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

Mana Momenilandi; Romain Lévy; Steicy Sobrino; Jingwei Li; Chantal Lagresle-Peyrou; Hossein Esmaeilzadeh; Antoine Fayand; Corentin Le Floc'h; Antoine Guérin; Erika Della Mina; Debra Shearer; Ottavia M. Delmonte; Ahmad Yatim; Kevin Mulder; Mathieu Mancini; Darawan Rinchai; Adeline Denis; Anna Lena Neehus; Karla Balogh; Sarah Brendle; Hassan Rokni-Zadeh; Majid Changi-Ashtiani; Yoann Seeleuthner; Caroline Deswarte; Boris Bessot; Cassandre Cremades; Marie Materna; Axel Cederholm; Masato Ogishi; Quentin Philippot; Omer Beganovic; Mania Ackermann; Margareta Wuyts; Taushif Khan; Sébastien Fouéré; Florian Herms; Johan Chanal; Boaz Palterer; Julie Bruneau; Thierry J. Molina; Stéphanie Leclerc-Mercier; Jean Luc Prétet; Leila Youssefian; Hassan Vahidnezhad; Nima Parvaneh; Kristl G. Claeys; Rik Schrijvers; Marine Luka; Philippe Pérot; Jacques Fourgeaud; Céline Nourrisson; Philippe Poirier; Emmanuelle Jouanguy; Stéphanie Boisson-Dupuis; Jacinta Bustamante; Luigi D. Notarangelo; Neil Christensen; Nils Landegren; Laurent Abel; Nico Marr; Emmanuelle Six; David Langlais; Tim Waterboer; Florent Ginhoux; Cindy S. Ma; Stuart G. Tangye; Isabelle Meyts; Nico Lachmann; Jiafen Hu; Mohammad Shahrooei; Xavier Bossuyt; Jean Laurent Casanova; Vivien Béziat

doi:10.1016/j.cell.2024.04.009

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

Mana Momenilandi, Romain Lévy, Steicy Sobrino, Jingwei Li, Chantal Lagresle-Peyrou, Hossein Esmaeilzadeh, Antoine Fayand, Corentin Le Floc'h, Antoine Guérin, Erika Della Mina, Debra Shearer, Ottavia M. Delmonte, Ahmad Yatim, Kevin Mulder, Mathieu Mancini, Darawan Rinchai, Adeline Denis, Anna Lena Neehus, Karla Balogh, Sarah BrendleHassan Rokni-Zadeh, Majid Changi-Ashtiani, Yoann Seeleuthner, Caroline Deswarte, Boris Bessot, Cassandre Cremades, Marie Materna, Axel Cederholm, Masato Ogishi, Quentin Philippot, Omer Beganovic, Mania Ackermann, Margareta Wuyts, Taushif Khan, Sébastien Fouéré, Florian Herms, Johan Chanal, Boaz Palterer, Julie Bruneau, Thierry J. Molina, Stéphanie Leclerc-Mercier, Jean Luc Prétet, Leila Youssefian, Hassan Vahidnezhad, Nima Parvaneh, Kristl G. Claeys, Rik Schrijvers, Marine Luka, Philippe Pérot, Jacques Fourgeaud, Céline Nourrisson, Philippe Poirier, Emmanuelle Jouanguy, Stéphanie Boisson-Dupuis, Jacinta Bustamante, Luigi D. Notarangelo, Neil Christensen, Nils Landegren, Laurent Abel, Nico Marr, Emmanuelle Six, David Langlais, Tim Waterboer, Florent Ginhoux, Cindy S. Ma, Stuart G. Tangye, Isabelle Meyts, Nico Lachmann, Jiafen Hu, Mohammad Shahrooei, Xavier Bossuyt, Jean Laurent Casanova, Vivien Béziat

Research output: Contribution to journal › Article › peer-review

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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.

