Imaging findings of bile duct hamartomas: a case report and

Int J Clin Exp Med 2015;8(8):13145-13153
www.ijcem.com /ISSN:1940-5901/IJCEM0012001
Original Article
Imaging findings of bile duct hamartomas: a case report
and literature review
Qiu-Sheng Shi1*, Ling-Xi Xing1*, Li-Fang Jin1, Han Wang2, Xiu-Hong Lv3, Lian-Fang Du1
Department of Ultrasound, Shanghai First People’s Hospital Affiliated to Shanghai Jiao Tong University School
of Medicine, Shanghai 200080, China; 2Department of Radiology, Shanghai First People’s Hospital Affiliated to
Shanghai Jiao Tong University School of Medicine, Shanghai 200080, China; 3Department of Pathology, Shanghai
First People’s Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai 200080, China.
*
Co-first authors.
1
Received June 27, 2015; Accepted August 11, 2015; Epub August 15, 2015; Published August 30, 2015
Abstract: Bile duct hamartomas (BHs), also called von Meyenburg complex (VMC), are benign biliary malformations
that originate from disorganization of the small intrahepatic bile ducts. This disorganization is often associated with
the abnormal involution of embryonic ductal end plates in the liver. This is clinically significant, as the development
of BHs can cause diagnostic confusion with liver metastases and small hepatocellular carcinoma (SHCC). Currently,
we report a specific case of BHs and review the literature to better define and diagnose BHs. In the following case,
a 37 year-old male bearing a lesion in his liver is presented and undergoes both radiological and pathological diagnosis. The lesion is preliminarily suspected to be a hepatic hemangioma by examination of conventional ultrasound
(US), contrast enhanced ultrasound (CEUS), computerized tomographic scanning (CT) and magnetic resonance
imaging (MRI). However, SHCC is suspected by follow-up analysis of US and CEUS, due to the patient’s background
history of hepatitis B and growth of the lesion and a tumor-feeding vessel in BHs via CEUS. However, BHs are finally
diagnosed by biopsy pathology under the guidance of ultrasound. Therefore, we believe pathology is imperative for
correct diagnosis of BHs over other similar diseases when the imaging findings are atypical. Here we report the
novel and unique detection of a tumor-feeding vessel, which mimicked SHCC strongly, during the course of CEUS.
We also present a comprehensive review of the previous reported radiological examination related to BHs.
Keywords: Bile duct hamartomas (BHs), pathology, ultrasound (US), contrast enhanced ultrasound (CEUS), computerized tomographic (CT), magnetic resonance imaging (MRI)
Introduction
Bile duct hamartomas (BHs), also known as von
Meyenburg complex (VMC), were initially
described by the Swiss pathologist Hanns von
Meyenburg [1] in 1918. BHs have been previously described as benign liver malformations
resulting from maldevelopment of the ductal
end plates, which remain in a persistent double-layered cylindrical structure similar to the
fetal ductal plate [2, 3]. Therefore, pathologically, BHs can be defined by examining dilated
intrahepatic bile ductules and the surrounding
fibrous stroma [4, 5].
BHs are typically detected during laparotomy or
autopsy, and the prevalence of BH formation is
age-dependent, varying from 1% in children to
5.6% in adults in a large series of autopsy samples [4, 6, 7]. Macroscopically, BHs can be characterized by the visualization of many grayishwhitish or yellow small nodules scattered
throughout the liver, either intraparenchymally
or subcapsularly [6, 8-10]. BHs may be detected as single or multiple hamartomas, with a
diameter ranging from 1 to 15 mm [11]. During
diagnosis, its characteristics can mimic that of
liver metastases, especially when the patient
has previously suffered a primary malignancy
[9, 11].
Patients with BHs are usually symptomatic.
Jaundice, epigastralgia, and fever can occur.
Jaundice, a major symptom, is caused by
obstruction of the biliary tract owing to the
mucus secreted by biliary papillomas [4, 6, 12].
