AJR 2003; 181:464-466
© American Roentgen Ray Society
Late Complication of a Large Simple Cyst of the Liver Mimicking Cystadenocarcinoma After Sclerotherapy
Kenichi Takayasu1,
Yasunori Mizuguchi1,
Yukio Muramatsu1,
Susumu Yamasaki2,
Teiichi Sugiura2,
Chise Sato1 and
Michiie Sakamoto3
1 Department of Diagnostic Radiology, National Cancer Center Hospital, 5-1-1,
Tsukiji, Chuo-ku, Tokyo, 104-0045 Japan.
2 Department of Hepatobiliary Surgery, National Cancer Center Hospital, Tokyo
104-0045, Japan.
3 Pathology Division, National Cancer Center Research Institute, Tokyo 104-0045,
Japan.
Received October 2, 2002;
accepted after revision January 2, 2003.
Address correspondence to K. Takayasu.
Introduction
For nonparasitic or nonneoplastic cysts of the liver in a symptomatic
patient, sclerotherapy with absolute ethanol has been widely performed as a
minimally invasive treatment
[1,
2]. Most reported cysts were
successfully reduced with no major complication. In one patient, however,
cystadenocarcinoma developed after sclerotherapy with ethanol
[3]. We report a patient in
whom a mural nodule emerged—and then gradually increased in
size—in a cyst that had been reduced with minocycline hydrochloride and
absolute ethanol. Contrast enhancement of the nodule 5 years after
sclerotherapy suggested the development of biliary cystadenocarcinoma and led
to hepatic resection.
Case Report
A 72-year-old woman who presented at the outpatient department with a large
hepatic cyst measuring 21 x 19 cm in diameter in the right lobe had been
followed up for 5 years. Because the patient complained of abdominal fullness
after meals, sclerotherapy was performed three times, with two kinds of
sclerosants, under sonographic guidance. On two occasions, 1000 mg of
minocycline hydrochloride was administered because of the patient's
hypersensitivity to ethanol; 3 days later, 105 mL of absolute ethanol was
administered because the minocycline hydrochloride had proven ineffective.
Cytology of the removed fluid was negative for cancer cells. Finally, 10
months later, the cyst was reduced to 5 x 2.5 cm in diameter.
Five years after the third sclerotherapy, sonography revealed a
triangle-shaped mural nodule measuring 2 x 2 cm in diameter in the
residual cyst (Fig. 1A). Even
though CT showed the mural nodule, it was not enhanced by iopamidol
(Iopamiron, 300 mg I/mL, Schering, Tokyo, Japan). Repeated sonography showed
that the mural nodule grew and filled the cystic cavity completely. The
patient had no tendency to bleed, nor was she receiving anticoagulants.
Hepatic arteriography alone and a combination of CT and hepatic arteriography
failed to show enhancement of the mural nodule. At a 21-gauge needle biopsy,
only a blood clot was aspirated. Dynamic CT performed 5 months later disclosed
that fine neovasculature had developed in the nodule
(Fig. 1B).
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Fig. 1A. —72-year-old woman complaining of abdominal fullness and having
unilocular 21 x 19 cm hepatic cyst. Subcostal sonogram reveals echogenic
mass (straight arrow) measuring 2 x 2 cm emerging in residual
cyst measuring 5 x 2.5 cm. Strong echo (arrowheads), suggesting
calcification of cyst wall, and middle hepatic vein (curved arrow)
are seen.
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Fig. 1B. —72-year-old woman complaining of abdominal fullness and having
unilocular 21 x 19 cm hepatic cyst. Dynamic axial CT scan obtained
during arterial phase shows two stellar-shaped enhancements in medial
component (arrow). Calcifications of cyst wall laterally and
associated cyst (arrowhead) are seen.
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T1-weighted MR imaging performed with a superconducting system at 1.5 T
(MRT 200FX, Toshiba, Tokyo, Japan) 4 months after dynamic CT showed a 5
x 4 cm round mass just behind an associated small cyst. The mass
consisted of a round hypointense area medially and a crescent-shaped
hyperintense region laterally (Fig.
1C). T2-weighted MR imaging revealed that the medial area
comprised a central hyperintense portion and a peripheral hypointense one, and
the lateral area consisted of a crescent-shaped hyperintense portion
(Fig. 1D). Dynamic MR imaging
with gadopentetate dimeglumine showed the central region of the medial area to
be enhanced (Fig. 1E), which
was coincident with the fine neovasculature recognized on dynamic CT
(Fig. 1B).
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Fig. 1C. —72-year-old woman complaining of abdominal fullness and having
unilocular 21 x 19 cm hepatic cyst. Axial T1-weighted fast spin-echo MR
image (TR/TE, 600/12) reveals hypointense area (arrow) in medial
portion of lesion and crescent-shaped hyperintense region in lateral
portion.
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Fig. 1D. —72-year-old woman complaining of abdominal fullness and having
unilocular 21 x 19 cm hepatic cyst. Axial T2-weighted fast spin-echo MR
image (3000/120) shows hypointense area with central hyperintensity
(arrow) in medial and crescent-shaped lateral portions.
