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Severe hypercalcemia in a 9-year-old Brazilian girl due to a novel inactivating mutation of the calcium-sensing receptor.
[familial hypocalciuric hypercalcemia]
Familial
hypocalciuric
hypercalcemia
(
FHH
)
and
neonatal
severe
hyperparathyroidism
(
NSHPT
)
are
consequent
to
inactivating
mutations
of
the
calcium-sensing
receptor
(
CaR
)
gene
.
FHH
is
usually
associated
with
heterozygous
inactivating
mutations
of
the
CaR
gene
,
whereas
NSHPT
is
usually
due
to
homozygous
inactivation
of
the
CaR
gene
.
FHH
is
generally
asymptomatic
and
is
characterized
by
mild
to
moderate
lifelong
hypercalcemia
,
relative
hypocalciuria
,
and
normal
intact
PTH
,
whereas
individuals
with
NSHPT
frequently
show
life-threatening
hypercalcemia
.
In
this
study
,
we
report
a
novel
inactivating
mutation
of
the
CaR
gene
,
identified
in
a
9
-
yr
-old
Brazilian
girl
who
was
found
to
be
severely
hypercalcemic
during
investigation
of
a
6
-
month
history
of
headaches
and
vomits
.
Direct
sequencing
of
the
CaR
gene
from
this
patient
showed
a
novel
homozygous
mutation
(
L
13
P
)
in
exon
2
.
Functional
characterization
by
intracellular
calcium
measurement
by
fluorometry
showed
that
the
mutant
receptor
had
a
dose-response
curve
shifted
to
the
right
relative
to
that
of
wild
type
.
The
proband
's
consanguineous
parents
,
who
had
mild
asymptomatic
hypercalcemia
,
showed
the
same
mutation
in
the
heterozygous
form
.
The
mutation
described
in
this
study
is
the
inactivating
missense
mutation
present
at
the
most
N-
terminal
end
among
the
known
CaR
missense
mutations
.
This
study
reinforces
the
fact
that
patients
with
homozygous
inactivation
of
the
CaR
gene
may
present
with
severe
hypercalcemia
in
different
phases
of
life
.
Diseases
Validation
Diseases presenting
"headaches"
symptom
alexander disease
cadasil
cutaneous mastocytosis
familial hypocalciuric hypercalcemia
hydrocephalus with stenosis of the aqueduct of sylvius
sneddon syndrome
thoracic outlet syndrome
This symptom has already been validated