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@ARTICLE{Chatain:280470,
author = {Chatain, N. and Perera, R. C. and Rossetti, G. and Rossa,
J. and Carloni, P. and Schemionek, M. and Haferlach, T. and
Brümmendorf, T. H. and Schnittger, S. and Koschmieder, S.},
title = {{R}are {FLT}3 deletion mutants may provide additional
treatment options to patients with {AML}: an approach to
individualized medicine},
journal = {Leukemia},
volume = {29},
number = {12},
issn = {1476-5551},
address = {Basingstoke},
publisher = {Nature Publ. Group},
reportid = {FZJ-2016-00244},
pages = {2434 - 2438},
year = {2015},
abstract = {The receptor tyrosine kinase (RTK) FLT3 (Fms-like tyrosine
kinase-3) is essential for the proliferation,
differentiation and survival of hematopoietic cells.1, 2
Mutations in the gene for FLT3 have been described in acute
myeloid leukemia (AML) as internal tandem duplications (ITD)
localized in the juxtamembrane (JM) domain and FLT3 tyrosine
kinase domain (TKD) mutations amounting to $2–30\%$ and
$7–10\%$ of patients, respectively. In patients with AML,
FLT3-ITD confers an unfavorable prognosis.3 In AML, recently
discovered FLT3 mutations involve point mutations as well as
short deletions in the JM domain, highlighting the
importance for a deeper understanding and further studies of
these aberrations.4, 5 The FLT3 JM domain (Supplementary
Figure 1a) is indeed the critical regulator region of
receptor autoinhibition.6 Kiyoi et al.7 had shown
constitutive activation of the FLT3 receptor by deletion of
a stretch of amino acids in the JM domain, generally
duplicated in FLT3-ITD.In this study, during diagnostic
screening for ITD and TKD mutations, we have identified and
functionally characterized deletion mutations of FLT3 in the
diagnostic work-up of over 6843 AML patients. Deletions were
detected in seven patients $(0.1\%)$ with newly diagnosed or
relapsed AML. All deletions detected by gene scan were in
the range of 3–18 base pairs (Supplementary Table 1 and
Figure 1a). Subsequent sequencing revealed that all but one
of these deletions, which was situated in the transmembrane
domain, were in the JM domain, and were simple deletions
(n=5) or indels (n=2). In three cases out of seven, the
mutations resulted in a premature stop codon. As loss of the
wild-type (WT) allele in FLT3-ITD knock-in mice led to a
more severe phenotype,8 we analyzed whether the FLT3
mutations were biallelic. However, we could not detect ITD
or TKD mutations on the other allele of these patients.
Remission samples of patient Id 2–7 confirmed the somatic
character of the mutations. We analyzed the transforming
potential of such deletions and performed computational
modeling to predict the structural effect of two deletions
(Id 5: $p.Glu598_Tyr599del—delEY;$ Id 6:
$p.Phe590_Asp593delinsLeuTyr—delIns).$},
cin = {JSC / INM-9 / IAS-5},
ddc = {610},
cid = {I:(DE-Juel1)JSC-20090406 / I:(DE-Juel1)INM-9-20140121 /
I:(DE-Juel1)IAS-5-20120330},
pnm = {511 - Computational Science and Mathematical Methods
(POF3-511) / 572 - (Dys-)function and Plasticity (POF3-572)},
pid = {G:(DE-HGF)POF3-511 / G:(DE-HGF)POF3-572},
typ = {PUB:(DE-HGF)16},
UT = {WOS:000366393900030},
doi = {10.1038/leu.2015.131},
url = {https://juser.fz-juelich.de/record/280470},
}