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1029
Case Report
Cholangitis
Candida D. Randall
Radin1
and
Meade
in a Diabetic
Woman
B. Johnson
formed. The patient was seronegative for antibodies to HIV. Repeat endoscopic retrograde cholangiography 1 week after the sphincterotomy showed no change (Fig. 1 B). Because of progressive increase of serum levels of alkaline phosphatase and bilirubin, a biliary stent
Candidiasis of the biliary tract is rare; only about a dozen well-documented cases have been published [1 -3]. We report the sonographic and cholangiographic findings in a diabetic woman with Candida cholangitis without infection elsewhere.
was placed. Biopsy specimen necrotic
Case Report A 39-year-old woman was admitted episode in a week. She had a 24-year
to another
hospital
had had cholecystectomy tive
cholangiogram
tamed
multiple
was
stones,
because
and placement reported
Fungal
pneumoniae.
cholangitis
of a T tube.
to be normal.
and pathologic
acute and chronic cholecystitis. Klebsiella
of ascending
evaluation
The
showed
stains
were
negative.
be severely hypotension,
of the distal common
and debris decreased,
biliary
was
of aspirated
photericin B was bilirubin increased,
but
dilatation
repeated
bile yielded
bile duct showed The serum level
with Candida. the
serum
level
was seen when
1 1 days
after
Candida
albicans,
the
of
bilirubin
retrograde
second
study.
Candida
krusei,
Staphylococcus epidermidis. Treatment with ambegun. Serum levels of alkaline phosphatase and but the patient was afebrile and did not appear to
ill or to have sepsis. One week later, hypothermia, and severe hypoglycemia developed. The patient died
5 weeks after admission. Autopsy showed invasive biliary candidiasis and multiple liver abscesses caused by C. albicans and bacteria. No other site of Candida infection was present.
con-
necrotizing
bile yielded
Serum
Increased
enterococcus, and
An intraopera-
Culture of intracholecystic
phosphatase
increased. Cultures
and
gallbladder
tissue
of alkaline cholangiography
with a third hypoglycemic history of insulin-dependent
diabetes mellitus, complicated by multiple episodes of ketoacidosis, severe retinopathy, enteropathy, neuropathy, and nephropathy, which required hemodialysis. Three weeks earlier she had been admitted
bile-stained
bilirubin
level decreased from 13.2 to 2.8 mg/dl. Hepatobiliary scintigraphy showed accumulation of radionuclide in the small bowel. The T tube
Discussion
was removed, and the patient was discharged 2 weeks after surgery. On this admission, she was afebrile but icteric, with marked elevation of serum levels of alkaline phosphatase (2599 U/I) and bilirubin (1 4.6 mg/dl). Sonography showed thickening of the wall of the common duct and nonshadowing echogenic material throughout the lumen (Fig. 1 A). Fever developed, and treatment with ampicillin, gentamicin, and metronidazole was begun. Endoscopic retrograde
Although virtually every organ may be involved, candidiasis of the biliary tract is rare, even in large series of patients with disseminated infection [1]. In 1 0 of 1 2 reported cases of biliary candidiasis ([1 -3] and this case), the infection was not systemic. In 1 1 of the 1 2 patients, one or more of these predisposing factors was present: previous cholecystectomy or other major operation, treatment with broad-spectrum antibiotics, diabetes mellitus, and acute leukemia. None of the patients had AIDS. No apparent predisposing factor was present in a 3V2-year-old girl who had a fungus ball in the common bile duct 2 months after blunt abdominal trauma [2].
cholangiography
mural irregularity,
a diffusely abnormal biliary tree with marked multiple strictures, and segments of mild dilatation.
showed
Apparent dissection bile duct was seen. gitis
and
primary
of contrast Differential sclerosing
material diagnosis cholangitis.
in the wall of the common included infectious cholanSphincterotomy
was
per-
Received August 15, 1991 ; accepted after revision September 23, 1991. 1 Both authors: Department of Radiology, University of Southern California Center, 1200 N. State St., Los Angeles, CA 90033-1 084. AJR 158:1029-1030,
May
1992 0361-8o3x/92/1585-1029
C American
School
Roentgen
of Medicine,
Ray Society
Los Angeles
County-University
of Southern
California
Medical
RADIN
1030
AND
JOHNSON
AJR:158,
May 1992
Downloaded from www.ajronline.org by 117.253.210.40 on 11/10/15 from IP address 117.253.210.40. Copyright ARRS. For personal use only; all rights reserved
Fig. 1.-A, Transverse sonogram shows thickened hypoechoic wall of common duct (arrows) and nonshadowing echogenic material filling lumen. Arrowhead = hepatic artery, P = portal vein, C = inferior vena cava, A = aorta, S = spine. B, Endoscopic retrograde cholangiogram
shows
Involvement
marked
mural
tree by strictures and segments of mild dilatation. Channels of contrast material parallel to common duct may represent
In one previously reported case [2], sonography showed a nonshadowing polypoid mass of 7-8 mm in the lumen of the distal common bile duct. Pathologic examination of the contents of the common duct showed Candida embedded in bile sludge material. Sonographic findings in our patient differed in two respects. Thickening of the wall of the common duct reflected the invasive nature of the infection. The echogenic material filling the lumen of the entire common duct probably represented a combination of intraluminal debris and ulcerated mucosa. Cholangiography in four reported cases of biliary candidiasis [1 4] showed one or more intraluminal filling defects representing fungus balls in the common bile duct. In one of these cases [4], several strictures of the intrahepatic and extrahepatic bile ducts also were present. Cholangiographic findings in our patient were far more severe and extensive, consistent with diffuse invasive candidiasis. The radiologic appearance was suggestive of primary sclerosing cholangitis [5] or AIDS-related cholangitis [6]. Candidiasis was not suspected until biopsy was performed. Although rare, the diagnosis of biliary candidiasis should be considered when radiologic findings show cholangitis or an ,
of entire
irregularity
intramural
biliary
with multiple
dissection.
