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Small nerve fiber involvement is frequent in X-linked adrenoleukodystrophy.
[adrenomyeloneuropathy]
To
investigate
the
presence
of
small
nerve
fiber
dysfunction
in
subjects
with
X-
linked
adrenoleukodystrophy
.
Cross-sectional
study
in
which
11
Norwegian
subjects
(
3
males
,
8
females
)
with
X-
linked
adrenoleukodystrophy
,
phenotypes
ranging
from
asymptomatic
to
wheelchair-bound
with
adrenomyeloneuropathy
,
were
investigated
with
neurophysiologic
studies
including
EMG
,
nerve
conduction
velocities
,
quantitative
sensory
testing
,
tests
of
autonomic
function
,
and
skin
biopsy
for
intraepidermal
nerve
fiber
density
measurements
.
We
found
small
nerve
fiber
dysfunction
in
10
of
11
subjects
,
increasing
with
age
and
more
pronounced
in
males
.
Low
intraepidermal
nerve
fiber
densities
were
found
in
5
of
11
subjects
,
indicating
a
loss
of
thin
unmyelinated
nerve
fibers
peripherally
.
Five
of
11
subjects
showed
small
nerve
fiber
dysfunction
despite
normal
nerve
fiber
densities
,
suggesting
possible
involvement
of
the
spinothalamic
tracts
.
Two
subjects
showed
moderate
abnormalities
in
autonomic
function
tests
.
Evidence
of
small
nerve
fiber
dysfunction
was
widespread
in
this
cohort
of
subjects
with
X-
linked
adrenoleukodystrophy
,
with
findings
indicating
loss
of
peripheral
small
nerve
fibers
and
possibly
also
fibers
of
the
spinothalamic
tracts
.
The
results
support
the
theory
of
primary
axonal
degeneration
in
adrenomyeloneuropathy
.
Evidence
of
nervous
system
involvement
was
found
in
all
heterozygotes
,
with
severity
increasing
with
age
.
Clinicians
caring
for
these
patients
should
be
alert
to
signs
of
small
nerve
fiber
involvement
.
Diseases
Validation
Diseases presenting
"skin biopsy"
symptom
adrenomyeloneuropathy
cadasil
cutaneous mastocytosis
dentinogenesis imperfecta
dracunculiasis
epidermolysis bullosa simplex
erdheim-chester disease
erythropoietic protoporphyria
fabry disease
harlequin ichthyosis
inclusion body myositis
junctional epidermolysis bullosa
kindler syndrome
lamellar ichthyosis
malignant atrophic papulosis
monosomy 21
oligodontia
primary effusion lymphoma
primary hyperoxaluria type 1
pyruvate dehydrogenase deficiency
sneddon syndrome
waldenström macroglobulinemia
x-linked adrenoleukodystrophy
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