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Data from a large European study indicate that the outcome of primary hyperoxaluria type 1 correlates with the AGXT mutation type.
[primary hyperoxaluria type 1]
Primary
hyperoxaluria
type
1
displays
a
heterogeneous
phenotype
,
likely
to
be
affected
by
genetic
and
non-genetic
factors
,
including
timeliness
of
diagnosis
and
quality
of
care
.
As
previous
genotype-phenotype
studies
were
hampered
by
limited
patient
numbers
the
European
OxalEurope
Consortium
was
constituted
.
This
preliminary
retrospective
report
is
based
on
526
patients
of
which
410
have
the
AGXT
genotype
defined
.
We
grouped
mutations
by
the
predicted
effect
as
null
,
missense
leading
to
mistargeting
(
G
170
R
)
,
and
other
missense
,
and
analyzed
their
phenotypic
correlations
.
Median
age
of
end-
stage
renal
disease
increased
from
9
.
9
for
88
homozygous
null
patients
,
11
.
5
for
42
heterozygous
null
/
missense
,
16
.
9
for
116
homozygous
missense
patients
,
25
.
1
for
61
G
170
R
/
null
patients
,
31
.
2
for
32
G
170
R
/
missense
patients
,
and
33
.
9
years
for
71
homozygous
G
170
R
patients
.
The
outcome
of
some
recurrent
missense
mutations
(
p
.
I
244
T
,
p
.
F
152
I
,
p
.
M
195
R
,
p
.
D
201
E
,
p
.
S
81
L
,
p
.
R
36
C
)
and
an
unprecedented
number
of
G
170
R
homozygotes
is
described
in
detail
.
Diagnosis
is
still
delayed
and
actions
aimed
at
increasing
awareness
of
primary
hyperoxaluria
type
1
are
recommended
.
Thus
,
in
addition
to
G
170
R
,
other
causative
mutations
are
associated
with
later
onset
of
end-
stage
renal
disease
.
The
OxalEurope
registry
will
provide
necessary
tools
for
characterizing
those
genetic
and
non-genetic
factors
through
a
combination
of
genetic
,
functional
,
and
biostatistical
approaches
.
Kidney
International
advance
online
publication
,
2
July
2014
;
doi
:
10
.
1038
/
ki
.
2014
.
222
.
Diseases
Validation
Diseases presenting
"end-stage renal disease"
symptom
cystinuria
fabry disease
familial mediterranean fever
heparin-induced thrombocytopenia
primary hyperoxaluria type 1
von hippel-lindau disease
This symptom has already been validated