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[Current understanding and perspectives of lymphangioleiomyomatosis].
[lymphangioleiomyomatosis]
Lymphangioleiomyomatosis
(
LAM
)
is
a
slowly
progressive
neoplastic
disease
characterized
with
proliferation
of
abnormal
smooth
muscle
-like
cells
(
LAM
cells
)
in
the
lungs
and
along
axial
lymphatics
.
Proliferation
of
LAM
cells
are
considered
to
be
driven
by
dysregulated
mTORC
1
signaling
,
that
is
caused
by
mutations
in
either
the
TSC
1
or
TSC
2
gene
in
LAM
cells
.
The
MILES
trial
has
successfully
demonstrated
that
sirolimus
,
a
mTORC
1
inhibitor
,
can
stabilize
pulmonary
function
in
LAM
,
but
its
effect
disappears
once
sirolimus
is
discontinued
.
Limited
ability
of
sirolimus
may
be
due
to
concomitant
activation
of
autophagy
in
LAM
cells
when
mTORC
1
activity
is
suppressed
by
sirolimus
.
Recently
animal
models
for
LAM
have
been
independently
established
by
several
groups
,
which
may
provide
a
platform
for
developing
drugs
interfering
various
steps
in
disease
progression
.
Diseases
Validation
Diseases presenting
"lymphangioleiomyomatosis"
symptom
lymphangioleiomyomatosis
proteus syndrome
This symptom has already been validated