FLT3L governs the development of partially overlapping hematopoietic lineages in humans and mice.

B cells FLT3 FLT3L FLT3LG NK cells dendritic cells hematopoiesis human papillomavirus primary immunodeficiency

Journal

Cell
ISSN: 1097-4172
Titre abrégé: Cell
Pays: United States
ID NLM: 0413066

Informations de publication

Date de publication:
02 May 2024
Historique:
received: 26 07 2023
revised: 04 03 2024
accepted: 10 04 2024
medline: 4 5 2024
pubmed: 4 5 2024
entrez: 3 5 2024
Statut: aheadofprint

Résumé

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.

Identifiants

DOI: 10.1016/j.cell.2024.04.009 PMID: 38701783
pubmed: 38701783
pii: S0092-8674(24)00404-5
doi: 10.1016/j.cell.2024.04.009
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

Copyright © 2024 The Authors. Published by Elsevier Inc. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of interests J.-L.C. serves on the scientific advisory boards of ADMA Biologics Inc., Kymera Therapeutics, and Elixiron Immunotherapeutics.

Auteurs

Mana 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.

Romain 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, France.

Steicy 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.

Jingwei 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.

Chantal Lagresle-Peyrou (C)

Paris Cité University, Imagine Institute, Paris, France; Biotherapy Clinical Investigation Center, Groupe Hospitalier Universitaire Ouest, AP-HP, INSERM, Paris, France.

Hossein 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.

Antoine 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.

Corentin 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.

Antoine Guérin (A)

Garvan Institute of Medical Research, Darlinghurst, NSW, Australia; St. Vincent's Clinical School, Faculty of Medicine, University of NSW, Sydney, NSW, Australia.

Erika Della Mina (ED)

Garvan Institute of Medical Research, Darlinghurst, NSW, Australia; St. Vincent's Clinical School, Faculty of Medicine, University of NSW, Sydney, NSW, Australia.

Debra 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.

Ottavia M Delmonte (OM)

Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.

Ahmad Yatim (A)

St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.

Kevin Mulder (K)

Gustave Roussy Cancer Campus, Villejuif, France; Paris-Saclay University, Ile-de-France, France.

Mathieu Mancini (M)

Dahdaleh Institute of Genomic Medicine, McGill University, Montreal, QC, Canada; Department of Microbiology and Immunology, McGill University, Montreal, QC, Canada.

Darawan Rinchai (D)

St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.

Adeline Denis (A)

Laboratory of Human Lymphohematopoiesis, INSERM, Imagine Institute, Paris, France.

Anna-Lena 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.

Karla 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.

Sarah 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 University College of Medicine, Hershey, PA, USA.

Hassan Rokni-Zadeh (H)

Department of Medical Biotechnology, School of Medicine, Zanjan University of Medical Sciences (ZUMS), Zanjan, Iran.

Majid Changi-Ashtiani (M)

School of Mathematics, Institute for Research in Fundamental Sciences (IPM), Tehran, Iran.

Yoann 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.

Caroline 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.

Boris Bessot (B)

Paris Cité University, Imagine Institute, Paris, France; Biotherapy Clinical Investigation Center, Groupe Hospitalier Universitaire Ouest, AP-HP, INSERM, Paris, France.

Cassandre Cremades (C)

Laboratory of Human Genetics of Infectious Diseases, Necker Branch, INSERM UMR 1163, Necker Hospital for Sick Children, Paris, France.

Marie 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.

Axel Cederholm (A)

Science for Life Laboratory, Department of Medical Biochemistry and Microbiology, Uppsala University, Uppsala, Sweden.

Masato Ogishi (M)

St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.

Quentin 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.

Omer Beganovic (O)

Laboratoire d'Onco-hématologie, Necker Hospital for Sick Children, AP-HP, Paris, France.

Mania 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.

Margareta Wuyts (M)

Department of Microbiology and Immunology, Clinical and Diagnostic Immunology, KU Leuven, Leuven, Belgium.

Taushif Khan (T)

Research Branch, Sidra Medicine, Doha, Qatar.

Sébastien Fouéré (S)

Groupe Hospitalier Saint-Louis, Lariboisière, Fernand-Widal, CeGIDD, AP-HP, Paris, France.

Florian Herms (F)

Dermatology Department, Paris-Cité University, INSERM 976, Saint Louis Hospital, Paris, France.

Johan Chanal (J)

Dermatology Department, Cochin Hospital, INSERM U1016, AP-HP, Paris, France.

Boaz Palterer (B)

Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.

Julie Bruneau (J)

Department of Pathology, Necker Hospital for Sick Children, AP-HP, Paris-Cité University, Paris, France.

Thierry J Molina (TJ)

Department of Pathology, Necker Hospital for Sick Children, AP-HP, Paris-Cité University, Paris, France.

Stéphanie Leclerc-Mercier (S)

Department of Pathology, Necker Hospital for Sick Children, AP-HP, Paris-Cité University, Paris, France.

