Liver International ISSN 1478-3223

CLINICAL STUDIES

Hepatobiliary cystadenomas and cystadenocarcinomas:

a report of 33 cases
Xinting Sang1, Yongliang Sun2, Yilei Mao1, Zhiying Yang2, Xin Lu1, Huayu Yang1, Haifeng Xu1, Shouxian Zhong1
and Jiefu Huang1
1 Department of Liver Surgery, Peking Union Medical College (PUMC) Hospital, Chinese Academy of Medical Sciences & PUMC, Beijing, China
2 Department of Hepatobiliary Surgery, China–Japan Friendship Hospital, Beijing, China

Keywords Abstract
CA19-9 – diagnosis – hepatobiliary Background: Hepatobiliary cystadenomas and cystadenocarcinomas are rare
cystadenocarcinoma – hepatobiliary and often misdiagnosed. Aims: We report our experience with 33 cases over
cystadenoma – treatment 20 years to discuss an algorithm for these diseases. Methods: Patients present-
ing with a diagnosis of hepatobiliary cystadenomas and cystadenocarcinomas
were retrospectively reviewed from January 1991 to October 2010. Clinical
Correspondence
data were collected by examining hospital records and by follow-up ques-
Yilei Mao, MD, PhD, Department of Liver
Surgery, Peking Union Medical College
tionnaire interviews. Results: Thirty-three patients had pathologically diag-
Hospital, Chinese Academy of Medical Sciences
nosed hepatobiliary cystadenomas (19/33, 17 females and two males) or
& PUMC, Shuai-Fu-Yuan 1, Dongcheng District, cystadenocarcinomas (14/33, five females and nine males). Symptoms of
Beijing 100730, China cystadenomas at hospitalization were abdominal bloating or pain (9/19). Nine
Tel:186 10 65296042 patients had an elevated level of carbohydrate antigen (CA) 19-9. The surgical
Fax:186 10 65296043 procedures, i.e. cyst enucleation, segmentectomy, sectionectomy and hemi-
e-mail: sunyongliang1982@hotmail.com; hepatectomy, were performed with satisfactory outcomes. Symptoms of
maoy@public3.bta.net.cn cystadenocarcinomas included abdominal bloating or pain (8/14) and fever
(3/14). Seven patients had elevated CA19-9. The imaging characteristics of
Received 28 March 2011 cystadenocarcinomas were similar to those of cystadenomas. The clinical
Accepted 17 May 2011 outcomes for cystadenocarcinomas were mostly poor after either surgical or
conservative treatment. Conclusions: Clinical symptoms are unreliable for
DOI:10.1111/j.1478-3231.2011.02560.x these diagnoses and their differential diagnosis. Imaging evaluations and
CA19-9 are of value for the recognition of cystadenoma and cystadenocarci-
noma, but not for their differential diagnosis. Any recurrence of liver cyst after
surgery or other treatments should lead one to suspect one of these diseases.
Invasive examination and percutaneous fine-needle aspiration cytology are
not recommended. Complete excision or careful enucleation should be the
first treatment choice for a better prognosis.

Hepatobiliary cystadenomas and cystadenocarcinomas
are rare neoplasms, with the first case having been
reported in 1892 by Keen (1). They account for 1% of
liver cystic lesions and approximately 5% of symptomatic
hepatic cysts (2–4). Because of their rarity and presenta-
tions similar to those of other cystic liver lesions, the
diagnosis is often delayed; this results in inappropriate
treatment modalities, such as aspiration, causing unne-
cessary morbidity and mortality (5, 6). A second-stage
operation is sometimes needed because of the misdiag-
nosis of the disease. Reports of these diseases have been
increasing because of advances in imaging techniques
such as computed tomography (CT) and magnetic
resonance imaging (MRI) in recent years. Here, we
present our experience with 19 hepatobiliary cystadeno-
mas and 14 cystadenocarcinomas over the last 20 years in
Contributed equally.
a leading teaching hospital in China, offer a pertinent
review of the literature and discuss current treatment
modalities. This series is one of the largest clinical reports
in the published literature.
Methods
This study was approved by the Ethics Committee of the
Peking Union Medical College (PUMC) Hospital. The
hospital records were searched for patients with diag-
noses of hepatobiliary cystadenoma, cystadenocarcino-
ma and other hepatic cysts according to the International
Classification of Diseases (Ninth Revision). Once identi-
fied from the hospital records, the pathological speci-
mens from operative resection were retrieved and
reviewed by two specialists in gastrointestinal pathology
who were blinded to the original diagnoses; no final
pathological diagnoses were changed after reviewing.

