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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
"early stage"
symptom
adrenomyeloneuropathy
allergic bronchopulmonary aspergillosis
aromatase deficiency
cadasil
carcinoma of the gallbladder
child syndrome
cholangiocarcinoma
congenital adrenal hyperplasia
esophageal adenocarcinoma
esophageal carcinoma
esophageal squamous cell carcinoma
familial hypocalciuric hypercalcemia
familial mediterranean fever
gm1 gangliosidosis
harlequin ichthyosis
hereditary cerebral hemorrhage with amyloidosis
hodgkin lymphoma, classical
kindler syndrome
lymphangioleiomyomatosis
neonatal adrenoleukodystrophy
pyomyositis
scrub typhus
sneddon syndrome
typhoid
von hippel-lindau disease
zellweger syndrome
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