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Onfroy, A.

Publications and source records attributed to Onfroy, A..

3 recordsLinked to original sources

A systematic benchmark of bioinformatics methods for single-cell and spatial RNA-seq Nanopore long-read data

Alternative splicing plays a crucial role in transcriptomic complexity, yet remains difficult to resolve at the single-cell level due to the limitations of short-read technologies. Coupling single-cell with long-read sequencing offers full-length transcript coverage, enabling more accurate isoform detection. Multiple specialized computational tools tailored for single-cell and spatial long-read transcriptomics have been developed, with diverse strategies. To compare the effectiveness of these approaches, we generated paired short-read and Nanopore long-read single-cell datasets, tailored for benchmarking bioinformatics tools. We evaluated ten state-of-the-art methods, spanning four analytical dimensions: barcodes and UMI detection, demultiplexing and UMI clustering, gene-level expression profiling, and isoform detection and quantification. Using real and simulated datasets across different protocols, sequencing depths and chemistries, we assessed the accuracy, robustness, and scalability of each tool. Our results revealed method-specific trade-offs, and highlight the importance of sequencing quality and UMI correction strategies. This benchmark provides a practical resource for optimizing isoform analysis and accurate gene expression profiling in single-cell and spatial transcriptomics using long-read sequencing. Our benchmarking workflow is designed to be reusable, thereby enabling method developers to compare their own approaches against the set of reference methods evaluated in this work.

bioinformatics↗

Exploring the mechanisms governing the initiation of plexiform neurofibromas using Prss56Cre,Nf1-KO mouse model at a single-cell resolution

About half of patients with the genetic disease Neurofibromatosis type 1 (NF1) develop benign nerve sheath tumors, called plexiform neurofibromas (pNFs). Despite important advances in understanding the pathogenesis of pNFs, mainly due to the plethora of dedicated mouse models, the mechanisms responsible for the initiation of this process remain poorly understood. Here, we used a Nf1-KO mouse model targeting biallelic loss of Nf1 in boundary cap cells on a wild type and heterozygous Nf1 background to explore the early events driving pNFs development. All mutants develop subcutaneous hyperplastic nerves with some progressing into pNFs from one year of age. We discovered that skin trauma accelerates this process, highlighting the role of inflammation. While skin trauma has no effect on control subcutaneous nerves, in the mutant nerves on a wildtype background, we observed an expansion of mutant Schwann cells expressing a profibrotic program. A similar response was observed in the mutant nerves on a heterozygous background without skin trauma, highlighting the impact of Nf1 heterozygosity in the nerve micro-environment on the development of pNFs. O_FIG O_LINKSMALLFIG WIDTH=200 HEIGHT=64 SRC="FIGDIR/small/662123v1_ufig1.gif" ALT="Figure 1"> View larger version (18K): org.highwire.dtl.DTLVardef@18709d0org.highwire.dtl.DTLVardef@955a59org.highwire.dtl.DTLVardef@15e9bd2org.highwire.dtl.DTLVardef@481d9f_HPS_FORMAT_FIGEXP M_FIG C_FIG

cancer biology↗

Hair follicle stem cell fate supports distinct clinical endotypes in Hidradenitis Suppurativa

Hidradenitis suppurativa (HS) is a severe skin disorder affecting 1% of the global population, with a complex and poorly understood pathogenesis involving aberrant keratinization and autoinflammation. It remains unclear whether autoinflammatory events precede or follow hyperkeratotic changes in hair follicle (HF) epithelia. Using single-cell RNA sequencing, we characterized HF cell populations in HS patients and investigated their role in disease pathogenesis. We uncovered two distinct differentiation trajectories of HF stem cells (HF-SCs): one leading to interfollicular epidermis (IFE) basal cells enriched in inflammatory pathways, and another giving rise to outer root sheath (ORS) cells associated with keratinization. In HS lesions, both populations displayed altered inflammatory phenotypes and were closely linked to immune cell infiltration, pointing to a role in disease heterogeneity. By integrating clinical features with HF cell composition from 49 HS patients, we identified three major endotypes: (i) an inflammatory subtype, marked by T cell infiltration and an expansion of IFE basal cells; (ii) a keratinizing subtype, characterized by ORS enrichment and minimal inflammation; and a mixed subtype, exhibiting features of follicular remodeling, fistula formation, and variable immune involvement. These findings provide novel insights into the epithelial-immune interactions that drive HS and support a stratified therapeutic approach tailored to the specific HF dysfunctions of each patient subgroup. O_FIG O_LINKSMALLFIG WIDTH=200 HEIGHT=49 SRC="FIGDIR/small/656362v1_ufig1.gif" ALT="Figure 1"> View larger version (14K): org.highwire.dtl.DTLVardef@4e8af9org.highwire.dtl.DTLVardef@85f61borg.highwire.dtl.DTLVardef@e085a7org.highwire.dtl.DTLVardef@66d804_HPS_FORMAT_FIGEXP M_FIG C_FIG

immunology↗