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Hepatobiliary cystadenomas and cystadenocarcinomas Sang et al.

Patient records were examined for the details of demo- Table 1. Patient characteristics
graphical characteristics, medical history, symptoms, Cystadenoma
pre-operative examination, treatments including details subgroup Cystadenocarcinoma
of surgeries and patient outcomes. (n = 19) subgroup (n = 14)
Follow-up data were obtained by reviewing the hospi- Age 44.2  14.4 57.0  14.0
tal records and filling out a questionnaire via telephone Gender (M/F) 2/17 9/5
interviews. The follow-up period was defined from the Length of symptoms 18.1  25.6 29.4  43.8
date of the operation to that of the patient’s death or the (months)
last follow-up point. Deaths from other causes were HBV (  ) 1/18 0/14
treated as censored cases. The length of hospitalization Serum CA19-9 (U/ml) 838.4  2485.7 337.9  723.3
(LOH) was defined as the time from admission to Tumour size (cm) 13.0  8.1 8.3  2.9
discharge of the patients. Definitive surgery
Cyst enucleation 3 0
Fenestration 1 1
Surgery Segmentectomy 5 1
The procedure terminology of the liver resections per- Sectionectomy 5 2
Hemihepatectomy 5 4
formed in this series was standardized according the
Trilobectomy 0 1
Brisbane Terminology (7). All surgeries were performed Operating time (hours) 3.9  1.7 4.2  1.9
following a standardized operation method. Taking left Volume of blood 514.2  621.1 556.8  592.5
hemihepatectomy as an example, after dissecting the porta loss (ml)
hepatis, the left hepatic artery and left portal vein branches TACE 0 2
were transected. The left biliary duct was also transected Length of hospitalization 18.4  9.4 23.1  15.0
only if it could be clearly dissected in the hilar plate. The (days)
second porta hepatis of hepatic veins was then fully Follow-up time (months) 22.5 (0.4–79.5) 5.5 (0.2–37.7)
dissected and exposed. The left hepatic vein was ligated
F, female; HBV, hepatitis B virus; TACE, transcatheter arterial chemoem-
and amputated if the left hepatic vein and middle hepatic bolization; M, male.
vein were not in the same outflow; an electrocoagulator was
used to divide the liver tissue within a depth of 0.7 cm from two males. Symptoms upon admission had been present
the liver surface, and then a Cavitron ultrasonic surgical for 18.1  25.6 months (range, 3 days to 7 years). Nine
aspirator (CUSA, Soering GmbH Medizintechnik, Justus- had abdominal bloating or pain (range, 2 weeks to 7
von-Liebig-Ring, Quickborn, Germany) was applied after months); among them, one had a palpable abdominal
the coagulator to further dissect the liver tissue. All the mass for 3 weeks, one showed intermittent nausea/
branches of blood vessels and bile ducts in the cutting edge vomiting for 6 months and jaundice for 1 month, and
were carefully explored and ligated or clipped. The middle one was accompanied by aggravated jaundice for 2
hepatic vein was protected, and the small branches were weeks. Ten patients were asymptomatic. LOH was
closed with fine Prolene sutures. The cutting edges were 18.4  9.4 days (range, 7–49 days) (Table 1).
either left open or closed with fine sutures according to the Pre-operative evaluations, such as laboratory studies
actual conditions. Sometimes, a drainage tube was not including complete blood count and serum chemistries,
needed if the cutting edge was treated satisfactorily. were unremarkable in all patients; hepatitis virus markers
were negative in all but one patient, who was positive for
Results the hepatitis B virus. The tumour marker carbohydrate
From January 1991 to October 2010, physicians at our antigen (CA) 19-9 was tested in 11 patients; among
institution identified 33 patients with a diagnosis of them, nine (81.8%) had levels higher than those in the
hepatobiliary cystadenomas or cystadenocarcinomas. normal range (0–37 U/ml), with an average value of
The cases were divided into a cystadenoma subgroup 838.4  2485.7 U/ml (range, 0–10 000 U/ml). Other
and a cystadenocarcinoma subgroup. The 19 hepatobili- tumour markers, including carcinoembryonic antigen
ary cystadenoma patients were hospitalized from April (CEA), a-fetoprotein (AFP), CA153 and CA242, were
2002 to October 2010, while the 14 cystadenocarcinoma unremarkable.
patients were hospitalized from October 1991 to Septem- Different abdominal imaging studies were performed
ber 2009 (Table 1). The serum levels of CA19-9 in 36 before definitive treatment. Ultrasound in 16 and CT in
patients with simple hepatic cysts were also measured. 14 patients were the most commonly used imaging
They were all within the normal range, with an average modalities; MRI was performed in six patients. The
value of 11.4  8.7 U/ml (range, 3.2–20.9 U/ml). common manifestations on ultrasound included multi-
locular or unilocular hypoechoic cysts with thickened,
irregular walls; numerous hyperechoic intracystic straps;
Cystadenoma subgroup
and thin internal septations with high echogenicity
The 19 patients were 44.2  14.4 years of age at the time within the cystic masses. Similarly, CT findings revealed
of presentation (range, 19–67 years), with 17 females and low-density, well-defined, lobulated, multilocular, thick-