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Fig. 1E. —72-year-old woman complaining of abdominal fullness and having
unilocular 21 x 19 cm hepatic cyst. Gadolinium-enhanced T1-weighted
image (143/5; 60° flip angle) reveals two nodular areas of enhancement
(arrows) surrounded by hypointense area in medial portion of
lesion.
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Finally, cystadenocarcinoma derived from a simple cyst treated by
sclerotherapy was suspected, and surgery was performed. The right lobe of the
liver was markedly atrophied, and the left lobe was compensatorily
hypertrophic. The cyst was strikingly firm and smaller than it had been, and
it seemed to be hanging from the hypertrophic left lobe. The lesion was
resected with a small portion of the attached left medial segment.
On the cut surface of the resected specimen, the tumor was surrounded by a
firm capsule and contained dark-red and dark-brown materials in the medial and
lateral portions, respectively. Histopathologic study showed that the lesion
was surrounded by hyalinized fibrous tissue with calcification and consisted
of a mixture of old organized hemorrhage (medially) and relatively fresh
hemorrhage (laterally). In the organized portion, which consisted of
hyalinized fibrous tissue and old hemorrhage, abundant clefts and a bizarrely
shaped dilated vessellike structure with exuding blood covered by endothelial
lining cells was recognized (Fig.
1F). No cancer cells were found within or outside the lesion. The
small associated cyst consisted of a thin cuboidal epithelium layer,
consistent with a simple cyst.
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Fig. 1F. —72-year-old woman complaining of abdominal fullness and having
unilocular 21 x 19 cm hepatic cyst. Photomicrograph of histologic
specimen shows multiple bizarrely shaped vessels (arrows) with RBCs
covered by endothelial cell lining in hematoma. (H and E, x 40)
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Three years after surgery, the patient is doing well with no sign of
malignant tumor.
Discussion
In the past, resection was the treatment of choice for a large simple cyst
of the liver, especially a symptomatic one. Now, however, sclerotherapy is
preferred because of its minimum invasiveness and low cost
[1,
2]. For the treatment, absolute
ethanol is preferred over other sclerosants, such as minocycline hydrochloride
[4] and iophendylate, because
it is highly effective in a single session
[2]. Intracystic hemorrhage can
occur after sclerotherapy [1,
2], and it can be
spontaneous.
Dohchin et al. [3] reported
the development of cystadenocarcinoma in a residual cyst 9 months after
sclerotherapy as an untoward event. However, the authors speculated that the
cystadenocarcinoma had been present in the cyst at the time of
treatment—despite negative cytology results for aspirated
fluid—because carcinoma emerged shortly after the sclerotherapy.
Generally, cystadenocarcinoma develops from its counterpart, cystadenoma
[5]. But cystadenocarcinoma can
also arise from a simple cyst, although the incidence is low; papillary
adenocarcinoma has been reported to arise next to the columnar cuboidal
epithelium layer of the cyst
[6]. In our patient, the
development of cystadenocarcinoma from a simple cyst was strongly suspected
because the mural nodule gradually grew within the residual cyst and tumor
vessels eventually emerged, as shown by dynamic CT and MR imaging. These
imaging findings are consistent with cystadenocarcinoma
[7].
The intensity of the hemorrhagic cyst on MR imaging depends on the stage:
at an acute stage, the appearance of the cyst is hyperintense on T1-weighted
MR imaging and hypointense on T2-weighted MR imaging, gradually changing to
hyperintense on both T1- and T2-weighted MR imaging. When chronic, it is
hypointense on T2-weighted MR imaging. In our patient, the lateral portion of
the lesion was seen as a crescent-shaped hyperintense area on both T1- and
T2-weighted MR imaging (Figs.
1C and
1D), which was coincident with
fresh hemorrhage. The medial portion appeared hypointense on T1-weighted MR
imaging (Fig. 1C) and
hypointense with a central hyperintense area on T2-weighted imaging
(Fig. 1D). The hyperintense
area, enhanced on dynamic CT and MR imaging, histopathologically consisted of
abundant capillaries containing blood in an organized hematoma. No cancer
cells were found.
Neovasculature in the chronic thrombi in the left atrium or aortic aneurysm
has been reported [8]. In our
patient, a similar mechanism can be presumed to have occurred within the cyst
that was reduced by sclerotherapy. The bizarre or dilated capillaries in the
organized or organizing hematoma were presumed to be tumor vessels, which
corresponded to the enhanced nodules on dynamic CT and MR imaging.
The mural nodule observed with contrast enhancement on dynamic CT and MR
imaging developed in a simple hepatic cyst 5 years after sclerotherapy. Its
presence suggested cystadenocarcinoma, and therefore, hepatic resection was
performed. The pathologic diagnosis of chronic hematoma with bizarre-shaped
capillaries showed that an enhanced lesion on images is not always
malignant.
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