Bile duct hamartomas
Figure 1. Imaging of patient findings via US and CEUS. (A) At initial examination, a heterogeneous hypoechoic nodule
was located in the liver parenchyma via US (red arrow). (B) At initial examination, there was no blood flow in the interior of the nodule (red arrow). (C) The result of a second examination shows a similar heterogeneous nodule located
in the liver parenchyma (red arrow). (D) The results of a second examination show sporadic internal blood flow in
the interior of the nodule (red arrow). (E-H) At first CEUS examination, enhancement of the hypervascular nodule is
earlier than the surrounding liver parenchyma. The arrival time, peak time and regression time of Sonovue® are (E)
12 seconds, (F) 19 seconds and (G) 24 seconds, respectively (red arrows). (H) A tumor-feeding vessel connecting
the branch of hepatic artery and the lesion (red arrow). (I-L) At second CEUS examination, enhancement of the hypervascular nodule is also earlier than the surrounding liver parenchyma. The arrival time, peak time and regression
time of Sonovue® are (I) 10 seconds, (J) 17 seconds, and (K) 24 seconds, respectively. (L) The tumor-feeding vessel
is again detected (red arrows).
However, these symptoms are nonspecific. A
close relationship has been reported to exist
between BHs and the following congenital disorders: Caroli’s disease, polycystic liver disease, congenital hepatic fibrosis, mesenchymal
hamartomas, bile duct atresia and autosomal
recessive polycystic kidney disease [5, 8, 13,
14]. While VMC is a benign condition, there is
an ongoing debate as to whether BHs have the
potential to induce malignant degeneration to
cholangiocarcinoma and hepatocellular carcinoma [6, 8, 11, 13-15]. Here, we report a case
of VMC diagnosed through several radiological
examinations, including ultrasound (US), contrast-enhance ultrasound (CEUS), computerized tomographic scanning (CT) and magnetic
resonance imaging (MRI). We present novel
results via CEUS that suggest the occurrence of
a tumor-feeding vessel. The imaging is
enhanced in the arterial phase without early
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disappearance in the delay phase in CEUS.
Additionally, we believe imaging strategies are
not sufficient to achieve a most definitive diagnosis of BHs in the absence of typical imaging
findings.
Case report
Here, we report the occurrence of a hypoechonical solid nodule in the right liver lobe detected
by B-mode ultrasonography in a 37 year-old
male. Our study had been approved by the ethics committee of Shanghai First People’s
Hospital and the informed consent had been
waived. Considering that the patient had a history of Hepatitis B, development of a malignant
tumor could not be ruled out. At the initial
screening, CT scanning missed the nodule
because it was imaged in only one frame, which
was reluctantly discovered in a later retrospec-
Int J Clin Exp Med 2015;8(8):13145-13153
Bile duct hamartomas
Figure 2. Imaging of patient findings via CT and MRI. (A, B) CECT examination displayed no enhancement in both of
hepatic arterial phase and portal venous phase (red arrows). (C-E) Conventinal MRI examination. (C) T1WI, the lesion
is hypointense (red arrow). (D) T2WI, the lesion is hyperintense (red arrow). (E) DWI, the lesion shows free diffusion
(red arrow). (F-H) Gadolinium-enhanced T1WI. (F) The lesion displays rim enhancement in the hepatic arterial phase,
and marginal tuberculiform enhancement in both (G) portal venous phase and (H) hepatic vein (red arrows).
tive analysis of the CT imaging. Additionally, the
patient refused to undergo a MRI examination.
CEUS was further implemented, with the primary consideration being liver benign lesion.
Six months later, follow-up with US, CEUS and
MRI showed that the lesion had slightly
increased in size (on US and CEUS) and indicated the presence of a tumor-feeding vessel.
The MRI findings suggested that the lesion was
benign. Because SHCC could not be ruled out
on US and CEUS, considering the patient’s history of hepatitis B, a biopsy was indispensable
in order to identify the final diagnosis. The
patient had suffered right abdominal discomfort, but did not present with other clinical
symptoms such as fever, nausea or vomiting,
cough and expectoration, gastrointestinal dysfunction, jaundice, hematemesis or melena.
