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@ARTICLE{Rhiel:274186,
author = {M. Rhiel and K. Geiger and G. Andrieux$^*$ and J. Rositzka
and M. Börries$^*$ and T. Cathomen and T. I. Cornu},
title = {{T}-{CAST}: {A}n optimized {CAST}-{S}eq pipeline for
{TALEN} confirms superior safety and efficacy of
obligate-heterodimeric scaffolds.},
journal = {Frontiers in genome editing},
volume = {5},
issn = {2673-3439},
address = {Lausanne},
publisher = {Frontiers Media},
reportid = {DKFZ-2023-00481},
pages = {1130736},
year = {2023},
abstract = {Transcription activator-like effector nucleases (TALENs)
are programmable nucleases that have entered the clinical
stage. Each subunit of the dimer consists of a DNA-binding
domain composed of an array of TALE repeats fused to the
catalytically active portion of the FokI endonuclease. Upon
DNA-binding of both TALEN arms in close proximity, the FokI
domains dimerize and induce a staggered-end DNA double
strand break. In this present study, we describe the
implementation and validation of TALEN-specific CAST-Seq
(T-CAST), a pipeline based on CAST-Seq that identifies
TALEN-mediated off-target effects, nominates off-target
sites with high fidelity, and predicts the TALEN pairing
conformation leading to off-target cleavage. We validated
T-CAST by assessing off-target effects of two promiscuous
TALENs designed to target the CCR5 and TRAC loci. Expression
of these TALENs caused high levels of translocations between
the target sites and various off-target sites in primary T
cells. Introduction of amino acid substitutions to the FokI
domains, which render TALENs obligate-heterodimeric
(OH-TALEN), mitigated the aforementioned off-target effects
without loss of on-target activity. Our findings highlight
the significance of T-CAST to assess off-target effects of
TALEN designer nucleases and to evaluate mitigation
strategies, and advocate the use of obligate-heterodimeric
TALEN scaffolds for therapeutic genome editing.},
keywords = {FokI (Other) / TALEN (Other) / chromosomal rearrangements
(Other) / chromosomal translocations (Other) / designer
nuclease (Other) / obligate heterodimer (Other) / off-target
effects (Other) / preclinical risk assessment (Other)},
cin = {FR01},
ddc = {630},
cid = {I:(DE-He78)FR01-20160331},
pnm = {899 - ohne Topic (POF4-899)},
pid = {G:(DE-HGF)POF4-899},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:36890979},
pmc = {pmc:PMC9986454},
doi = {10.3389/fgeed.2023.1130736},
url = {https://inrepo02.dkfz.de/record/274186},
}