ArticleNeuron2025
Role of clonal inflammatory microglia in histiocytosis-associated neurodegeneration.
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Article in Neuron, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers, 1 of them a synthesis that pooled it.
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12citing papers in PubMed, 1 pooled it
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Who cites it
12 citing papers in PubMed, 1 synthesis or guideline pooled it.
- Impulsivity, Compulsivity, Social Inappropriateness, and Other Neuropsychiatric Features in Cerebellar Histiocytosis.Cerebellum (London, England) · 2026Pooled it
- Article
- Somatic mosaicism in the brain: linking development, ageing and neurodegeneration.Nature reviews. Neurology · 2026Review
- Histiocytosis development and clinical variation through the lens of genomics.The Journal of pathology · 2026Review
- Article
- Article
- Efficacy of MAPK inhibitors in children with neurodegenerative Langerhans cell histiocytosis: Results from the European Consortium for Histiocytosis.HemaSphere · 2026Article
- Long-term MAPK inhibition of childhood refractory-Langerhans cell histiocytosis: an observational study of 288 patients.Blood advances · 2026Observational
- Progress over perfection: evolving treatments in LCH.Blood advances · 2026Article
- Characterization of the mammalian prodefinitive angio-hematopoietic lineage.Science immunology · 2025Article
- Histiocyte Society blueprint for Langerhans cell histiocytosis research: from cell-of-origin to a more comprehensive cure.Haematologica · 2025Review
- Effective vemurafenib monotherapy for refractory Langerhans cell histiocytosis with sustained results post-withdrawal for over two years: a case report.Frontiers in oncology · 2025Article
Corrections and comments
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Authors and funding
37 authors.
Rocio VicarioImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Stamatina FragkogianniImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Maria PokrovskiiImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Carina MeyerImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Estibaliz Lopez-RodrigoImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Yang HuDepartment of Physiology and Biophysics, Institute for Computational Biomedicine, Weill Cornell, New York, NY 10021, USA.
Masato OgishiSt. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY 10065, USA.
Araitz AlberdiImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Ann BaakoImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Oyku AyImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA.
Isabelle PluDepartment of Neuropathology, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Véronique SazdovitchDepartment of Neuropathology, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Sebastien HeritierFrench Langerhans cell histiocytosis registry, Department of Pediatric Hematology and Oncology, Trousseau Hospital, AP-HP, Paris, France.
Fleur Cohen-AubartDepartment of Internal Medicine & Institut E3M, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Natalia ShorDepartment of Neuroradiology, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Makoto MiyaraCenter for Immunology and Infectious Diseases (CIMI-PARIS), Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Florence Nguyen-KhacDepartment of Hematology, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Agnes VialeMarie-Josée & Henry R. Kravis Center for Molecular Oncology, MSKCC, New York, NY 10065, USA.
Ahmed IdbaihSorbonne Université, Inserm, CNRS, UMR S 1127, Institut du Cerveau et de la Moelle épinière, ICM, AP-HP, Hôpitaux Universitaires La Pitié-Salpêtrière - Charles Foix, Service de Neurologie 2-Mazarin, 75013 Paris, France.
Zahir AmouraDepartment of Neuroradiology, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Marc K RosenblumDepartment of Pathology, MSKCC, New York, NY 10065, USA.
Haochen ZhangHuman Oncology and Pathogenesis Program, MSKCC, New York, NY, USA.
Elias-Ramzey KarnoubHuman Oncology and Pathogenesis Program, MSKCC, New York, NY, USA.
Palash SashittalDepartment of Computer Science, Princeton University, Princeton, NJ, USA.
Akhil JakatdarDepartment of Computer Science, Princeton University, Princeton, NJ, USA.
Christine A Iacobuzio-DonahueDepartment of Pathology, MSKCC, New York, NY 10065, USA; Human Oncology and Pathogenesis Program, MSKCC, New York, NY, USA.
Omar Abdel-WahabHuman Oncology and Pathogenesis Program, MSKCC, New York, NY, USA.
Viviane TabarDepartment of Neurosurgery, and Center for Stem Cell Biology, MSKCC, New York, NY, USA; Department of Neurology, MSKCC, New York, NY 10065, USA.
Nicholas D SocciMarie-Josée & Henry R. Kravis Center for Molecular Oncology, MSKCC, New York, NY 10065, USA.
Olivier ElementoDepartment of Physiology and Biophysics, Institute for Computational Biomedicine, Weill Cornell, New York, NY 10021, USA.
Eli L DiamondDepartment of Neurosurgery, and Center for Stem Cell Biology, MSKCC, New York, NY, USA.
Bertrand BoissonSt. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY 10065, USA.
Jean-Laurent CasanovaSt. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, The Rockefeller University, New York, NY 10065, USA.
Danielle SeilheanDepartment of Neuropathology, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France.
Julien HarocheDepartment of Internal Medicine & Institut E3M, Pitié-Salpêtrière Hospital, APHP-Sorbonne Université, Paris, France. Electronic address: julien.haroche@aphp.fr.
Jean DonadieuFrench Langerhans cell histiocytosis registry, Department of Pediatric Hematology and Oncology, Trousseau Hospital, AP-HP, Paris, France.
Frederic GeissmannImmunology Program, Sloan Kettering Institute, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA. Electronic address: geissmaf@mskcc.org.
Funding
X-RAY CRYSTALLOGRAPHYP30CA008748 · NCI · SLOAN-KETTERING INSTITUTE FOR CANCER RES · PI SELWYN M VICKERS · 1985 to 2026
$347.4MMacrophage development and specification in mice.R01AI130345 · NIAID · SLOAN-KETTERING INST CAN RESEARCH · PI Frederic Geissmann · 2017 to 2026
$5.7MRole of Microglia Somatic Variants in Neurodegenerative DiseasesR01NS115715 · NINDS · SLOAN-KETTERING INST CAN RESEARCH · PI GEISSMANN, FREDERIC · 2019 to 2023
$5.0MLineages and pathophysiology of clonal histiocytic disordersR01HL138090 · NHLBI · SLOAN-KETTERING INST CAN RESEARCH · PI GEISSMANN, FREDERIC · 2017 to 2020
$2.9MNCI NIH HHS P30 CA008748NHLBI NIH HHS R01 HL138090NIAID NIH HHS R01 AI130345NINDS NIH HHS R01 NS115715
Abstract
Langerhans cell histiocytosis (LCH) and Erdheim-Chester disease (ECD) are clonal myeloid disorders associated with mitogen-activated protein (MAP)-kinase-activating mutations and an increased risk of neurodegeneration. We found microglial mutant clones in LCH and ECD patients, whether or not they presented with clinical symptoms of neurodegeneration, associated with microgliosis, astrocytosis, and neuronal loss, predominantly in the rhombencephalon gray nuclei. Neurological symptoms were associated with PU.1
Indexed as
Histiocytosis, Langerhans-CellMicrogliaNeurodegenerative DiseasesAdultAnimalsDisease Models, AnimalFemaleHumansMaleMiceMiddle AgedReceptors, Granulocyte-Macrophage Colony-Stimulating FactorReceptors, Granulocyte-Macrophage Colony-Stimulating FactorbrainstemhippocampusMAPK pathwaymicrogliaNeurodegenerationresident macrophages
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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.