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Therapeutic chaperone effect of N-octyl 4-epi-β-valienamine on murine G(M1)-gangliosidosis.
[gm1 gangliosidosis]
Therapeutic
chaperone
effect
of
a
valienamine
derivative
N-
octyl
4
-
epi-β-valienamine
(
NOEV
)
was
studied
in
G
(
M
1
)
-
gangliosidosis
model
mice
.
Phamacokinetic
analysis
revealed
rapid
intestinal
absorption
and
renal
excretion
after
oral
administration
.
Intracellular
accumulation
was
not
observed
after
continuous
treatment
.
NOEV
was
delivered
to
the
central
nervous
system
through
the
blood
-
brain
barrier
to
induce
high
expression
of
the
apparently
deficient
β-galactosidase
activity
.
NOEV
treatment
starting
at
the
early
stage
of
disease
resulted
in
remarkable
arrest
of
neurological
progression
within
a
few
months
.
Survival
time
was
significantly
prolonged
.
This
result
suggests
that
NOEV
chaperone
therapy
will
be
clinically
effective
for
prevention
of
neuronal
damage
if
started
early
in
life
hopefully
also
in
human
patients
with
G
(
M
1
)
-
gangliosidosis
.
Diseases
Validation
Diseases presenting
"stage of disease"
symptom
adrenomyeloneuropathy
coats disease
dedifferentiated liposarcoma
gm1 gangliosidosis
hodgkin lymphoma, classical
liposarcoma
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