ArticleCancer discovery2019
Epigenomics and Single-Cell Sequencing Define a Developmental Hierarchy in Langerhans Cell Histiocytosis.
Article in Cancer discovery, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 41 papers.
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Who cites it
41 citing papers in PubMed, 63 citations in OpenAlex.
- Langerhans cell histiocytosis: molecular mechanisms underlying pathogenesis and emerging targeted therapeutics.Cancer metastasis reviews · 2026Review
- Histiocytosis development and clinical variation through the lens of genomics.The Journal of pathology · 2026Review
- Article
- Pathogenic myeloid phenotypes drive disease pathology in a novel human neurohistiocytosis model.Blood · 2026Article
- Pre-diagnostic metabolites in peripheral blood of Langerhans cell histiocytosis children with posterior pituitary involvement.Metabolomics : Official journal of the Metabolomic Society · 2026Article
- A case of mixed histiocytosis (Erdheim-Chester disease and Langerhans cell histiocytosis) with STEAP3-associated anemia and type 4 hemochromatosis.Orphanet journal of rare diseases · 2026Article
- Histiocyte Society blueprint for Langerhans cell histiocytosis research: from cell-of-origin to a more comprehensive cure.Haematologica · 2025Review
- Langerhans cells infiltration in lymph nodes of patients with systemic lupus erythematosus.Lupus science & medicine · 2025Article
- BRAFV600E induces key features of LCH in iPSCs with cell type-specific phenotypes and drug responses.Blood · 2025Article
- Research progress and the prospect of using single-cell sequencing technology to explore the characteristics of the tumor microenvironment.Genes & diseases · 2025Review
- Article
- Classification of pediatric soft and bone sarcomas using DNA methylation-based profiling.BMC cancer · 2024Article
- Review
- Single-cell RNA sequencing reveals roles of unique retinal microglia types in early diabetic retinopathy.Diabetology & metabolic syndrome · 2024Article
- Myeloid cells from Langerhans cell histiocytosis patients exhibit increased vesicle trafficking and an altered secretome capable of activating NK cells.Haematologica · 2023Article
- Clinicogenomic associations in childhood Langerhans cell histiocytosis: an international cohort study.Blood advances · 2023Article
- Langerhans cell histiocytosis: current advances in molecular pathogenesis.Frontiers in immunology · 2023Review
- Detection of Immune Microenvironment Changes and Immune-Related Regulators in Langerhans Cell Histiocytosis Bone Metastasis.BioMed research international · 2023Article
- Notch-dependent cooperativity between myeloid lineages promotes Langerhans cell histiocytosis pathology.Science immunology · 2022Article
- Signaling pathways, microenvironment, and targeted treatments in Langerhans cell histiocytosis.Cell communication and signaling : CCS · 2022Review
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17 authors at 5 institutions in 2 countries.
Funding
Abstract
Langerhans cell histiocytosis (LCH) is a rare neoplasm predominantly affecting children. It occupies a hybrid position between cancers and inflammatory diseases, which makes it an attractive model for studying cancer development. To explore the molecular mechanisms underlying the pathophysiology of LCH and its characteristic clinical heterogeneity, we investigated the transcriptomic and epigenomic diversity in primary LCH lesions. Using single-cell RNA sequencing, we identified multiple recurrent types of LCH cells within these biopsies, including putative LCH progenitor cells and several subsets of differentiated LCH cells. We confirmed the presence of proliferative LCH cells in all analyzed biopsies using IHC, and we defined an epigenomic and gene-regulatory basis of the different LCH-cell subsets by chromatin-accessibility profiling. In summary, our single-cell analysis of LCH uncovered an unexpected degree of cellular, transcriptomic, and epigenomic heterogeneity among LCH cells, indicative of complex developmental hierarchies in LCH lesions. SIGNIFICANCE: This study sketches a molecular portrait of LCH lesions by combining single-cell transcriptomics with epigenome profiling. We uncovered extensive cellular heterogeneity, explained in part by an intrinsic developmental hierarchy of LCH cells. Our findings provide new insights and hypotheses for advancing LCH research and a starting point for personalizing therapy.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.