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[Hematopoietic cell transplantation in X-linked adrenoleukodystrophy].
[adrenomyeloneuropathy]
X-
linked
adrenoluekodystrophy
(
X-
ALD
)
is
a
clinically
heterogenous
disorder
ranging
from
the
rapidly
progressive
childhood
cerebral
form
to
the
milder
adrenomyeloneuropathy
in
adults
,
and
some
persons
with
ALD
protein
(
ALDP
)
defects
may
remain
asymptomatic
for
life
.
Hematopoietic
cell
transplantation
(
HCT
)
is
currently
the
only
effective
therapy
for
cerebral
X-
ALD
.
Stem
cell
sources
for
transplantation
are
bone
marrow
,
peripheral
blood
and
cord
blood
.
The
donor
should
be
HLA
identical
with
the
recipient
and
can
be
found
within
family
members
or
from
unrelated
bone
marrow
or
cord
blood
donor
pools
.
Preparative
conditioning
with
supralethal
chemoradiotherapy
is
needed
for
donor
cell
engraftment
,
and
immunosuppressive
agents
are
given
for
months
as
prophylaxis
against
graft-verus-host
disease
(
GVHD
)
.
Worldwide
data
collection
reported
56
%
of
long
term
survival
.
Neurologic
and
MRI
severity
at
the
time
of
transplantation
was
the
most
important
factor
for
survival
and
the
degree
of
improvement
after
transplantation
.
More
than
90
%
of
early
stage
patients
have
survived
with
good
quality
of
life
for
a
long
term
,
whereas
patients
with
advanced
disease
had
the
higher
mortality
rate
during
transplant
procedures
and
the
neurological
abnormalities
progressed
in
most
of
them
.
Patients
with
a
parietal
-
occipital
lobe
pattern
of
demyelination
demonstrated
a
greater
mean
loss
of
performance
IQ
(
PIQ
)
points
than
patients
with
a
frontal
lobe
pattern
of
demyelination
.
Thus
,
boys
with
early
-
stage
disease
benefit
from
HCT
,
whereas
boys
with
advanced
disease
may
be
candidates
for
experimental
therapies
.
Diseases
Validation
Diseases presenting
"loss of performance"
symptom
adrenomyeloneuropathy
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