Interleukin-7 receptor α mutational activation can initiate precursor B-cell acute lymphoblastic leukemia.


Journal

Nature communications
ISSN: 2041-1723
Titre abrégé: Nat Commun
Pays: England
ID NLM: 101528555

Informations de publication

Date de publication:
14 12 2021
Historique:
received: 22 04 2020
accepted: 03 11 2021
entrez: 15 12 2021
pubmed: 16 12 2021
medline: 11 1 2022
Statut: epublish

Résumé

Interleukin-7 receptor α (encoded by IL7R) is essential for lymphoid development. Whether acute lymphoblastic leukemia (ALL)-related IL7R gain-of-function mutations can trigger leukemogenesis remains unclear. Here, we demonstrate that lymphoid-restricted mutant IL7R, expressed at physiological levels in conditional knock-in mice, establishes a pre-leukemic stage in which B-cell precursors display self-renewal ability, initiating leukemia resembling PAX5 P80R or Ph-like human B-ALL. Full transformation associates with transcriptional upregulation of oncogenes such as Myc or Bcl2, downregulation of tumor suppressors such as Ikzf1 or Arid2, and major IL-7R signaling upregulation (involving JAK/STAT5 and PI3K/mTOR), required for leukemia cell viability. Accordingly, maximal signaling drives full penetrance and early leukemia onset in homozygous IL7R mutant animals. Notably, we identify 2 transcriptional subgroups in mouse and human Ph-like ALL, and show that dactolisib and sphingosine-kinase inhibitors are potential treatment avenues for IL-7R-related cases. Our model, a resource to explore the pathophysiology and therapeutic vulnerabilities of B-ALL, demonstrates that IL7R can initiate this malignancy.

Identifiants

pubmed: 34907175
doi: 10.1038/s41467-021-27197-5
pii: 10.1038/s41467-021-27197-5
pmc: PMC8671594
doi:

Substances chimiques

Antineoplastic Agents 0
Interleukin-7 Receptor alpha Subunit 0
Proto-Oncogene Proteins p21(ras) EC 3.6.5.2

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

7268

Subventions

Organisme : NCI NIH HHS
ID : R35 CA197695
Pays : United States

Informations de copyright

© 2021. The Author(s).

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Auteurs

Afonso R M Almeida (ARM)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

João L Neto (JL)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Ana Cachucho (A)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Mayara Euzébio (M)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.
Centro Infantil Boldrini, Campinas, SP, Brazil.

Xiangyu Meng (X)

Institut Curie, PSL Research University, CNRS, UMR144, Equipe Labellisée Ligue contre le Cancer, Paris, France.

Rathana Kim (R)

Hematology Laboratory, Saint-Louis Hospital, AP-HP, Paris, France, and Saint-Louis Research Institute, Université de Paris, INSERM U944/Centre National de la Recherche Scientifique (CNRS) Unité Mixte de Recherche (UMR) 7212, Paris, France.

Marta B Fernandes (MB)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Beatriz Raposo (B)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Mariana L Oliveira (ML)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Daniel Ribeiro (D)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Rita Fragoso (R)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Priscila P Zenatti (PP)

Centro Infantil Boldrini, Campinas, SP, Brazil.

Tiago Soares (T)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Mafalda R de Matos (MR)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Juliana Ronchi Corrêa (JR)

Centro Infantil Boldrini, Campinas, SP, Brazil.

Mafalda Duque (M)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal.

Kathryn G Roberts (KG)

Department of Pathology and Hematological Malignancies Program, St. Jude Children's Research Hospital, Memphis, TN, US.

Zhaohui Gu (Z)

Department of Pathology and Hematological Malignancies Program, St. Jude Children's Research Hospital, Memphis, TN, US.

Chunxu Qu (C)

Department of Pathology and Hematological Malignancies Program, St. Jude Children's Research Hospital, Memphis, TN, US.

Clara Pereira (C)

Smurfit Institute of Genetics, Trinity College Dublin, University of Dublin, Dublin 2, Ireland.

Susan Pyne (S)

Strathclyde Institute of Pharmacy and Biomedical Sciences (SIPBS), University of Strathclyde, Glasgow, Scotland, UK.

Nigel J Pyne (NJ)

Strathclyde Institute of Pharmacy and Biomedical Sciences (SIPBS), University of Strathclyde, Glasgow, Scotland, UK.

Vasco M Barreto (VM)

DNA Breaks Laboratory, CEDOC - Chronic Diseases Research Center, NOVA Medical School - Faculdade de Ciências Médicas, Universidade NOVA de Lisboa, Lisbon, Portugal.

Isabelle Bernard-Pierrot (I)

Institut Curie, PSL Research University, CNRS, UMR144, Equipe Labellisée Ligue contre le Cancer, Paris, France.

Emannuelle Clappier (E)

Hematology Laboratory, Saint-Louis Hospital, AP-HP, Paris, France, and Saint-Louis Research Institute, Université de Paris, INSERM U944/Centre National de la Recherche Scientifique (CNRS) Unité Mixte de Recherche (UMR) 7212, Paris, France.

Charles G Mullighan (CG)

Department of Pathology and Hematological Malignancies Program, St. Jude Children's Research Hospital, Memphis, TN, US.

Ana R Grosso (AR)

UCIBIO, Departamento de Ciências da Vida, Faculdade de Ciências e Tecnologia, Universidade NOVA de Lisboa, Caparica, Portugal.

J Andrés Yunes (JA)

Centro Infantil Boldrini, Campinas, SP, Brazil. andres@boldrini.org.br.

João T Barata (JT)

Instituto de Medicina Molecular João Lobo Antunes, Faculdade de Medicina, Universidade de Lisboa, Lisbon, Portugal. joao_barata@medicina.ulisboa.pt.

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