Laboratory examination found no abnormal
results, including α-FP and other tumor-related
index. Next, we present the strategies used for
diagnosis:
Conventional ultrasound (US)
The first ultrasound examination was conducted at initial screening. A solid non-homogeneous hypoechoic lesion was detected in the
liver, measuring 8.9×13.6 mm (Figure 1A). This
lesion was located in the upper segment of the
right anterior hepatic lobe, and was considered
a benign lesion due to its clear boundary, regular shape and the missing internal blood flow
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(Figure 1B). The ultrasound findings at the sixmonth follow-up were roughly consistent with
that of the first time, with a slightly increased
lesion size of 13.5×13.8 mm (Figure 1C).
However, we additionally detected an internally
sporadic blood flow (Figure 1D), which was
initially suggestive of a malignant transformation.
Contrast enhanced ultrasound (CEUS)
During the first examination, a 2.5 ml solution
of the microbubble contrast agent Sonovue®
was injected into the median cubital vein.
Twelve seconds later, the dye began to delineate the lesion outline (Figure 1E), and reached
peak contrast at 19 seconds (Figure 1F). The
contrast lasted 24 seconds and then began to
subside (Figure 1G). In the retrospective analysis six months later, a tumor-feeding vessel was
observed during the contrast enhancement
course, which was not detected at first examination of the CEUS data (Figure 1H).
At the six-month follow-up, both the arrival and
peak contrast times were two seconds earlier
than that of the first examination, 10 seconds
(Figure 1I) and 17 seconds (Figure 1J), respectively. Similar to the initial screening, imaging
started to subside at 24 seconds (Figure 1K).
Again, the tumor-feeding vessel was also
detected (Figure 1L). By CEUS examination, the
lesion demonstrated a slight increase in size,
Int J Clin Exp Med 2015;8(8):13145-13153
Bile duct hamartomas
Figure 3. Lesion biopsy under ultrasound guidance and pathological examination. (A) The lesion is clearly presented
by two-dimensional sonogram. (B) The biopsy needle is inserted into the lesion through the liver parenchyma. (C-F)
Pathological examination. (C, D) The lesion displays hyperplasia of the fibrocollagenous stroma and bile ducts ((H)
and (E) staining, (C) ×100, (D) ×400). (E, F) The bile duct epithelia is positive for CK (Immunohistochemistry of CK,
(E) ×100, (F) ×400).
which was an 11×15 mm in measurement, as
compared to the initial measurement of
8.9×13.6 mm. CEUS of the nodule displayed an
enhancement in the arterial phase, similar to
the majority of liver cancers. However, the
malignant liver lesions on CEUS typically disappear earlier than the surrounding liver parenchyma in the delay phase, but that trait did not
emerge in our patient. Thus, valid conclusions
could not be drawn to differentiate BHs and
liver metastases via US and CEUS alone.
Because of the changes in size and hemodynamics of lesion, a malignant lesion (SHCC or
liver metastasis) was initially suspected.
Contrast enhanced computed tomographic
(CECT)
To perform CECT, the arterial phase scanning
and the porto-venous phase scanning were
respectively started at 8 s and 35 s after a
bolus of Ultravist 370 was injected. At the initial
screening, CT imaging did not recognize the
liver nodule as it was detected in only one
cross-section. However, the nodule was detected in the following retrospective study of CT
imaging by improving the brightness and contrast. Compared with the surrounding liver
parenchyma, the density of the lesion was
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slightly lower and non-homogeneous. There
was no enhancement in either hepatic arterial
phase or in portal venous phase of iohexolenhanced imaging (Figure 2A, 2B).
Magnetic resonance imaging (MRI)
A MRI examination was performed at the sixmonth follow-up. A round lesion measuring 15
mm in diameter was found in the upper segment of the right anterior hepatic lobe, with
hypo-intense signals on T1-weighted images
(T1WI) (Figure 2C), hyper-intense signals on
T2-weighted images (T2WI) (Figure 2D), and
high diffusion on diffusion-weighted imaging
(DWI) (Figure 2E), respectively. After Magnevist
was injected intravenously, the lesion was
clearly imaged with rim enhancement in the
hepatic arterial phase (Figure 2F) of enhanced
T1WI, and marginally nodular enhancement
was detected in portal venous phase (Figure
2G) and delayed phase (Figure 2H). Therefore,
a liver hemangioma was suggested via the MRI
results.
