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FLT3L governs the development of partially overlapping hematopoietic lineages in humans and mice.
Momenilandi, Mana; Lévy, Romain; Sobrino, Steicy; Li, Jingwei; Lagresle-Peyrou, Chantal; Esmaeilzadeh, Hossein; Fayand, Antoine; Le Floc'h, Corentin; Guérin, Antoine; Mina, Erika Della; Shearer, Debra; Delmonte, Ottavia M; Yatim, Ahmad; Mulder, Kevin; Mancini, Mathieu; Rinchai, Darawan; Denis, Adeline; Neehus, Anna-Lena; Balogh, Karla; Brendle, Sarah; Rokni-Zadeh, Hassan; Changi-Ashtiani, Majid; Seeleuthner, Yoann; Deswarte, Caroline; Bessot, Boris; Cremades, Cassandre; Materna, Marie; Cederholm, Axel; Ogishi, Masato; Philippot, Quentin; Beganovic, Omer; Ackermann, Mania; Wuyts, Margareta; Khan, Taushif; Fouéré, Sébastien; Herms, Florian; Chanal, Johan; Palterer, Boaz; Bruneau, Julie; Molina, Thierry J; Leclerc-Mercier, Stéphanie; Prétet, Jean-Luc; Youssefian, Leila; Vahidnezhad, Hassan; Parvaneh, Nima; Claeys, Kristl G; Schrijvers, Rik; Luka, Marine; Pérot, Philippe; Fourgeaud, Jacques.
Affiliation
  • Momenilandi M; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France.
  • Lévy R; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France; Pediatric Hematology-Immunology and Rheumatology Unit, Necker Hospital for Sick Children, AP-HP, Paris, Fran
  • Sobrino S; Laboratory of Chromatin and Gene Regulation During Development, Paris Cité University, UMR1163 INSERM, Imagine Institute, Paris, France; Laboratory of Human Lymphohematopoiesis, INSERM, Imagine Institute, Paris, France.
  • Li J; Jake Gittlen Laboratories for Cancer Research, Pennsylvania State University College of Medicine, Hershey, PA, USA; Department of Pathology and Laboratory Medicine, Pennsylvania State University College of Medicine, Hershey, PA, USA.
  • Lagresle-Peyrou C; Paris Cité University, Imagine Institute, Paris, France; Biotherapy Clinical Investigation Center, Groupe Hospitalier Universitaire Ouest, AP-HP, INSERM, Paris, France.
  • Esmaeilzadeh H; Allergy Research Center, Shiraz University of Medical Sciences, Shiraz, Iran; Department of Allergy and Clinical Immunology, Namazi Hospital, Shiraz University of Medical Sciences, Shiraz, Iran.
  • Fayand A; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France; Sorbonne University, AP-HP, Tenon Hospital, Department of Internal Medicine, Paris, France.
  • Le Floc'h C; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France.
  • Guérin A; Garvan Institute of Medical Research, Darlinghurst, NSW, Australia; St. Vincent's Clinical School, Faculty of Medicine, University of NSW, Sydney, NSW, Australia.
  • Mina ED; Garvan Institute of Medical Research, Darlinghurst, NSW, Australia; St. Vincent's Clinical School, Faculty of Medicine, University of NSW, Sydney, NSW, Australia.
  • Shearer D; Jake Gittlen Laboratories for Cancer Research, Pennsylvania State University College of Medicine, Hershey, PA, USA; Department of Pathology and Laboratory Medicine, Pennsylvania State University College of Medicine, Hershey, PA, USA.
  • Delmonte OM; Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
  • Yatim A; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.
  • Mulder K; Gustave Roussy Cancer Campus, Villejuif, France; Paris-Saclay University, Ile-de-France, France.
  • Mancini M; Dahdaleh Institute of Genomic Medicine, McGill University, Montreal, QC, Canada; Department of Microbiology and Immunology, McGill University, Montreal, QC, Canada.
  • Rinchai D; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.
  • Denis A; Laboratory of Human Lymphohematopoiesis, INSERM, Imagine Institute, Paris, France.
  • Neehus AL; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France.
  • Balogh K; Jake Gittlen Laboratories for Cancer Research, Pennsylvania State University College of Medicine, Hershey, PA, USA; Department of Pathology and Laboratory Medicine, Pennsylvania State University College of Medicine, Hershey, PA, USA.
  • Brendle S; Jake Gittlen Laboratories for Cancer Research, Pennsylvania State University College of Medicine, Hershey, PA, USA; Department of Pathology and Laboratory Medicine, Pennsylvania State University College of Medicine, Hershey, PA, USA; Department of Microbiology and Immunology, Pennsylvania State Univ
  • Rokni-Zadeh H; Department of Medical Biotechnology, School of Medicine, Zanjan University of Medical Sciences (ZUMS), Zanjan, Iran.