intraluminal filling defect in the common bile duct in a patient with predisposing factors for Candida infection. Bile should be obtained for fungal stain and culture. With appropriate treatment, the majority of patients will make a complete recovery [1].
REFERENCES 1 . Irani M, Truong
2.
LD. Candidiasis
of the extrahepatic
Pathol Lab Med 1986;1 10: 1087-1 090 Carstensen H, Nilsson KO, Nettelblad SV, Cederlund
biliary tract. Arch CG, Hildell
J. Corn-
mon bile duct obstruction
due to an intraluminal mass of candidiasis in a previously healthy child. Pediatrics 1986;77:858-861 3. Chen CC, Chang PY, Chen CL. Refractory cholangitis after Kasai’s operation caused by candidiasis: a case report. J Pediatr Surg 1986;21: 736-737
4. Uflacker A, Wholey MH, Amaral NM, Lima S. Parasitic and mycotic causes of biliary obstruction. Gastrointest Radiol 1982;7: 173-1 79 5. MacCarty AL, LaRusso NF, Wiesner RH, Ludwig J. Primary sclerosing cholangitis: findings 1983;149:39-44
6. Dolmatch
on cholangiography
and pancreatography.
Radiology
BL, Laing FC, Federle MP, Jeffrey RB, Cello J. AIDS-related
cholangitis: radiographic 1987;163:313-316
findings
in
nine
patients.
Radiology
1029
Case Report
Cholangitis
Candida D. Randall
Radin1
and
Meade
in a Diabetic
Woman
B. Johnson
formed. The patient was seronegative for antibodies to HIV. Repeat endoscopic retrograde cholangiography 1 week after the sphincterotomy showed no change (Fig. 1 B). Because of progressive increase of serum levels of alkaline phosphatase and bilirubin, a biliary stent
Candidiasis of the biliary tract is rare; only about a dozen well-documented cases have been published [1 -3]. We report the sonographic and cholangiographic findings in a diabetic woman with Candida cholangitis without infection elsewhere.
was placed. Biopsy specimen necrotic
Case Report A 39-year-old woman was admitted episode in a week. She had a 24-year
to another
hospital
had had cholecystectomy tive
cholangiogram
tamed
multiple
was
stones,
because
and placement reported
Fungal
pneumoniae.
cholangitis
of a T tube.
to be normal.
and pathologic
acute and chronic cholecystitis. Klebsiella
of ascending
evaluation
The
showed
stains
were
negative.
be severely hypotension,
of the distal common
and debris decreased,
biliary
was
of aspirated
photericin B was bilirubin increased,
but
dilatation
repeated
bile yielded
bile duct showed The serum level
with Candida. the
serum
level
was seen when
1 1 days
after
Candida
albicans,
the
of
bilirubin
retrograde
second
study.
Candida
krusei,
Staphylococcus epidermidis. Treatment with ambegun. Serum levels of alkaline phosphatase and but the patient was afebrile and did not appear to
ill or to have sepsis. One week later, hypothermia, and severe hypoglycemia developed. The patient died
5 weeks after admission. Autopsy showed invasive biliary candidiasis and multiple liver abscesses caused by C. albicans and bacteria. No other site of Candida infection was present.