Original language	English (US)
Pages (from-to)	2817-2837.e31
Journal	Cell
Volume	187
Issue number	11
DOIs	https://doi.org/10.1016/j.cell.2024.04.009
State	Published - May 23 2024

All Science Journal Classification (ASJC) codes

General Biochemistry, Genetics and Molecular Biology

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10.1016/j.cell.2024.04.009

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Momenilandi, M., Lévy, R., Sobrino, S., Li, J., Lagresle-Peyrou, C., Esmaeilzadeh, H., Fayand, A., Le Floc'h, C., Guérin, A., Mina, E. D., Shearer, D., Delmonte, O. M., Yatim, A., Mulder, K., Mancini, M., Rinchai, D., Denis, A., Neehus, A. L., Balogh, K., ... Béziat, V. (2024). FLT3L governs the development of partially overlapping hematopoietic lineages in humans and mice. Cell, 187(11), 2817-2837.e31. https://doi.org/10.1016/j.cell.2024.04.009

@article{34e2bb7b41654b3c99c77870abd99fa0,

title = "FLT3L governs the development of partially overlapping hematopoietic lineages in humans and mice",

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{\textquoteright} 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{\textquoteright} 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.",

author = "Mana Momenilandi and Romain L{\'e}vy and Steicy Sobrino and Jingwei Li and Chantal Lagresle-Peyrou and Hossein Esmaeilzadeh and Antoine Fayand and {Le Floc'h}, Corentin and Antoine Gu{\'e}rin and Mina, {Erika Della} and Debra Shearer and Delmonte, {Ottavia M.} and Ahmad Yatim and Kevin Mulder and Mathieu Mancini and Darawan Rinchai and Adeline Denis and Neehus, {Anna Lena} and Karla Balogh and Sarah Brendle and Hassan Rokni-Zadeh and Majid Changi-Ashtiani and Yoann Seeleuthner and Caroline Deswarte and Boris Bessot and Cassandre Cremades and Marie Materna and Axel Cederholm and Masato Ogishi and Quentin Philippot and Omer Beganovic and Mania Ackermann and Margareta Wuyts and Taushif Khan and S{\'e}bastien Fou{\'e}r{\'e} and Florian Herms and Johan Chanal and Boaz Palterer and Julie Bruneau and Molina, {Thierry J.} and St{\'e}phanie Leclerc-Mercier and Pr{\'e}tet, {Jean Luc} and Leila Youssefian and Hassan Vahidnezhad and Nima Parvaneh and Claeys, {Kristl G.} and Rik Schrijvers and Marine Luka and Philippe P{\'e}rot and Jacques Fourgeaud and C{\'e}line Nourrisson and Philippe Poirier and Emmanuelle Jouanguy and St{\'e}phanie Boisson-Dupuis and Jacinta Bustamante and Notarangelo, {Luigi D.} and Neil Christensen and Nils Landegren and Laurent Abel and Nico Marr and Emmanuelle Six and David Langlais and Tim Waterboer and Florent Ginhoux and Ma, {Cindy S.} and Tangye, {Stuart G.} and Isabelle Meyts and Nico Lachmann and Jiafen Hu and Mohammad Shahrooei and Xavier Bossuyt and Casanova, {Jean Laurent} and Vivien B{\'e}ziat",