  • Changi-Ashtiani M; School of Mathematics, Institute for Research in Fundamental Sciences (IPM), Tehran, Iran.
  • Seeleuthner Y; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France.
  • Deswarte C; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France.
  • Bessot B; Paris Cité University, Imagine Institute, Paris, France; Biotherapy Clinical Investigation Center, Groupe Hospitalier Universitaire Ouest, AP-HP, INSERM, Paris, France.
  • Cremades C; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France.
  • Materna M; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France.
  • Cederholm A; Science for Life Laboratory, Department of Medical Biochemistry and Microbiology, Uppsala University, Uppsala, Sweden.
  • Ogishi M; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.
  • Philippot Q; Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France; Paris Cité University, Imagine Institute, Paris, France.
  • Beganovic O; Laboratoire d'Onco-hématologie, Necker Hospital for Sick Children, AP-HP, Paris, France.
  • Ackermann M; Hannover Medical School, Department of Pediatric Pulmonology, Allergology and Neonatology, Hannover, Germany; Hannover Medical School, Cluster of Excellence RESIST (EXC 2155), Hannover, Germany; Fraunhofer Institute for Toxicology and Experimental Medicine (ITEM), Hannover, Germany.
  • Wuyts M; Department of Microbiology and Immunology, Clinical and Diagnostic Immunology, KU Leuven, Leuven, Belgium.
  • Khan T; Research Branch, Sidra Medicine, Doha, Qatar.
  • Fouéré S; Groupe Hospitalier Saint-Louis, Lariboisière, Fernand-Widal, CeGIDD, AP-HP, Paris, France.
  • Herms F; Dermatology Department, Paris-Cité University, INSERM 976, Saint Louis Hospital, Paris, France.
  • Chanal J; Dermatology Department, Cochin Hospital, INSERM U1016, AP-HP, Paris, France.
  • Palterer B; Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.
  • Bruneau J; Department of Pathology, Necker Hospital for Sick Children, AP-HP, Paris-Cité University, Paris, France.
  • Molina TJ; Department of Pathology, Necker Hospital for Sick Children, AP-HP, Paris-Cité University, Paris, France.
  • Leclerc-Mercier S; Department of Pathology, Necker Hospital for Sick Children, AP-HP, Paris-Cité University, Paris, France.
  • Prétet JL; Papillomavirus National Reference Center, Besançon Hospital, Besançon, France.
  • Youssefian L; Department of Pathology and Laboratory Medicine, UCLA Clinical Genomics Center, David Geffen School of Medicine at UCLA, Los Angeles, CA, USA.
  • Vahidnezhad H; Department of Pediatrics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA; Department of Dermatology, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA; Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA
  • Parvaneh N; Department of Pediatrics, Children's Medical Center, Tehran University of Medical Sciences, Tehran, Iran.
  • Claeys KG; Department of Neurology, University Hospitals Leuven, Leuven, Belgium; Laboratory for Muscle Diseases and Neuropathies, Department of Neurosciences, KU Leuven, and Leuven Brain Institute (LBI), Leuven, Belgium.
  • Schrijvers R; Department of Microbiology, Immunology and Transplantation, Allergy and Clinical Immunology Research Group, KU Leuven, Leuven, Belgium.
  • Luka M; Labtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, 75015 Paris, France.
  • Pérot P; Pathogen Discovery Laboratory, Institut Pasteur, Paris Cité University, Paris, France.
  • Fourgeaud J; Paris Cité University, URP 7328 FETUS, Paris, France; Microbiology Department, AP-HP, Necker Hospital for Sick Children, Paris, France.
Cell ; 187(11): 2817-2837.e31, 2024 May 23.
Article in En | MEDLINE | ID: mdl-38701783
ABSTRACT
FMS-related tyrosine kinase 3 ligand (FLT3L), encoded by FLT3LG, is a hematopoietic factor essential for the development of natural killer (NK) cells, B cells, and dendritic cells (DCs) in mice. We describe three humans homozygous for a loss-of-function FLT3LG variant with a history of various recurrent infections, including severe cutaneous warts. The patients' bone marrow (BM) was hypoplastic, with low levels of hematopoietic progenitors, particularly myeloid and B cell precursors. Counts of B cells, monocytes, and DCs were low in the patients' blood, whereas the other blood subsets, including NK cells, were affected only moderately, if at all. The patients had normal counts of Langerhans cells (LCs) and dermal macrophages in the skin but lacked dermal DCs. Thus, FLT3L is required for B cell and DC development in mice and humans. However, unlike its murine counterpart, human FLT3L is required for the development of monocytes but not NK cells.
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Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Killer Cells, Natural / Membrane Proteins Limits: Animals / Female / Humans / Male Language: En Journal: Cell Year: 2024 Document type: Article Affiliation country:

Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Killer Cells, Natural / Membrane Proteins Limits: Animals / Female / Humans / Male Language: En Journal: Cell Year: 2024 Document type: Article Affiliation country:
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