Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells

Abstract Clinical application of lentiviral vector (LV)‐based hematopoietic stem and progenitor cells (HSPC) gene therapy is rapidly becoming a reality. Nevertheless, LV‐mediated signaling and its potential functional consequences on HSPC biology remain poorly understood. We unravel here a remarkabl...

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Main Authors: Francesco Piras, Michela Riba, Carolina Petrillo, Dejan Lazarevic, Ivan Cuccovillo, Sara Bartolaccini, Elia Stupka, Bernhard Gentner, Davide Cittaro, Luigi Naldini, Anna Kajaste‐Rudnitski
Format: Article
Language:English
Published: Springer Nature 2017-06-01
Series:EMBO Molecular Medicine
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Online Access:https://doi.org/10.15252/emmm.201707922
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author Francesco Piras
Michela Riba
Carolina Petrillo
Dejan Lazarevic
Ivan Cuccovillo
Sara Bartolaccini
Elia Stupka
Bernhard Gentner
Davide Cittaro
Luigi Naldini
Anna Kajaste‐Rudnitski
author_facet Francesco Piras
Michela Riba
Carolina Petrillo
Dejan Lazarevic
Ivan Cuccovillo
Sara Bartolaccini
Elia Stupka
Bernhard Gentner
Davide Cittaro
Luigi Naldini
Anna Kajaste‐Rudnitski
author_sort Francesco Piras
collection DOAJ
description Abstract Clinical application of lentiviral vector (LV)‐based hematopoietic stem and progenitor cells (HSPC) gene therapy is rapidly becoming a reality. Nevertheless, LV‐mediated signaling and its potential functional consequences on HSPC biology remain poorly understood. We unravel here a remarkably limited impact of LV on the HSPC transcriptional landscape. LV escaped innate immune sensing that instead led to robust IFN responses upon transduction with a gamma‐retroviral vector. However, reverse‐transcribed LV DNA did trigger p53 signaling, activated also by non‐integrating Adeno‐associated vector, ultimately leading to lower cell recovery ex vivo and engraftment in vivo. These effects were more pronounced in the short‐term repopulating cells while long‐term HSC frequencies remained unaffected. Blocking LV‐induced signaling partially rescued both apoptosis and engraftment, highlighting a novel strategy to further dampen the impact of ex vivo gene transfer on HSPC. Overall, our results shed light on viral vector sensing in HSPC and provide critical insight for the development of more stealth gene therapy strategies.
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spelling doaj-art-7e75a85b5c0347b0ba9cf82f60a719be2025-08-20T03:05:53ZengSpringer NatureEMBO Molecular Medicine1757-46761757-46842017-06-01991198121110.15252/emmm.201707922Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cellsFrancesco Piras0Michela Riba1Carolina Petrillo2Dejan Lazarevic3Ivan Cuccovillo4Sara Bartolaccini5Elia Stupka6Bernhard Gentner7Davide Cittaro8Luigi Naldini9Anna Kajaste‐Rudnitski10San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific InstituteCenter for Translational Genomics and Bioinformatics, IRCCS San Raffaele Scientific InstituteSan Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific InstituteCenter for Translational Genomics and Bioinformatics, IRCCS San Raffaele Scientific InstituteSan Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific InstituteSan Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific InstituteCenter for Translational Genomics and Bioinformatics, IRCCS San Raffaele Scientific InstituteSan Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific InstituteCenter for Translational Genomics and Bioinformatics, IRCCS San Raffaele Scientific InstituteSan Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific InstituteSan Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific InstituteAbstract Clinical application of lentiviral vector (LV)‐based hematopoietic stem and progenitor cells (HSPC) gene therapy is rapidly becoming a reality. Nevertheless, LV‐mediated signaling and its potential functional consequences on HSPC biology remain poorly understood. We unravel here a remarkably limited impact of LV on the HSPC transcriptional landscape. LV escaped innate immune sensing that instead led to robust IFN responses upon transduction with a gamma‐retroviral vector. However, reverse‐transcribed LV DNA did trigger p53 signaling, activated also by non‐integrating Adeno‐associated vector, ultimately leading to lower cell recovery ex vivo and engraftment in vivo. These effects were more pronounced in the short‐term repopulating cells while long‐term HSC frequencies remained unaffected. Blocking LV‐induced signaling partially rescued both apoptosis and engraftment, highlighting a novel strategy to further dampen the impact of ex vivo gene transfer on HSPC. Overall, our results shed light on viral vector sensing in HSPC and provide critical insight for the development of more stealth gene therapy strategies.https://doi.org/10.15252/emmm.201707922gene therapyhematopoietic stem and progenitor cellsinnate sensinglentiviral vectorsp53 signaling
spellingShingle Francesco Piras
Michela Riba
Carolina Petrillo
Dejan Lazarevic
Ivan Cuccovillo
Sara Bartolaccini
Elia Stupka
Bernhard Gentner
Davide Cittaro
Luigi Naldini
Anna Kajaste‐Rudnitski
Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells
EMBO Molecular Medicine
gene therapy
hematopoietic stem and progenitor cells
innate sensing
lentiviral vectors
p53 signaling
title Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells
title_full Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells
title_fullStr Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells
title_full_unstemmed Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells
title_short Lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells
title_sort lentiviral vectors escape innate sensing but trigger p53 in human hematopoietic stem and progenitor cells
topic gene therapy
hematopoietic stem and progenitor cells
innate sensing
lentiviral vectors
p53 signaling
url https://doi.org/10.15252/emmm.201707922
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