note = "Publisher Copyright: {\textcopyright} 2024 The Authors",

year = "2024",

month = may,

day = "23",

doi = "10.1016/j.cell.2024.04.009",

language = "English (US)",

volume = "187",

pages = "2817--2837.e31",

journal = "Cell",

issn = "0092-8674",

publisher = "Elsevier B.V.",

number = "11",

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Momenilandi, M, Lévy, R, Sobrino, S, Li, J, Lagresle-Peyrou, C, Esmaeilzadeh, H, Fayand, A, Le Floc'h, C, Guérin, A, Mina, ED, Shearer, D, Delmonte, OM, Yatim, A, Mulder, K, Mancini, M, Rinchai, D, Denis, A, Neehus, AL, Balogh, K, Brendle, S, Rokni-Zadeh, H, Changi-Ashtiani, M, Seeleuthner, Y, Deswarte, C, Bessot, B, Cremades, C, Materna, M, Cederholm, A, Ogishi, M, Philippot, Q, Beganovic, O, Ackermann, M, Wuyts, M, Khan, T, Fouéré, S, Herms, F, Chanal, J, Palterer, B, Bruneau, J, Molina, TJ, Leclerc-Mercier, S, Prétet, JL, Youssefian, L, Vahidnezhad, H, Parvaneh, N, Claeys, KG, Schrijvers, R, Luka, M, Pérot, P, Fourgeaud, J, Nourrisson, C, Poirier, P, Jouanguy, E, Boisson-Dupuis, S, Bustamante, J, Notarangelo, LD, Christensen, N, Landegren, N, Abel, L, Marr, N, Six, E, Langlais, D, Waterboer, T, Ginhoux, F, Ma, CS, Tangye, SG, Meyts, I, Lachmann, N, Hu, J, Shahrooei, M, Bossuyt, X, Casanova, JL & Béziat, V 2024, 'FLT3L governs the development of partially overlapping hematopoietic lineages in humans and mice', Cell, vol. 187, no. 11, pp. 2817-2837.e31. https://doi.org/10.1016/j.cell.2024.04.009

TY - JOUR

T1 - FLT3L governs the development of partially overlapping hematopoietic lineages in humans and mice

AU - Momenilandi, Mana

AU - Lévy, Romain

AU - Sobrino, Steicy

AU - Li, Jingwei

AU - Lagresle-Peyrou, Chantal

AU - Esmaeilzadeh, Hossein

AU - Fayand, Antoine

AU - Le Floc'h, Corentin

AU - Guérin, Antoine

AU - Mina, Erika Della

AU - Shearer, Debra

AU - Delmonte, Ottavia M.

AU - Yatim, Ahmad

AU - Mulder, Kevin

AU - Mancini, Mathieu

AU - Rinchai, Darawan

AU - Denis, Adeline

AU - Neehus, Anna Lena

AU - Balogh, Karla

AU - Brendle, Sarah

AU - Rokni-Zadeh, Hassan

AU - Changi-Ashtiani, Majid

AU - Seeleuthner, Yoann

AU - Deswarte, Caroline

AU - Bessot, Boris

AU - Cremades, Cassandre

AU - Materna, Marie

AU - Cederholm, Axel

AU - Ogishi, Masato

AU - Philippot, Quentin

AU - Beganovic, Omer

AU - Ackermann, Mania

AU - Wuyts, Margareta

AU - Khan, Taushif

AU - Fouéré, Sébastien

AU - Herms, Florian

AU - Chanal, Johan

AU - Palterer, Boaz

AU - Bruneau, Julie

AU - Molina, Thierry J.

AU - Leclerc-Mercier, Stéphanie

AU - Prétet, Jean Luc

AU - Youssefian, Leila

AU - Vahidnezhad, Hassan

AU - Parvaneh, Nima

AU - Claeys, Kristl G.

AU - Schrijvers, Rik

AU - Luka, Marine

AU - Pérot, Philippe

AU - Fourgeaud, Jacques

AU - Nourrisson, Céline

AU - Poirier, Philippe

AU - Jouanguy, Emmanuelle

AU - Boisson-Dupuis, Stéphanie

AU - Bustamante, Jacinta

AU - Notarangelo, Luigi D.

AU - Christensen, Neil

AU - Landegren, Nils

AU - Abel, Laurent

AU - Marr, Nico

AU - Six, Emmanuelle

AU - Langlais, David

AU - Waterboer, Tim

AU - Ginhoux, Florent

AU - Ma, Cindy S.

AU - Tangye, Stuart G.

AU - Meyts, Isabelle

AU - Lachmann, Nico

AU - Hu, Jiafen

AU - Shahrooei, Mohammad

AU - Bossuyt, Xavier

AU - Casanova, Jean Laurent

AU - Béziat, Vivien

PY - 2024/5/23

Y1 - 2024/5/23

N2 - 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.

AB - 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.

UR - http://www.scopus.com/inward/record.url?scp=85192102496&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85192102496&partnerID=8YFLogxK

U2 - 10.1016/j.cell.2024.04.009

DO - 10.1016/j.cell.2024.04.009

M3 - Article

C2 - 38701783

AN - SCOPUS:85192102496

SN - 0092-8674

VL - 187

SP - 2817-2837.e31

JO - Cell

JF - Cell

IS - 11

ER -

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

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