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[Follow-up of benign recurrent intrahepatic cholestasis (Summerskill-Walshe-Tygstrup syndrome) over 46 years].
[benign recurrent intrahepatic cholestasis]
Benign
recurrent
intrahepatic
cholestasis
(
BRIC
or
Summerskill-
Walshe-
Tygstrup-syndrome
)
is
a
rare
autosomal
recessive
form
of
liver
disease
,
which
usually
becomes
manifest
in
childhood
.
Characteristic
are
recurrent
episodes
of
jaundice
and
itching
of
different
duration
.
Number
and
duration
of
episodic
attack
and
asymptomatic
period
develop
individually
.
For
diagnosis
of
BRIC
following
criteria
are
proposed
:
At
least
three
episodes
of
severe
jaundice
and
pruritus
with
biochemical
evidence
of
cholestasis
,
normal
intra-and
extrahepatic
bile
ducts
on
cholangiography
,
absence
of
a
factor
known
to
produce
intrahepatic
cholestasis
and
symptom-free
intervals
of
several
months
or
years
.
Often
the
diagnosis
of
BRIC
is
made
very
late
and
patients
have
to
suffer
invasive
investigations
(
explorative
laparotomy
)
.
Because
of
the
unknown
pathophysiological
mechanism
there
is
no
specific
treatment
.
We
report
on
a
53
-
year
-old
patient
with
jaundice
,
severe
pruritus
,
vomiting
,
loss
of
hair
and
weight
,
extreme
sleeplessness
and
intractable
cough
.
At
the
onset
of
the
attack
an
increase
of
serum
bilirubin
concentration
and
serum
alkaline
phosphatase
was
observed
,
whereas
aspartate
and
alanine
aminotransferase
and
gamma-glutamyltransferase
were
normal
.
Histological
findings
of
liver
biopsy
revealed
accumulation
of
bile
plugs
in
bile
canaliculi
.
The
long
-term
follow-up
of
our
patient
confirms
that
the
prognosis
is
good
.
Diseases
Validation
Diseases presenting
"vomiting"
symptom
22q11.2 deletion syndrome
alexander disease
alpha-thalassemia
aromatase deficiency
benign recurrent intrahepatic cholestasis
cadasil
carcinoma of the gallbladder
child syndrome
cholangiocarcinoma
congenital toxoplasmosis
cutaneous mastocytosis
dedifferentiated liposarcoma
esophageal squamous cell carcinoma
homocystinuria without methylmalonic aciduria
kallmann syndrome
locked-in syndrome
megacystis-microcolon-intestinal hypoperistalsis syndrome
neonatal adrenoleukodystrophy
primary hyperoxaluria type 1
proteus syndrome
scrub typhus
severe combined immunodeficiency
systemic capillary leak syndrome
triple a syndrome
typhoid
von hippel-lindau disease
This symptom has already been validated