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[Clinical features and genotype-phenotype studies of 89 Chinese patients with X-linked adrenoleukodystrophy].
[adrenomyeloneuropathy]
X-
linked
adrenoleukodystrophy
(
X-
ALD
)
is
the
most
common
inherited
disorder
of
peroxisomal
metabolism
which
is
characterized
by
demyelination
of
central
nervous
system
,
impaired
adrenal
cortex
and
abnormal
accumulation
of
very
long
chain
fatty
acids
in
body
fluid
and
tissues
.
In
this
study
,
the
clinical
features
and
the
genotype-phenotype
relationship
were
investigated
so
as
to
help
early
diagnosis
.
Clinical
data
of
89
Chinese
patients
with
X-
ALD
were
analyzed
and
mutation
spectrums
in
53
of
the
patients
were
investigated
by
polymerase
chain
reaction
and
DNA
sequencing
.
Of
the
89
cases
,
60
(
67
.
4
%
)
had
childhood
cerebral
ALD
(
CCALD
,
mean
age
of
onset
was
6
.
5
years
,
range
2
-
10
years
)
,
18
(
20
.
2
%
)
had
adolescent
cerebral
ALD
(
ACALD
,
mean
age
of
onset
12
.
1
years
,
range
11
-
19
years
)
,
7
(
7
.
9
%
)
had
adrenomyeloneuropathy
(
AMN
,
mean
age
of
onset
23
years
,
range
6
-
39
.
5
years
)
,
and
two
cases
were
asymptomatic
and
another
2
had
simple
Addison
's
disease
only
.
CCALD
was
the
most
common
and
severe
phenotype
,
visual
impairment
was
the
most
common
initial
symptom
in
CCALD
and
ACALD
patients
.
Twenty
four
cases
(
63
%
)
in
whom
hydrocortisone
and
ACTH
were
measured
showed
adrenal
insufficiency
.
Forty
five
different
mutations
were
identified
in
53
patients
.
Missense
mutations
were
the
most
common
.
No
hotspot
mutation
was
found
in
these
patients
and
1415
delAG
,
the
most
frequent
mutation
found
worldwide
seemed
not
to
be
the
real
"
hotspot
"
in
these
Chinese
patients
.
The
same
phenotype
may
be
due
to
diverse
genomic
mutations
.
A
single
mutation
may
result
in
different
phenotypes
even
within
a
family
.
The
phenotype
distribution
,
initial
symptom
and
gene
mutation
spectrum
of
Chinese
patients
may
not
be
completely
consistent
with
those
in
other
countries
.
The
clinical
phenotype
of
the
disease
had
no
definite
relationship
with
the
nature
of
gene
mutations
.
Diseases
Validation
Diseases presenting
"central nervous system"
symptom
22q11.2 deletion syndrome
adrenomyeloneuropathy
alexander disease
aniridia
aromatase deficiency
canavan disease
child syndrome
classical phenylketonuria
congenital toxoplasmosis
cowden syndrome
cushing syndrome
cystinuria
dracunculiasis
erdheim-chester disease
fabry disease
gm1 gangliosidosis
hereditary cerebral hemorrhage with amyloidosis
hirschsprung disease
hodgkin lymphoma, classical
kabuki syndrome
kallmann syndrome
kindler syndrome
krabbe disease
lamellar ichthyosis
legionellosis
liposarcoma
malignant atrophic papulosis
monosomy 21
neonatal adrenoleukodystrophy
phenylketonuria
proteus syndrome
scrub typhus
severe combined immunodeficiency
sneddon syndrome
triple a syndrome
von hippel-lindau disease
waldenström macroglobulinemia
well-differentiated liposarcoma
wiskott-aldrich syndrome
wolf-hirschhorn syndrome
x-linked adrenoleukodystrophy
zellweger syndrome
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