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The FEN1 E359K germline mutation disrupts the FEN1-WRN interaction and FEN1 GEN activity, causing aneuploidy-associated cancers.
[werner syndrome]
Polymorphisms
and
somatic
mutations
in
Flap
Endonuclease
1
(
FEN
1
)
,
an
essential
enzyme
involved
in
DNA
replication
and
repair
,
can
lead
to
functional
deficiencies
of
the
FEN
1
protein
and
a
predisposition
to
cancer
.
We
identified
a
FEN
1
germline
mutation
that
changed
residue
E
359
to
K
in
a
patient
whose
family
had
a
history
of
breast
cancer
.
We
determined
that
the
E
359
K
mutation
,
which
is
in
the
protein-protein
domain
of
FEN
1
,
abolished
the
interaction
of
FEN
1
with
Werner
syndrome
protein
(
WRN
)
,
an
interaction
that
is
critical
for
resolving
stalled
DNA
replication
forks
.
Furthermore
,
although
the
flap
endonuclease
activity
of
FEN
1
E
359
K
was
unaffected
,
it
failed
to
resolve
bubble
structures
,
which
require
the
FEN
1
gap-dependent
endonuclease
activity
.
To
determine
the
etiological
significance
of
E
359
K
,
we
established
a
mouse
model
containing
this
mutation
.
E
359
K
mouse
embryonic
fibroblasts
(
MEF
)
were
more
sensitive
to
DNA
crosslinking
agents
that
cause
replication
forks
to
stall
.
Cytological
analysis
suggested
that
the
FEN
1
-
WRN
interaction
was
also
required
for
telomere
stability
;
mutant
cell
lines
had
fragile
telomeres
,
increased
numbers
of
spontaneous
chromosomal
anomalies
and
higher
frequencies
of
transformation
.
Moreover
,
the
incidence
of
cancer
was
significantly
higher
in
mice
homozygous
for
FEN
1
E
359
K
than
in
wild-
type
mice
,
suggesting
that
the
FEN
1
E
359
K
mutation
is
oncogenic
.
Oncogene
advance
online
publication
,
10
March
2014
;
doi
:
10
.
1038
/
onc
.
2014
.
19
.
Diseases
Validation
Diseases presenting
"cancer"
symptom
achondroplasia
acute rheumatic fever
adrenal incidentaloma
alpha-thalassemia
benign recurrent intrahepatic cholestasis
cadasil
canavan disease
carcinoma of the gallbladder
cholangiocarcinoma
coats disease
congenital adrenal hyperplasia
congenital diaphragmatic hernia
cowden syndrome
cushing syndrome
cutaneous mastocytosis
dedifferentiated liposarcoma
dystrophic epidermolysis bullosa
epidermolysis bullosa simplex
erdheim-chester disease
erythropoietic protoporphyria
esophageal adenocarcinoma
esophageal carcinoma
esophageal squamous cell carcinoma
familial hypocalciuric hypercalcemia
familial mediterranean fever
gm1 gangliosidosis
heparin-induced thrombocytopenia
hereditary cerebral hemorrhage with amyloidosis
hirschsprung disease
hodgkin lymphoma, classical
inclusion body myositis
junctional epidermolysis bullosa
kabuki syndrome
kallmann syndrome
kindler syndrome
lamellar ichthyosis
liposarcoma
locked-in syndrome
lymphangioleiomyomatosis
monosomy 21
neuralgic amyotrophy
oculocutaneous albinism
oligodontia
oral submucous fibrosis
papillon-lefèvre syndrome
pendred syndrome
pleomorphic liposarcoma
primary effusion lymphoma
proteus syndrome
pyomyositis
pyruvate dehydrogenase deficiency
severe combined immunodeficiency
sneddon syndrome
systemic capillary leak syndrome
triple a syndrome
von hippel-lindau disease
waldenström macroglobulinemia
well-differentiated liposarcoma
werner syndrome
wiskott-aldrich syndrome
wolf-hirschhorn syndrome
x-linked adrenoleukodystrophy
This symptom has already been validated