Biopsy under ultrasound guidance
Due to inconclusive diagnosis by the imaging
studies, a biopsy was performed with a disposInt J Clin Exp Med 2015;8(8):13145-13153
Bile duct hamartomas
able core tissue biopsy needle, MN1820,
Bard® Magnium®, USA, under the guidance of
ultrasound instrument equipped with a puncture probe, Hitachi, Japan (Figure 3A, 3B).
Three strips of tissue samples measuring 1 cm
in diameter and 21 mm in length were obtained.
The sample strips were placed on sterilized
glass and turned over to prepare smear glass.
Next, the strips were dipped in absolute alcohol
to fix the specimens for pathological examination.
The resulting pathological sections were
reviewed by specialists in hepatobiliary pathology. It was determined that the morphology and
structure of the hepatic cells had no obvious
abnormalities. However, the small bile duct
cells were in an irregular arrangement surrounded by plenty of fibrocollagenous stroma.
In addition, the bile duct epithelia were significantly hyperplastic without cellular heteromorphism (Figure 3C, 3D). The results of the immunohistochemistry demonstrated that cytokeratins (CK) CK8, CK18 and CK19 staining were
all positive, while hepatocyte staining was negative (Figure 3E, 3F). Due to this staining, the
histological origin of the lesion was considered
to be bile duct epithelium. Therefore, bile duct
hamartoma was finally defined by pathology.
The patients were additionally followed-up both
four months and nine months after the biopsy.
At these times, the size of the lesion was
13.6×14.9 mm and 13.7×15.7 mm, respectively. The nodule increased one to two mm in nine
months. Due to the benign results of biopsy
before, BHs were considered the correct pathologic results.
Discussion
As mentioned above, BHs are considered
benign liver malformations resulting from
derangement of brephic bile duct cells, which is
proposed to be triggered by a disruptive or ischemic factor during bile duct lamina remodeling
[8, 13]. These nodules typically measure less
than 1 cm on gross observation, strongly simulating liver metastases on macroscopical
inspection and in imaging studies [9, 10].
Microscopically, BHs consist of a variable numbers of dilated, tortuous or branching intraheatic bile ducts surrounded by monoptychial
columnar or cubical bile duct epithelium. The
background is composed of abundant fibrocol-
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lagenous stroma with little vascular proliferation. Furthermore, the dilated bile ducts may
have some bile in their lumina [6, 10].
Two kinds of classification methods for BHs
have been mentioned in the literature. Based
on the degree of biliary tract dilatation within
the lesions, BHs can be divided into three
classes: (i) predominantly solid, (ii) predominantly cystic and (iii) intermediate lesions [4, 8,
16, 17]. The connections between the abnormal dilated cysts and the draining bile ducts
BHs are also divided in two types: (i) draining or
(ii) non-draining [8, 18]. However, it is also
strongly believed that there is no communication either among the deformed bile ducts or
between them and the normal biliary system
[19].
Sonographic findings of BHs detect hypoechoic, hyperechoic, or mixed non-homogeneous
echoic lesions [10, 11, 13, 20, 21]. A specific
ultrasonic characteristic of BHs is the so-called
“multiple comet-tail echoes” that have been
inferred to be strong diagnostic evidence
towards BHs [10, 11, 21]. However, this finding
was not significant in our case study. Instead,
CEUS was used to evaluate the microcirculation inside the lesion. Recently, MurphyLavallee et al. [11, 22] have suggested a unique
phenomenon wherein all metastases display
degrees of enhancement in the arterial phase
on CEUS, no matter the appearances on contrast enhanced CT and contrast enhanced MRI
[11]. There has been no contrast enhancement
reported in most cases of CEUS examination
for BHs, but this finding is currently being disputed [10, 13]. For example, Berry et al. [11,
23] and Phillip et al. [11] reported cases of multiple BHs that did not demonstrate any
enhancement in all three phases of the process (arterial, portal venous, and delayed
phase) via CEUS. On the other hand, Hohmann
et al. [11, 24] highlighted the presence of contrast enhancement on CEUS in a BH measuring
5 mm in size. Imaging of the lesion was synchronized and consistent with that of the normal liver parenchyma in arterial phase and portal venous phase, and ‘washout’ in the delayed
phase [11]. Our results of CEUS roughly agree
with the findings of Hohmann et al. The imaging
of the lesion displayed enhancement in arterial
phase, similar to imaging in liver malignancies.
