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Clinical approach to inherited peroxisomal disorders.
[neonatal adrenoleukodystrophy]
At
least
21
genetic
disorders
have
now
been
found
that
are
linked
to
peroxisomal
dysfunction
.
Whatever
the
genetic
defect
might
be
,
peroxisomal
disorders
should
be
considered
in
various
clinical
conditions
,
dependent
on
the
age
of
onset
.
The
prototype
of
peroxisomal
disorders
is
represented
by
'
classical
'
Zellweger
syndrome
(
ZS
)
which
is
the
most
severe
disorder
combining
all
the
characteristic
symptoms
.
ZS
is
characterized
by
the
association
of
errors
of
morphogenesis
,
severe
neurological
dysfunction
,
neurosensory
defects
,
regressive
changes
,
hepatodigestive
involvement
with
failure
to
thrive
,
usually
early
death
,
and
absence
of
recognizable
liver
peroxisomes
.
Other
peroxisomal
disorders
(
pseudo-
Zellweger
syndrome
,
neonatal
adrenoleukodystrophy
(
NALD
)
,
pseudo-
neonatal
adrenoleukodystrophy
,
rhizomelic
chondrodysplasia
punctata
(
RCDP
)
,
and
hyperpipecolic
acidaemia
)
share
some
of
these
symptoms
,
but
with
varying
organ
involvement
,
severity
of
dysfunction
,
and
duration
of
survival
.
The
diagnosis
should
not
cause
difficulty
when
all
the
characteristic
manifestations
are
present
.
Depending
on
the
main
presenting
sign
,
peroxisomal
disorders
in
neonates
should
be
suspected
in
two
categories
of
circumstances
:
polymalformative
syndrome
with
craniofacial
dysmorphism
,
and
severe
neurological
dysfunction
.
During
the
first
6
months
of
life
,
the
predominant
symptoms
may
be
hepatomegaly
,
prolonged
jaundice
,
liver
failure
,
anorexia
,
vomiting
and
diarrhoea
leading
to
failure
to
thrive
resembling
a
malabsorption
syndrome
;
severe
psychomotor
retardation
,
hearing
loss
and
ocular
abnormalities
become
evident
.
Beyond
4
years
of
age
,
behavioural
changes
,
intellectual
deterioration
,
visual
impairment
and
gait
abnormalities
may
be
the
presenting
symptoms
.
Independently
of
the
clinical
symptoms
and
age
of
onset
,
most
peroxisomal
disorders
described
so
far
can
be
clinically
screened
by
recordings
of
electroretinogram
,
visual
-evoked
responses
,
and
brain
auditory-evoked
responses
,
which
are
almost
always
abnormal
.
Nine
of
the
17
peroxisomal
disorders
with
neurological
involvement
are
associated
with
an
accumulation
of
very
long
-chain
fatty
acids
(
VLCFA
)
,
which
suggests
that
assay
of
plasma
VLCFA
should
be
used
as
a
primary
test
.
However
,
assays
of
plasma
phytanic
acid
and
plasma
/
urine
bile
acid
intermediates
should
also
be
performed
in
view
of
the
recent
reports
of
atypical
chondrodysplasia
variants
(
without
rhizomelic
shortening
)
and
isolated
trihydroxycholestanoic
aciduria
.
The
differential
diagnoses
in
various
clinical
conditions
and
age
periods
are
discussed
.
Diseases
Validation
Diseases presenting
"hepatomegaly"
symptom
carcinoma of the gallbladder
congenital toxoplasmosis
cutaneous mastocytosis
lamellar ichthyosis
neonatal adrenoleukodystrophy
scrub typhus
typhoid
waldenström macroglobulinemia
zellweger syndrome
This symptom has already been validated