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Sang et al. Hepatobiliary cystadenomas and cystadenocarcinomas

Fig. 1. (A) Convex papillate on the cyst wall of a well-defined, unilocular cystic mass on computed tomography (CT) scan; the convex papillate
can be enhanced with an intravenous intensifier. The final pathological diagnosis was cystadenoma. (B) Low-density, well-defined,
unilobulated, thick-walled cystic mass with internal septa on CT scan; the internal septa and wall could be enhanced with an intravenous
intensifier. The final pathological diagnosis was cystadenoma. (C) Exogenous, unilocular cystic mass with convex papillate on the thickened
wall on CT scan. The characteristics on CT scan were mostly similar to those of cystadenomas, but the final pathological diagnosis was
cystadenocarcinoma. (D) Multiloculated cystic mass located in segment VI with an irregular, thickened wall, convex papillate and internal septa;
coarse calcifications could be seen on the internal septa. The final pathological diagnosis was cystadenocarcinoma, as we suspected
pre-operatively.

walled cystic masses with internal septae and convex
papillae. The internal septae and walls were enhanced
after the administration of an intravenous contrast (Figs
1A and B). On MRI examination, a fluid-containing,
multilocular, septated mass with a homogenous low-
signal intensity was present on T1-weighted images, the
wall and septa of which became enhanced after the
administration of gadolinium–DTPA.
The pre-operative work-up included liver-protective
and nutritional support treatments. The liver-protective
medications of Polyene phosphatidylcholine and/or ade-
metionine 1,4-butanedisulphonate were used 3 days
before surgery in the patients with Child–Pugh scores
Z7; no patient in our series had cirrhosis or Child–Pugh
scores 4 9. Nutritional supportive treatment was im-
plemented 3 days before surgery at 83.7 kj/kg/day of
energy in patients with nutritional risks after a risk
assessment or those older than 65 years.
Five patients had previous surgical procedures that
resulted in incomplete surgery or recurrences, including
percutaneous aspiration in two, laparoscopic fenestra-
tion of the cyst in two, one attempted fenestration twice
and percutaneous aspiration four times.
For definitive surgeries, cyst enucleation was per-
formed in three, left segmentectomy in two, left biseg-
mentectomy in two, left lateral sectionectomy in two, left
medial sectionectomy in one, left hemihepatectomy in
four, right segmentectomy in one, right anterior sectio-
nectomy in one, right posterior sectionectomy in one,
right hemihepatectomy in one (Fig. 2) and laparoscopic
fenestration in one. Tumours averaged 13.0  8.1 cm
(range, 2–32 cm). The cystic fluid was clear, faint yellow
or brown, or jelly-like. The cystic fluid CA19-9 levels
were tested in three patients; they were 83.42, 5000 and
4 10 000 U/ml respectively. The operative duration was
3.9  1.7 h (range, 1.75–9 h). Blood loss during surgery
was 514.2  621.1 ml (range, 20–2500 ml). Ten patients
required no transfusion during the operative procedure
or subsequent hospital stay. Of the nine patients who
required transfusion, the mean requirement was
6.1  3.8 units of packed red blood cells and
866.7  722.9 ml of plasma. Regretfully, only four pa-
tients underwent frozen-section biopsy intra-operatively;
three of them were proven hepatobiliary cystadenomas,
while the other patient was diagnosed with a simple
hepatic cyst. All of the final pathological diagnoses were
hepatobiliary cystadenomas.
Follow-up information was available for all 19 pa-
tients. The follow-up time ranged from 0.4 to 79.5
months, with a median follow-up time of 22.5 months.
Two patients were treated by percutaneous drainage
because of post-operative biloma. One patient had a

