Removal of innate immune barriers allows efficient transduction of quiescent human hematopoietic stem cells.

antiviral restriction factors gene therapy hematopoietic stem cells innate immunity lentiviral vectors

Journal

Molecular therapy : the journal of the American Society of Gene Therapy
ISSN: 1525-0024
Titre abrégé: Mol Ther
Pays: United States
ID NLM: 100890581

Informations de publication

Date de publication:
20 Nov 2023
Historique:
received: 23 05 2023
revised: 29 09 2023
accepted: 17 11 2023
pubmed: 22 11 2023
medline: 22 11 2023
entrez: 22 11 2023
Statut: aheadofprint

Résumé

Quiescent human hematopoietic stem cells (HSC) are ideal targets for gene therapy applications due to their preserved stemness and repopulation capacities; however, they have not been exploited extensively because of their resistance to genetic manipulation. We report here the development of a lentiviral transduction protocol that overcomes this resistance in long-term repopulating quiescent HSC, allowing their efficient genetic manipulation. Mechanistically, lentiviral vector transduction of quiescent HSC was found to be restricted at the level of vector entry and by limited pyrimidine pools. These restrictions were overcome by the combined addition of cyclosporin H (CsH) and deoxynucleosides (dNs) during lentiviral vector transduction. Clinically relevant transduction levels were paired with higher polyclonal engraftment of long-term repopulating HSC as compared with standard ex vivo cultured controls. These findings identify the cell-intrinsic barriers that restrict the transduction of quiescent HSC and provide a means to overcome them, paving the way for the genetic engineering of unstimulated HSC.

Identifiants

pubmed: 37990494
pii: S1525-0016(23)00621-4
doi: 10.1016/j.ymthe.2023.11.020
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

Copyright © 2023 The Authors. Published by Elsevier Inc. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of interests E.V., G.U., F.P., and A.K.-R. are inventors on pending and issued patents on lentiviral gene transfer filed by the Telethon Foundation and the San Raffaele Scientific Institute.

Auteurs

Erika Valeri (E)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy; Vita-Salute San Raffaele University, School of Medicine, 20132 Milan, Italy.

Giulia Unali (G)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy; Vita-Salute San Raffaele University, School of Medicine, 20132 Milan, Italy.

Francesco Piras (F)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Monah Abou-Alezz (M)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Giulia Pais (G)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Fabrizio Benedicenti (F)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Maria Rosa Lidonnici (MR)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Ivan Cuccovillo (I)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Ilaria Castiglioni (I)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Sergio Arévalo (S)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Giulio Spinozzi (G)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Ivan Merelli (I)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Rayk Behrendt (R)

Institute for Clinical Chemistry and Clinical Pharmacology, University Hospital Bonn, 53127 Bonn, Germany.

Adrian Oo (A)

Department of Pediatrics, Emory University School of Medicine, Atlanta, GA 30322, USA.

Baek Kim (B)

Department of Pediatrics, Emory University School of Medicine, Atlanta, GA 30322, USA.

Nathaniel R Landau (NR)

Department of Microbiology, NYU School of Medicine, New York, NY 10016, USA.

Giuliana Ferrari (G)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy; Vita-Salute San Raffaele University, School of Medicine, 20132 Milan, Italy.

Eugenio Montini (E)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy.

Anna Kajaste-Rudnitski (A)

San Raffaele Telethon Institute for Gene Therapy, IRCCS San Raffaele Scientific Institute, 20132 Milan, Italy; Department of Biology and Biotechnology, University of Pavia, Via Ferrata 9A, 27100 Pavia, Italy. Electronic address: anna.kajaste@unipv.it.

Classifications MeSH