con-
necrotizing
bile yielded
Serum
Increased
enterococcus, and
An intraopera-
Culture of intracholecystic
phosphatase
increased. Cultures
and
gallbladder
tissue
of alkaline cholangiography
with a third hypoglycemic history of insulin-dependent
diabetes mellitus, complicated by multiple episodes of ketoacidosis, severe retinopathy, enteropathy, neuropathy, and nephropathy, which required hemodialysis. Three weeks earlier she had been admitted
bile-stained
bilirubin
level decreased from 13.2 to 2.8 mg/dl. Hepatobiliary scintigraphy showed accumulation of radionuclide in the small bowel. The T tube
Discussion
was removed, and the patient was discharged 2 weeks after surgery. On this admission, she was afebrile but icteric, with marked elevation of serum levels of alkaline phosphatase (2599 U/I) and bilirubin (1 4.6 mg/dl). Sonography showed thickening of the wall of the common duct and nonshadowing echogenic material throughout the lumen (Fig. 1 A). Fever developed, and treatment with ampicillin, gentamicin, and metronidazole was begun. Endoscopic retrograde
Although virtually every organ may be involved, candidiasis of the biliary tract is rare, even in large series of patients with disseminated infection [1]. In 1 0 of 1 2 reported cases of biliary candidiasis ([1 -3] and this case), the infection was not systemic. In 1 1 of the 1 2 patients, one or more of these predisposing factors was present: previous cholecystectomy or other major operation, treatment with broad-spectrum antibiotics, diabetes mellitus, and acute leukemia. None of the patients had AIDS. No apparent predisposing factor was present in a 3V2-year-old girl who had a fungus ball in the common bile duct 2 months after blunt abdominal trauma [2].
cholangiography
mural irregularity,
a diffusely abnormal biliary tree with marked multiple strictures, and segments of mild dilatation.
showed
Apparent dissection bile duct was seen. gitis
and
primary
of contrast Differential sclerosing
material diagnosis cholangitis.
in the wall of the common included infectious cholanSphincterotomy
was
per-
Received August 15, 1991 ; accepted after revision September 23, 1991. 1 Both authors: Department of Radiology, University of Southern California Center, 1200 N. State St., Los Angeles, CA 90033-1 084. AJR 158:1029-1030,
May
1992 0361-8o3x/92/1585-1029
C American
School
Roentgen
of Medicine,
Ray Society
Los Angeles
County-University
of Southern
California
Medical
RADIN
1030
AND
JOHNSON
AJR:158,
May 1992
Downloaded from www.ajronline.org by 117.253.210.40 on 11/10/15 from IP address 117.253.210.40. Copyright ARRS. For personal use only; all rights reserved
Fig. 1.-A, Transverse sonogram shows thickened hypoechoic wall of common duct (arrows) and nonshadowing echogenic material filling lumen. Arrowhead = hepatic artery, P = portal vein, C = inferior vena cava, A = aorta, S = spine. B, Endoscopic retrograde cholangiogram
shows
Involvement
marked
mural
tree by strictures and segments of mild dilatation. Channels of contrast material parallel to common duct may represent
In one previously reported case [2], sonography showed a nonshadowing polypoid mass of 7-8 mm in the lumen of the distal common bile duct. Pathologic examination of the contents of the common duct showed Candida embedded in bile sludge material. Sonographic findings in our patient differed in two respects. Thickening of the wall of the common duct reflected the invasive nature of the infection. The echogenic material filling the lumen of the entire common duct probably represented a combination of intraluminal debris and ulcerated mucosa. Cholangiography in four reported cases of biliary candidiasis [1 4] showed one or more intraluminal filling defects representing fungus balls in the common bile duct. In one of these cases [4], several strictures of the intrahepatic and extrahepatic bile ducts also were present. Cholangiographic findings in our patient were far more severe and extensive, consistent with diffuse invasive candidiasis. The radiologic appearance was suggestive of primary sclerosing cholangitis [5] or AIDS-related cholangitis [6]. Candidiasis was not suspected until biopsy was performed. Although rare, the diagnosis of biliary candidiasis should be considered when radiologic findings show cholangitis or an ,
of entire
irregularity
intramural
biliary
with multiple
dissection.
intraluminal filling defect in the common bile duct in a patient with predisposing factors for Candida infection. Bile should be obtained for fungal stain and culture. With appropriate treatment, the majority of patients will make a complete recovery [1].
REFERENCES 1 . Irani M, Truong
2.
LD. Candidiasis
of the extrahepatic
Pathol Lab Med 1986;1 10: 1087-1 090 Carstensen H, Nilsson KO, Nettelblad SV, Cederlund
biliary tract. Arch CG, Hildell
J. Corn-
mon bile duct obstruction
due to an intraluminal mass of candidiasis in a previously healthy child. Pediatrics 1986;77:858-861 3. Chen CC, Chang PY, Chen CL. Refractory cholangitis after Kasai’s operation caused by candidiasis: a case report. J Pediatr Surg 1986;21: 736-737
4. Uflacker A, Wholey MH, Amaral NM, Lima S. Parasitic and mycotic causes of biliary obstruction. Gastrointest Radiol 1982;7: 173-1 79 5. MacCarty AL, LaRusso NF, Wiesner RH, Ludwig J. Primary sclerosing cholangitis: findings 1983;149:39-44
6. Dolmatch
on cholangiography
and pancreatography.
Radiology
BL, Laing FC, Federle MP, Jeffrey RB, Cello J. AIDS-related
cholangitis: radiographic 1987;163:313-316
findings
in
nine
patients.
Radiology