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Hepatobiliary cystadenomas and cystadenocarcinomas Sang et al.

Fig. 2. Procedures of the operation of a patient with right hepatic cystadenoma who underwent right hemihepatectomy. (A) Pre-operative
computed tomography (CT) scan that showed a low-density, well-defined, unilobulated, thick-walled cystic mass with internal septa; the
internal septa and wall could be enhanced with an intravenous intensifier. (B) Overall appearance of the cystadenoma after laparotomy.
(C) Dissection of the portal triad. (D) Dissection of the right hepatic vein in the second porta hepatis. (E) Hepatic cut surface after right
hemihepatectomy. (F) Macroscopic appearance of the cystadenoma after surgery; the final pathological diagnosis was cystadenoma.

laparoscopic fenestration, and the intra-operative frozen-
section biopsy revealed a simple hepatic cyst; however,
she refused to undergo a second resection after the final
pathological diagnosis of hepatobiliary cystadenoma and
showed no recurrence after 14.4 months of follow-up.
Eight months after left lobectomy, one patient died of
widespread metastasis of a malignant tumour despite an
original pathological diagnosis of hepatobiliary cystade-
noma combined with moderate dysplasia. The other 17
patients showed no recurrence or clinical symptoms
upon our last follow-up (Fig. 3).
Cystadenocarcinoma subgroup
The 14 patients were 57.0  14.0 years of age at the time
of presentation (range, 26–73 years), with five females
and nine males. Symptoms upon admission had been
present for 29.4  43.8 months (range, 20 days to 10
years). Eight had abdominal bloating or pain (range,
2 months to 10 years), and among them, three were
accompanied by fever (20 days, 9 months and 12 months
respectively). Three patients were asymptomatic. LOH
was 23.1  15.0 days (range, 10–69 days) (Table 1).
The pre-operative blood evaluations were unremark-
able, as in the cystadenoma subgroup; hepatitis virus
markers were negative in all patients. Tumour marker
CA19-9 levels were tested in 11 patients. Seven (63.6%)
had values higher than those of the normal range, and
the average value was 337.9  723.3 U/ml (range,
4.8–2461 U/ml). Other tumour markers, such as AFP,
CA153 and CA242, were also unremarkable.
Abdominal imaging evaluations included ultrasound
in 10, CT in 10, positron emission tomography (PET) in
one, nuclear medicine uptake study in one and angio-
graphy in two. The characteristics of the cystadenocarci-
nomas on imaging evaluations were mostly similar to
those of cystadenomas (Fig. 1C). Rarely, capsular, mural
or septal coarse calcifications could be seen on