Jean-Luc Prétet (JL)

Papillomavirus National Reference Center, Besançon Hospital, Besançon, France.

Leila Youssefian (L)

Department of Pathology and Laboratory Medicine, UCLA Clinical Genomics Center, David Geffen School of Medicine at UCLA, Los Angeles, CA, USA.

Hassan 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.

Nima Parvaneh (N)

Department of Pediatrics, Children's Medical Center, Tehran University of Medical Sciences, Tehran, Iran.

Kristl G 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.

Rik Schrijvers (R)

Department of Microbiology, Immunology and Transplantation, Allergy and Clinical Immunology Research Group, KU Leuven, Leuven, Belgium.

Marine Luka (M)

Labtech Single-Cell@Imagine, Imagine Institute, INSERM UMR 1163, 75015 Paris, France.

Philippe Pérot (P)

Pathogen Discovery Laboratory, Institut Pasteur, Paris Cité University, Paris, France.

Jacques Fourgeaud (J)

Paris Cité University, URP 7328 FETUS, Paris, France; Microbiology Department, AP-HP, Necker Hospital for Sick Children, Paris, France.

Céline Nourrisson (C)

Clermont Auvergne University, INSERM U1071, M2iSH, USC INRAE 1382, CHU Clermont-Ferrand, 3IHP, Department of Parasitology-Mycology, Clermont-Ferrand, France; National Reference Center for Cryptosporidiosis, Microsporidia and Other Digestive Protozoa, Clermont-Ferrand, France.

Philippe Poirier (P)

Clermont Auvergne University, INSERM U1071, M2iSH, USC INRAE 1382, CHU Clermont-Ferrand, 3IHP, Department of Parasitology-Mycology, Clermont-Ferrand, France; National Reference Center for Cryptosporidiosis, Microsporidia and Other Digestive Protozoa, Clermont-Ferrand, France.

Emmanuelle Jouanguy (E)

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; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.

Stéphanie Boisson-Dupuis (S)

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; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.

Jacinta Bustamante (J)

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; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA; Study Center for Primary Immunodeficiencies, Necker Hospital for Sick Children, AP-HP, Paris, France.

Luigi D Notarangelo (LD)

Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, USA.

Neil Christensen (N)

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.

Nils Landegren (N)

Science for Life Laboratory, Department of Medical Biochemistry and Microbiology, Uppsala University, Uppsala, Sweden; Centre for Molecular Medicine, Department of Medicine, Karolinska Institutet, Stockholm, Sweden.

Laurent Abel (L)

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; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA.

Nico Marr (N)

Research Branch, Sidra Medicine, Doha, Qatar; College of Health and Life Sciences, Hamad Bin Khalifa University, Doha, Qatar.

Emmanuelle Six (E)

Biotherapy Clinical Investigation Center, Groupe Hospitalier Universitaire Ouest, AP-HP, INSERM, Paris, France.

David Langlais (D)

Dahdaleh Institute of Genomic Medicine, McGill University, Montreal, QC, Canada; Department of Microbiology and Immunology, McGill University, Montreal, QC, Canada; Department of Human Genetics, McGill University, Montreal, QC, Canada.

Tim Waterboer (T)

Infections and Cancer Epidemiology, Infection, Inflammation and Cancer Program, German Cancer Research Center (DKFZ), Heidelberg, Germany.

Florent Ginhoux (F)

Gustave Roussy Cancer Campus, Villejuif, France; Paris-Saclay University, Ile-de-France, France.

Cindy S Ma (CS)

Garvan Institute of Medical Research, Darlinghurst, NSW, Australia; St. Vincent's Clinical School, Faculty of Medicine, University of NSW, Sydney, NSW, Australia.

Stuart G Tangye (SG)

Garvan Institute of Medical Research, Darlinghurst, NSW, Australia; St. Vincent's Clinical School, Faculty of Medicine, University of NSW, Sydney, NSW, Australia.

Isabelle Meyts (I)

Laboratory of Inborn Errors of Immunity, Department of Microbiology, Immunology and Transplantation, KU Leuven, Leuven, Belgium; Department of Pediatrics, Leuven University Hospitals, Leuven, Belgium.

Nico Lachmann (N)

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.

Jiafen Hu (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.

Mohammad Shahrooei (M)

Department of Microbiology and Immunology, Clinical and Diagnostic Immunology, KU Leuven, Leuven, Belgium; Specialized Immunology Laboratory of Dr. Shahrooei, Tehran, Iran.

Xavier Bossuyt (X)

Department of Microbiology and Immunology, Clinical and Diagnostic Immunology, KU Leuven, Leuven, Belgium; Department of Laboratory Medicine, University Hospitals Leuven, Leuven, Belgium.

Jean-Laurent Casanova (JL)

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, France; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA; Howard Hughes Medical Institute, New York, NY, USA.

Vivien Béziat (V)

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; St. Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller Branch, Rockefeller University, New York, NY, USA. Electronic address: vivien.beziat@inserm.fr.

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