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Intravenous immunoglobulin treatment in a patient with adrenomyeloneuropathy.
[adrenomyeloneuropathy]
Adrenomyeloneuropathy
(
AMN
)
is
one
of
several
phenotypes
of
the
adrenoleukodystrophy
spectrum
caused
by
mutations
in
the
ABCD
1
gene
on
the
X
chromosome
.
An
inflammatory
component
is
part
of
the
disease
complex
ranging
from
severe
childhood
CNS
demyelination
to
spinal
cord
and
peripheral
nerve
degeneration
.
We
present
a
patient
with
clinical
progressive
AMN
and
severe
lower
limb
pain
.
Longitudinal
brain
magnetic
resonance
spectroscopy
showed
a
constant
slightly
elevated
myoinositol
/
total
creatine
ratio
during
the
five
year
treatment
period
,
probably
reflecting
demyelination
,
microglial
activation
and
gliosis
,
indicating
an
inflammatory
response
.
The
pain
was
refractory
to
conventional
therapy
but
intravenous
immunoglobulin
(
IVIG
)
treatment
was
highly
efficient
.
IVIG
may
be
considered
as
a
last
resort
for
treatment
of
refractory
pain
in
AMN
patients
with
indications
of
an
inflammatory
component
.
Diseases
Validation
Diseases presenting
"severe lower limb pain"
symptom
adrenomyeloneuropathy
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