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Sang et al.
Fig. 3. (A) Right posterior branch of the portal vein after cyst
enucleation to preserve the important vessels. (B) Gross internal
appearance of the irregular, thickened wall of a cystadenoma after
left hemihepatectomy.
ultrasound or CT in cystadenocarcinomas (Fig. 1D).
Imaging differentiation criteria between hepatobiliary
cystadenoma and cystadenocarcinoma were not estab-
lished. The PET indicated a hepatic malignant tumour in
the left liver, and the patient underwent left hemihepa-
tectomy plus cholangiojejunostomy using a Roux-en-Y
limb. Both angiographies showed abnormal hepatic
malignant tumour staining. No special characteristics
were identified by the nuclear medicine uptake study.
Pre-operative work-ups were similar to those in the
cystadenoma subgroup.
Six patients received previous inappropriate treat-
ments that resulted in incomplete surgery or accompany-
ing discomfort, including percutaneous aspiration in
two, cyst enucleation in one and open fenestration plus
complete fulguration in one; their previous final patho-
logical diagnoses in other hospitals were cystadenomas.
One underwent attempted drainage using a Roux-en-Y
limb, but the final pathological diagnosis for that surgery
was a simple hepatic cyst. One underwent hepatobiliary
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c 2011 John Wiley & Sons A/S
Hepatobiliary cystadenomas and cystadenocarcinomas
cystadenoma partial resection for cyst recurrence after
percutaneous aspiration, and the patient received percu-
taneous transhepatic catheter drainage for jaundice 3
months later.
As for the 14 cystadenocarcinoma patients, three were
discharged because the tumours were evaluated as un-
resectable and the patients refused further treatments,
while two late-stage patients received transcatheter arter-
ial chemoembolization (TACE). The nine remaining
patients received surgical treatments, including left bi-
segmentectomy in one, left lateral sectionectomy in one,
left hemihepatectomy in three, left hemihepatectomy
plus cholangiojejunostomy in one, left medial sectionect-
omy plus right anterior sectionectomy in one and open
fenestration in one; the patient on who attempted
drainage using a Roux-en-Y limb was performed pre-
viously received right trilobectomy for abdominal bloat-
ing and pain. One patient who experienced epigastric
and hepatic tumour recurrence underwent tumour re-
section 8 months after left bisegmentectomy. Tumours
averaged 8.3  2.9 cm (range, 3.5–13 cm). The operative
duration was 4.2  1.9 h (range, 2–7 hours). Blood loss
during surgery was 556.8  592.5 ml (range, 50–1500 ml).
Four patients required no transfusion during surgeries or
subsequent hospital stay. In the other four patients
who required transfusion, the mean requirement was
6.0  1.8 units of packed red blood cells and
600  200 ml of plasma. All six intra-operative frozen-
section biopsies were hepatobiliary cystadenocarcino-
mas. All nine patients’ final pathological diagnoses were
hepatobiliary cystadenocarcinomas.
Of the six cystadenocarcinoma patients who had been
followed up, four underwent surgery, one was treated
with TACE and one refused to accept any treatment.
Among them, three died, one had recurrence four
months after right trilobectomy, the late-stage patient
who refused any treatment was still alive and only one
operated patient was still alive and tumour-free (14.4
months after the operation). The follow-up time ranged
from 0.2 to 37.7 months, with a median follow-up time
of 5.5 months. The details of follow-up information are
shown below. One male patient who underwent left
hemihepatectomy died of cerebral haemorrhage and
ventricular fibrillation 6 days post-operatively, during
hospitalization. One late-stage male patient died 4
months after TACE of widespread metastasis. The female
patient who experienced epigastric and hepatic tumour
recurrence and underwent tumour resection 8 months
after left bisegmentectomy died of widespread metastasis
10 months later. One late-stage male patient who did not
undergo surgery, TACE or any other treatment showed
distention and vomiting, but was still alive 13.2 months
after discharge from our hospital. One female patient
who underwent right trilobectomy showed jaundice 4
months after the surgery; abdominal MRI indicated local
recurrence, but the patient refused any treatment. One
male patient who underwent left hemihepatectomy
