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A cross-sectional multicenter study of osteogenesis imperfecta in North America - results from the linked clinical research centers.
[dentinogenesis imperfecta]
Osteogenesis
imperfecta
(
OI
)
is
the
most
common
skeletal
dysplasia
that
predisposes
to
recurrent
fractures
and
bone
deformities
.
In
spite
of
significant
advances
in
understanding
the
genetic
basis
of
OI
,
there
have
been
no
large
-scale
natural
history
studies
.
To
better
understand
the
natural
history
and
improve
the
care
of
patients
,
a
network
of
Linked
Clinical
Research
Centers
(
LCRC
)
was
established
.
Subjects
with
OI
were
enrolled
in
a
longitudinal
study
,
and
in
this
report
,
we
present
cross-sectional
data
on
the
largest
cohort
of
OI
subjects
(
n
 
=
 
544
)
.
OI
type
III
subjects
had
higher
prevalence
of
dentinogenesis
imperfecta
,
severe
scoliosis
,
and
long
bone
deformities
as
compared
to
those
with
OI
types
I
and
IV
.
Whereas
the
mean
lumbar
spine
area
bone
mineral
density
(
LS
aBMD
)
was
low
across
all
OI
subtypes
,
those
with
more
severe
forms
had
lower
bone
mass
.
Molecular
testing
may
help
predict
the
subtype
in
type
I
collagen-related
OI
.
Analysis
of
such
well-collected
and
unbiased
data
in
OI
can
not
only
help
answering
questions
that
are
relevant
to
patient
care
but
also
foster
hypothesis-driven
research
,
especially
in
the
context
of
'
phenotypic
expansion
'
driven
by
next
-generation
sequencing
.
Diseases
Validation
Diseases presenting
"bone deformities"
symptom
dentinogenesis imperfecta
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