapies. However, existing airway epithelial cultures often show variable ciliogenesis, and ciliary function is frequently assessed using a single assay that does not capture the phenotypic heterogeneity of ciliary dysfunction. Here, we established a personalized, multi-assay in vitro platform using human nasal epithelial cells (HNECs) to assess ciliary function and therapeutic response, using primary ciliary dyskinesia (PCD) as a model disease. MethodsHNECs from 8 healthy individuals and 13 individuals with PCD carrying distinct disease-associated variants were obtained by nasal brushing. Cells were differentiated under optimized conditions, including {gamma}-secretase/Notch and BMP pathway inhibitors and a low liquid-liquid interface, to generate highly ciliated 2D epithelial cultures. Ciliary function was assessed using ciliary beat frequency, bead transport, and apical-out nasal organoid rotation assays. Therapeutic rescue was assessed in HNECs harboring DNAI1 alterations using DNAI1 mRNA-loaded lipid nanoparticles. ResultsOptimized differentiation yielded reproducibly multiciliated HNEC cultures. The multi-assay platform distinguished healthy from PCD-derived HNECs and revealed individual- and genotype-specific patterns of ciliary dysfunction not captured by a single assay. Basolateral administration of DNAI1 mRNA-loaded lipid nanoparticles resulted in partial, dose-dependent recovery of ciliary function in DNAI1-deficient HNECs. ConclusionThis study establishes a standardized, individual-specific multi-assay nasal epithelial platform for functional phenotyping of motile cilia and preclinical evaluation of emerging therapies, with demonstrated utility in PCD.

cell biology↗

Immortalization of human nasal and bronchial airway epithelial cells for genome editing applications

In vitro air-liquid interface culture of airway epithelial cells is used as a model system to study respiratory diseases. This culture system not only overcomes the need for animal models or continuous biopsies from individuals but also enables studies of pathophysiology associated with the disease in a patient background. Human airway basal cells serve as progenitor cells for a functional pseudostratified airway epithelium composed mainly of multiciliated and secretory cells. However, due to the limited ability of basal cells to proliferate and differentiate, the long-term use of primary material in culture is restricted. This challenges research that requires genome editing. Here, we describe airway stem cells from nasal and bronchial origin immortalized by hTERT overexpression followed by polyclonal expansion. We demonstrate that this diverse panel of cell lines shows differentiation patterns similar to primary stem cells and can be used for lentiviral and CRISPR/Cas9 genome editing. These cell lines and optimized protocols facilitate airway biology research and disease phenotyping.

cell biology↗

Small molecule-directed differentiation of submerged-cultured human nasal airway epithelia for respiratory disease modelling

Submerged cultures of undifferentiated or transformed epithelial cells are widely used in respiratory research due to their ease of use and scalability. However, these systems fail to capture the cellular diversity of the human airway epithelium. In this study, we developed an in vitro model where cryopreserved human nasal epithelial cells, collected by brushings, are differentiated under submerged conditions on standard plastic cultureware. By applying small-molecule inhibitors targeting Notch and BMP signaling, we achieved efficient differentiation of cultures containing basal, secretory, and ciliated cells. This approach supports scalable culturing of both 2D epithelial monolayers and 3D organoids, validated as (personalized) disease models for primary ciliary dyskinesia, cystic fibrosis, and respiratory syncytial virus infection. This model offers a cost-effective, scalable platform that combines the simplicity of traditional cultures with the cellular complexity of the human airway epithelium, providing a valuable tool for respiratory disease research.

cell biology↗

Drug repurposing for Cystic Fibrosis: identification of drugs that induce CFTR-independent fluid secretion in nasal organoids

Individuals with Cystic Fibrosis (CF) suffer from severe respiratory disease due to a genetic defect in the Cystic Fibrosis Transmembrane conductance Regulator (CFTR) gene, which impairs airway epithelial ion and fluid secretion. New CFTR modulators that restore mutant CFTR function have been recently approved for a large group of people with CF (pwCF), but [~]19% of pwCF cannot benefit from CFTR modulators [1]. Restoration of epithelial fluid secretion through non-CFTR pathways might be an effective treatment for all pwCF. Here we developed a medium-throughput 384-wells screening assay using nasal CF airway epithelial organoids, with the aim to repurpose FDA-approved drugs as modulators of non-CFTR dependent epithelial fluid secretion. From a [~]1400 FDA-approved drug library, we identified and validated 12 FDA-approved drugs that induced CFTR-independent fluid secretion. Among the hits were several cAMP-mediating drugs, including {beta}2-adrenergic agonists. The hits displayed no effects on chloride conductance measured in Ussing chamber, and fluid secretion was not affected by TMEM16A as demonstrated by knockout (KO) experiments in primary nasal epithelial cells. Altogether, our results demonstrate the use of primary nasal airway cells for mediumscale drug screening, target validation with a highly efficient protocol for generating CRISPR-Cas9 KO cells and identification of compounds which induce fluid secretion in a CFTR- and TMEM16A-indepent manner.

cell biology↗