showed no recurrence after 14.4 months of follow-up.
1341
Hepatobiliary cystadenomas and cystadenocarcinomas
Discussion
Hepatobiliary cystadenomas and cystadenocarcinomas
may occur at any age. Approximately 85–95% of those
affected are women (8, 9). However, our cystadenocarci-
noma subgroup did not show the same female tendency
(9/14 cystadenocarcinomas were in males), as did other
reports. The cases reported so far may be so limited that
the gender tendency could not be clarified. The appear-
ance of the initial manifestations of the lesions may take
years or the peak frequency was in the fifth decade of life
(10, 11). The ages at presentation in our cystadenocarci-
noma subgroup were older than those of our cystadeno-
ma subgroup (57.0  14.0 years vs. 44.2  14.4 years
respectively; P o 0.05). This phenomenon further sup-
ports the hypothesis of malignant transformation of
cystadenomas to cystadenocarcinomas.
Most of our cases were identified over the past 10
years, especially in the past 5 years. This may be because
of the advances and commonly applied tools in favour
of detection, as well as pathological awareness of the
diseases.
The exact aetiology of hepatobiliary cystadenomas
remains unknown. Both congenital and acquired causes
have been proposed. It is uncertain whether biliary
cystadenocarcinomas are de novo cancers or are derived
from cystadenomas. They are generally thought, how-
ever, to arise from pre-existing benign cystadenomas
because benign epithelium has been demonstrated in
over 90% of cystadenocarcinomas. Many cystadenocar-
cinomas contain areas of cystadenoma in the same
sample.
The most typical clinical manifestations in the litera-
ture are abdominal discomfort or pain, which were
present in 51.5% (17/33) of our patients. Asymptomatic
patients comprised 39.4% (13/33) in our series. Other
non-specific manifestations such as jaundice and fever
may be associated with biliary obstruction, rupture,
bacterial infection or intracystic haemorrhage (12, 13).
Thus, clinical manifestations are considered unreliable
for the diagnosis. We believe that any patient presenting
with recurrence of their liver cyst after surgery or other
treatments should be suspected of having a cystadenoma
or cystadenocarcinoma. Significantly increased levels of
serum CA19-9 can be encountered in cystadenomas and
cystadenocarcinomas (10, 14, 15). This was also true in
our series, with nine of 11 patients with cystadenomas
having increased CA19-9 values (838.4  2485.7 U/ml)
and seven of 11 patients with cystadenocarcinomas
having increased CA19-9 values (337.9  723.3 U/ml).
Other tumour markers, such as CEA, AFP, CA153 and
CA242, were unremarkable in both subgroups. We
compared serum CA19-9 in simple cysts, cystadenomas
and cystadenocarcinomas subgroups in order to evaluate
the role of CA19-9 in differentiating diagnosis (Table 2).
Based on the data from the comparisons, we conclude
that the measurement of serum CA19-9 could thus be
helpful in the differential diagnosis of simple hepatic
1342
Sang et al.
Table 2. Serum CA19-9 in patients with simple hepatic cyst, carbo-
hydrate antigen and cystadenocarcinoma
Cystadeno-
Cystadenoma carcinoma
subgroup subgroup
Serum (n = 11) (n = 11) P-value
CA19-9 838.4  2485.7 337.9  723.3 0.735
Simple hepatic 11.4  8.7
cyst (n = 36)
P-value 0.0001 0.001
cystic lesions, but not between cystadenoma and cysta-
denocarcinoma, or other cancers originating from the
biliary tract. Whereas the level of CA19-9 in the cystic
fluid may be invaluable in differential diagnoses among
liver cystic lesions, it may also be significantly elevated in
the fluid of a simple hepatic cyst in our experience and in
the published literature (16–18).
In addition, CT is less sensitive than ultrasound in
visualizing intracystic blood clots for differential diag-
nosis. Although the imaging characteristics of hepato-
biliary cystadenoma and cystadenocarcinoma have been
recognized as we described in our patients, the differen-
tial diagnosis between them on the basis of imaging alone
has appeared to be difficult (17, 19, 20, 21). CT or MRI
can delineate the anatomic relations within the liver that
may be helpful in planning the surgical procedure (22).
Intra-operative frozen sections may be more helpful than
imaging characteristics in the differentiation of cystade-
nomas and cystadenocarcinomas from other cystic hepa-
tic lesions. Careful histopathological evaluation of the
