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Hallopeau-Siemens dystrophic epidermolysis bullosa due to homozygous 5818delC mutation in the COL7A gene.
[dystrophic epidermolysis bullosa]
Epidermolysis
bullosa
(
EB
)
is
a
group
of
inherited
mechanobullous
skin
disease
.
The
dystrophic
EB
(
DEB
)
,
one
subtype
of
EB
,
is
inherited
in
an
autosomal
dominant
DEB
or
in
an
autosomal
recessive
(
RDEB
)
.
DEB
is
caused
by
mutations
in
the
COL
7
A
1
gene
encoding
type
VII
collagen
,
the
major
component
of
anchoring
fibrils
.
Over
300
pathogenic
mutations
have
been
detected
within
COL
7
A
in
DEB
.
Patients
with
the
Hallopeau-
Siemens
type
(
HS
-RDEB
)
,
most
severe
form
of
DEB
,
frequently
have
premature
termination
codon
(
PTC
)
mutations
on
both
alleles
.
PTC
mutations
on
both
alleles
result
in
depleted
mRNA
and
α
1
helix
,
and
failure
to
form
the
triple
helix
structure
characteristic
of
type
VII
collagen
.
As
patients
with
HS
-RDEB
usually
have
a
pair
of
heterozygous
PTC
mutations
,
there
have
been
rarely
reported
homozygous
ones
in
HS
-RDEB
.
We
report
the
first
case
of
HS
-RDEB
homozygous
PTC
mutations
of
5818
delC
in
both
COL
7
A
1
alleles
.
This
case
report
suggests
the
positional
effect
of
PTC
mutations
and
vigilance
against
early
infantile
death
in
EB
including
HS
-RDEB
.
Diseases
Validation
Diseases presenting
"reported homozygous ones"
symptom
dystrophic epidermolysis bullosa
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