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Confirmation of oxidative stress and fatty acid disturbances in two further Papillon-Lefèvre syndrome families with identification of a new mutation.
[papillon-lefèvre syndrome]
We
have
previously
reported
oxidative
and
fatty
acids
disturbances
in
one
Papillon-
Lefèvre
syndrome
(
PLS
)
family
.
This
Mendelian
condition
characterized
by
palmar
plantar
keratosis
and
severe
aggressive
periodontitis
,
is
caused
by
mutations
in
the
cathepsin
C
(
CTSC
)
gene
.
In
this
study
,
we
have
analysed
two
further
unrelated
PLS
families
to
confirm
this
association
.
Mutations
were
identified
by
direct
sequencing
of
CTSC
.
Biochemical
analyses
were
performed
in
probands
and
their
relatives
in
order
to
determine
plasma
levels
of
vitamin
E
,
CoQ
10
,
lipid
hydroperoxides
(
HP
)
and
fatty
acid
patterns
.
Pathogenic
CTSC
mutations
were
identified
in
both
families
including
a
new
mutation
(
c
504
C
>
G
)
.
Both
probands
showed
low
levels
of
vitamin
E
and
CoQ
10
,
and
high
levels
of
lipid
HP
,
and
also
very
low
levels
of
docohexaenoic
acid
.
The
previously
reported
oxidative
and
fatty
acids
disturbances
were
confirmed
as
a
feature
of
this
condition
in
two
further
families
.
There
are
low
levels
of
antioxidant
markers
and
high
levels
of
oxidative
markers
,
in
addition
of
low
levels
of
some
anti-
inflammatory
fatty
acids
in
persons
suffering
PLS
and
some
of
their
relatives
.
Diseases
Validation
Diseases presenting
"low levels"
symptom
22q11.2 deletion syndrome
adrenal incidentaloma
adrenomyeloneuropathy
aromatase deficiency
benign recurrent intrahepatic cholestasis
cadasil
congenital adrenal hyperplasia
congenital diaphragmatic hernia
esophageal carcinoma
hodgkin lymphoma, classical
liposarcoma
malignant atrophic papulosis
megacystis-microcolon-intestinal hypoperistalsis syndrome
neonatal adrenoleukodystrophy
omenn syndrome
oral submucous fibrosis
papillon-lefèvre syndrome
primary effusion lymphoma
sneddon syndrome
x-linked adrenoleukodystrophy
zellweger syndrome
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