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@ARTICLE{AlJanahi:169364,
author = {A. A. AlJanahi and C. R. Lazzarotto and S. Chen and T.-H.
Shin and S. Cordes and X. Fan and I. Jabara and Y. Zhou and
D. J. Young and B.-C. Lee and K.-R. Yu and Y. Li and B. Toms
and I. Tunc and S. G. Hong and L. L. Truitt and J. Klermund
and G. Andrieux$^*$ and M. Y. Kim and T. Cathomen and S.
Gill and S. Q. Tsai and C. E. Dunbar},
title = {{P}rediction and {V}alidation of {H}ematopoietic {S}tem and
{P}rogenitor {C}ell {O}ff-{T}arget {E}diting in
{T}ransplanted {R}hesus {M}acaques.},
journal = {Molecular therapy},
volume = {30},
number = {1},
issn = {1525-0016},
address = {New York, NY},
publisher = {Nature Publ. Group},
reportid = {DKFZ-2021-01444},
pages = {209-222},
year = {2022},
note = {2022 Jan 5;30(1):209-222},
abstract = {The programmable nuclease technology CRISPR/Cas9 has
revolutionized gene editing in the last decade. Due to the
risk of off-target editing, accurate and sensitive methods
for off-target characterization are crucial prior to
applying CRISPR/Cas9 therapeutically. Here, we utilized a
rhesus macaque model to compare the predictive values of
CIRCLE-seq, an in vitro off-target prediction method, with
in silico prediction (ISP) based solely on genomic sequence
comparisons. We use AmpliSeq HD error-corrected sequencing
to validate off-target sites predicted by CIRCLE-seq and ISP
for a CD33 gRNA with thousands of off-target sites predicted
by ISP and CIRCLE-seq. We found poor correlation between the
sites predicted by the two methods. When almost 500 sites
predicted by each method were analyzed by error-corrected
sequencing of hematopoietic cells following transplantation,
19 off-target sites revealed insertion/deletion mutations.
Of these sites, 8 were predicted by both methods, 8 by
CIRCLE-seq only, and 3 by ISP only. The levels of cells with
these off-target edits exhibited no expansion or abnormal
behavior in vivo in animals followed for up to 2 years. In
addition, we utilized an unbiased method termed CAST-Seq to
search for translocations between the on-target site and
off-target sites present in animals following
transplantation, detecting one specific translocation that
persisted in blood cells for at least one year following
transplantation. In conclusion, neither CIRCLE-seq or ISP
predicted all sites, and a combination of careful gRNA
design, followed by screening for predicted off-target sites
in target cells by multiple methods may be required for
optimizing safety of clinical development.},
cin = {FR01},
ddc = {610},
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:34174439},
doi = {10.1016/j.ymthe.2021.06.016},
url = {https://inrepo02.dkfz.de/record/169364},
}