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Reduction of plasma oxalate levels by oral application of Oxalobacter formigenes in 2 patients with infantile oxalosis.
[primary hyperoxaluria type 1]
The
spectrum
of
primary
hyperoxaluria
type
I
is
extremely
heterogeneous
,
ranging
from
singular
to
recurrent
urolithiasis
and
early
end-
stage
renal
disease
(
ESRD
)
.
In
infantile
oxalosis
,
the
most
devastating
form
,
ESRD
occurs
as
early
as
within
the
first
weeks
of
life
.
No
kidney
replacement
therapy
sufficiently
removes
endogenously
overproduced
oxalate
.
However
,
curative
combined
liver
-kidney
transplant
often
is
impracticable
in
small
infants
.
Oxalobacter
formigenes
(
O
formigenes
)
,
an
anaerobic
oxalate-degrading
bacterium
,
is
a
colonizer
of
the
healthy
human
colon
.
Oral
administration
of
O
formigenes
has
been
shown
to
significantly
decrease
urine
and
plasma
oxalate
levels
in
patients
with
primary
hyperoxaluria
.
We
report
compassionate
use
of
O
formigenes
in
two
11
-
month
-old
girls
with
infantile
oxalosis
and
ESRD
.
They
received
O
formigenes
twice
a
day
for
4
weeks
(
or
until
transplant
)
.
Dialysis
regimens
were
unchanged
.
Plasma
oxalate
levels
decreased
from
>
110
μmol
/
L
before
to
71
.
53
μmol
/
L
under
treatment
in
patient
1
and
from
>
90
to
68
.
56
μmol
/
L
(
first
treatment
period
)
and
50
.
05
μmol
/
L
(
second
treatment
period
)
in
patient
2
.
O
formigenes
was
well
tolerated
.
No
serious
side
effects
were
reported
.
Extremely
increased
plasma
oxalate
levels
in
patients
with
infantile
oxalosis
may
enable
intestinal
elimination
of
endogenous
oxalate
in
the
presence
of
O
formigenes
.
Therefore
,
O
formigenes
therapy
may
be
helpful
as
a
bridging
procedure
until
transplant
in
such
patients
.
Diseases
Validation
Diseases presenting
"is a colonizer of the healthy human colon"
symptom
primary hyperoxaluria type 1
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