Single-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s Macroglobulinemia

Abstract Waldenstrom’s Macroglobulinemia (WM) is an IgM-secreting bone marrow (BM) lymphoma that is preceded by an asymptomatic state (AWM). To dissect tumor-intrinsic and immune mechanisms of progression, we perform single-cell RNA-sequencing on 294,206 BM tumor and immune cells from 30 patients wi...

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Main Authors: Romanos Sklavenitis-Pistofidis, Yoshinobu Konishi, Daniel Heilpern-Mallory, Ting Wu, Nicholas Tsakmaklis, Michelle P. Aranha, Zachary R. Hunter, Alaa K. Ali, Junko Tsuji, Nicholas J. Haradhvala, Elizabeth D. Lightbody, Katherine Towle, Laura Hevenor, Rizwan Romee, Edward L. Briercheck, Eric L. Smith, Christine-Ivy Liacos, Efstathios Kastritis, Meletios A. Dimopoulos, Steven P. Treon, Gad Getz, Irene M. Ghobrial
Format: Article
Language:English
Published: Nature Portfolio 2025-02-01
Series:Nature Communications
Online Access:https://doi.org/10.1038/s41467-025-56323-w
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author Romanos Sklavenitis-Pistofidis
Yoshinobu Konishi
Daniel Heilpern-Mallory
Ting Wu
Nicholas Tsakmaklis
Michelle P. Aranha
Zachary R. Hunter
Alaa K. Ali
Junko Tsuji
Nicholas J. Haradhvala
Elizabeth D. Lightbody
Katherine Towle
Laura Hevenor
Rizwan Romee
Edward L. Briercheck
Eric L. Smith
Christine-Ivy Liacos
Efstathios Kastritis
Meletios A. Dimopoulos
Steven P. Treon
Gad Getz
Irene M. Ghobrial
author_facet Romanos Sklavenitis-Pistofidis
Yoshinobu Konishi
Daniel Heilpern-Mallory
Ting Wu
Nicholas Tsakmaklis
Michelle P. Aranha
Zachary R. Hunter
Alaa K. Ali
Junko Tsuji
Nicholas J. Haradhvala
Elizabeth D. Lightbody
Katherine Towle
Laura Hevenor
Rizwan Romee
Edward L. Briercheck
Eric L. Smith
Christine-Ivy Liacos
Efstathios Kastritis
Meletios A. Dimopoulos
Steven P. Treon
Gad Getz
Irene M. Ghobrial
author_sort Romanos Sklavenitis-Pistofidis
collection DOAJ
description Abstract Waldenstrom’s Macroglobulinemia (WM) is an IgM-secreting bone marrow (BM) lymphoma that is preceded by an asymptomatic state (AWM). To dissect tumor-intrinsic and immune mechanisms of progression, we perform single-cell RNA-sequencing on 294,206 BM tumor and immune cells from 30 patients with AWM/WM, 26 patients with Smoldering Myeloma, and 23 healthy donors. Despite their early stage, patients with AWM present extensive immune dysregulation, including in normal B cells, with disease-specific immune hallmarks. Patient T and NK cells show systemic hypo-responsiveness to interferon, which improves with interferon administration and may represent a therapeutic vulnerability. MYD88-mutant tumors show transcriptional heterogeneity, which can be distilled in a molecular classification, including a DUSP22/CD9-positive subtype, and progression signatures which differentiate IgM MGUS from overt WM and can help advance WM research and clinical practice.
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spelling doaj-art-c62f07b7c8c149ef8acc46413d18417f2025-08-20T02:59:54ZengNature PortfolioNature Communications2041-17232025-02-0116111910.1038/s41467-025-56323-wSingle-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s MacroglobulinemiaRomanos Sklavenitis-Pistofidis0Yoshinobu Konishi1Daniel Heilpern-Mallory2Ting Wu3Nicholas Tsakmaklis4Michelle P. Aranha5Zachary R. Hunter6Alaa K. Ali7Junko Tsuji8Nicholas J. Haradhvala9Elizabeth D. Lightbody10Katherine Towle11Laura Hevenor12Rizwan Romee13Edward L. Briercheck14Eric L. Smith15Christine-Ivy Liacos16Efstathios Kastritis17Meletios A. Dimopoulos18Steven P. Treon19Gad Getz20Irene M. Ghobrial21Department of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteBroad Institute of Massachusetts Institute of Technology (MIT) and HarvardDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteBroad Institute of Massachusetts Institute of Technology (MIT) and HarvardBroad Institute of Massachusetts Institute of Technology (MIT) and HarvardDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Medical Oncology, Dana-Farber Cancer InstituteDepartment of Clinical Therapeutics, National and Kapodistrian University of Athens, School of MedicineDepartment of Clinical Therapeutics, National and Kapodistrian University of Athens, School of MedicineDepartment of Clinical Therapeutics, National and Kapodistrian University of Athens, School of MedicineDepartment of Medical Oncology, Dana-Farber Cancer InstituteBroad Institute of Massachusetts Institute of Technology (MIT) and HarvardDepartment of Medical Oncology, Dana-Farber Cancer InstituteAbstract Waldenstrom’s Macroglobulinemia (WM) is an IgM-secreting bone marrow (BM) lymphoma that is preceded by an asymptomatic state (AWM). To dissect tumor-intrinsic and immune mechanisms of progression, we perform single-cell RNA-sequencing on 294,206 BM tumor and immune cells from 30 patients with AWM/WM, 26 patients with Smoldering Myeloma, and 23 healthy donors. Despite their early stage, patients with AWM present extensive immune dysregulation, including in normal B cells, with disease-specific immune hallmarks. Patient T and NK cells show systemic hypo-responsiveness to interferon, which improves with interferon administration and may represent a therapeutic vulnerability. MYD88-mutant tumors show transcriptional heterogeneity, which can be distilled in a molecular classification, including a DUSP22/CD9-positive subtype, and progression signatures which differentiate IgM MGUS from overt WM and can help advance WM research and clinical practice.https://doi.org/10.1038/s41467-025-56323-w
spellingShingle Romanos Sklavenitis-Pistofidis
Yoshinobu Konishi
Daniel Heilpern-Mallory
Ting Wu
Nicholas Tsakmaklis
Michelle P. Aranha
Zachary R. Hunter
Alaa K. Ali
Junko Tsuji
Nicholas J. Haradhvala
Elizabeth D. Lightbody
Katherine Towle
Laura Hevenor
Rizwan Romee
Edward L. Briercheck
Eric L. Smith
Christine-Ivy Liacos
Efstathios Kastritis
Meletios A. Dimopoulos
Steven P. Treon
Gad Getz
Irene M. Ghobrial
Single-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s Macroglobulinemia
Nature Communications
title Single-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s Macroglobulinemia
title_full Single-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s Macroglobulinemia
title_fullStr Single-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s Macroglobulinemia
title_full_unstemmed Single-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s Macroglobulinemia
title_short Single-cell RNA sequencing defines distinct disease subtypes and reveals hypo-responsiveness to interferon in asymptomatic Waldenstrom’s Macroglobulinemia
title_sort single cell rna sequencing defines distinct disease subtypes and reveals hypo responsiveness to interferon in asymptomatic waldenstrom s macroglobulinemia
url https://doi.org/10.1038/s41467-025-56323-w
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