resected specimen is the only safe diagnostic modality,
while malignant degeneration or transformation of cy-
stadenomas can only be detected after thorough section-
ing (23, 24). Histologically, both cystadenomas and
cystadenocarcinomas have been divided into two sub-
groups according to the presence of mesenchymal (ovar-
ian-like) stroma between the inner epithelial lining and
the outer connective tissue capsule (2, 11, 23). We also
consider that percutaneous fine-needle aspiration cytol-
ogy should not be generally recommended in cases of
overt or suspected cystadenocarcinoma, particularly
when surgery is planned. The characteristic of the fluid
content of the lesion is not meaningful to diagnosis in
our experience. Clear, haemorrhagical, faint yellow,
bilious and mixed fluid contents were observed in our
series.
Because distinguishing between cystadenoma and
cystadenocarcinoma pre-operatively and determining its
malignant potential are difficult, we suggest that total
excision is ideal, may offer the best chances of cure and
might be the only definitive therapy. Surgery also allows a
proper histological examination of the cyst and a detailed
classification. Once cystadenoma or cystadenocarcinoma
is diagnosed regardless of what the results are from the
intra-operative frozen section or/and the final pathology,
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Sang et al.
a radical resection is strongly recommended for the
patients. Because cystadenoma holds a risk of malignant
degeneration, and also part of the cystadenoma tissue
may have already been malignant transformed, that was
not found in frozen sections. However, the general
condition of the individual patient, anatomic position
of the cysts and experience of the surgeon should
determine the final therapeutic decision (10, 11, 18, 25,
26). Enucleations were performed when the lesions were
large sized and/or the blood vessels or bile ducts were
invaded. It may be difficult to separate from the wall of
cystadenoma, especially after earlier procedures or infec-
tion; the enucleations must be performed cautiously and
can offer a satisfactory outcome. Partial excision should
only be reserved for those cases in which total excision
might jeopardize the main liver structures. Partial resec-
tion with occasional ablation of the residual cyst using
electrocautery or argon beam coagulation have been
reported (10). However, we do not recommend these
procedures because partial resection may not confer a
prognostic benefit for the patients at all while having the
potential to increase the complication rate.
Our follow-up data showed that 18 of 19 cystadeno-
mas had no recurrence after an average of 22.5 months of
follow-up. The follow-up number and duration in our
cystadenocarcinoma subgroup was not sufficient for
further analysis or a firm conclusion. Although the post-
operative recurrent rate for cystadenocarcinomas is high,
and with poor prognosis, we advocate that a surgical
process with complete resection for the lesion is recom-
mended, as long as the condition of patients allows them
to tolerate the surgery.
In conclusion, with the advantage of better imaging
technologies, the recognition and detection rate of hepa-
tobiliary cystadenoma and cystadenocarcinoma have
increased in recent years. Any recurrence of a liver cyst
after surgery or other treatments should lead one to
suspect one of these diseases. The CA19-9 is a valuable
marker in the differential diagnosis of simple hepatic
cysts. We believe that invasive examinations should be
avoided and that percutaneous fine-needle aspiration
cytology should not be recommended. Complete exci-
sion or careful enucleation should be the first treatment
of choice to ensure a better prognosis, and partial
excision should only be reserved for those cases in which
total excision might jeopardize the main liver structures.
Acknowledgements
This work was supported by the China Medical Board in
New York (CMB, 06-837).
Yongliang Sun, Xinting Sang and Yilei Mao per-
formed research and wrote the paper; Huayu Yang helped
writing the paper; Zhiying Yang, Xin Lu, Haifeng Xu,
Shouxian Zhong and Jiefu Huang provided critical com-
ments and intellectual input.
Conflict of interest: Nothing to report.
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c 2011 John Wiley & Sons A/S
Hepatobiliary cystadenomas and cystadenocarcinomas
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