These differences may vary based on lesion
Int J Clin Exp Med 2015;8(8):13145-13153
Bile duct hamartomas
size. Typically, liver malignancies on CEUS show
that the lesion disappears earlier than the surrounding liver parenchyma in the delay phase.
In our case, they are synchronous in portal
venous phase and delay phase. Innovatively,
we have also found a tumor-feeding vessel during the course of CEUS, which has never been
reported. Thus, valid conclusions cannot be
drawn between imaging differences in BH and
liver malignancies.
BH lesions are always low-density on CT scanning images. In the published literature, no
enhancement of BHs has been captured in all
phases of CECT after intravenous injection of
iodine contrast medium [13], as in our case.
However, exceptions exist in two cases [13, 25]
in which the imaging of lesions was homogeneously enhanced on post-contrast imaging of
CECT.
The typical imaging features of BHs on MRI are
hypo signal on T1WI and hyper signal on T2WI
[11, 19]. However, controversy exists when considering enhancement of gadolinium-enhanced
T1WI [11]. Both enhancement [13, 18, 21, 26,
27] and non-enhancement [11, 13] have been
reported. In our case, rim enhancement was
detected in the hepatic arterial phase, and
marginal tuberculiform enhancement was
detected in both the portal venous phase and
the delayed phase of enhanced T1WI. This was
speculated to be resulting from the compressed
liver parenchyma surrounding the lesion and
inflammatory cell infiltration [21, 27]. As a
promising part of MRI, diffusion-weighted imaging (DWI) has attracted a lot of attention in differentiating benign lesions from malignant
lesions, due to diffusion rate. It is thought that
benign lesions display free diffusion on DWI,
while malignant lesions show restricted diffusion [11]. According to this theory, the lesion in
our case could be inferred to be benign.
Additionally, it has been suggested that the
false negative rate of CT and MRI remains high
for liver metastasis less than 1.5 cm in size,
which may also explain the discrepancies in the
T2W1 enhancement. This size coincides with
the vast majority of BHs, so the differential
diagnosis would be difficult and may vary with
lesion size [28].
Magnetic resonance cholangiopancreatography (MRCP) imaging has the ability to identify
the communication between BHs and the nor13150
mal biliary system, also known as the ‘starry
sky’ phenomenon. The dilated bile ducts of BHs
are hyper intense isolated nodules scattered in
both of the liver lobes, and may or may not communicate with the draining bile ducts [6, 10,
13, 21, 27, 28]. MRCP can facilitatefacilitate
the differential diagnosis of BHs and other bile
duct anomalies such as Caroli disease by analyzing the association with the draining bile
ducts [13]. However, based on whether there
are connections between the abnormal dilated
cysts and the draining bile ducts, some believe
that BHs can be divided in two types, existence
or absence of the communication [8, 13].
The most important differential diagnosis of
BHs would be to rule out liver metastases,
which would interfere with different treatment
strategies [29], especially when the patient has
a history of malignancy [2, 28]. Other similar
diseases include primary hepatocellular carcinoma, intrahepatic cholangiocarcinoma and
circumscribed inhomogeneous fatty liver [2,
28], simple hepatic cyst, small liver abscess,
and lymphoma. To our knowledge, CEUS of BHs
has been reported in few papers, and the accuracy of CEUS in the diagnosis of BHs has not
been reported. CT helps slightly in differentiating BHs from liver metastasis but has poor tissue contrast resolution [21]. Comparatively, if
accompanied with typical imaging, MRI may be
of the greatest value in the differential diagnosis of BHs and liver metastasis, although it
remains controversial, as described above. In
addition, the dilated bile ducts of BHs can display the characteristic “starry sky” appearance
in some cases on MRCP [6, 10, 13, 21]. We
believe that pathological examination is
required in those lesions with atypical radiological performance, such as solitary lesions or
enhanced post-gadolinium during MRI [21], as
in our case.
BHs may also be confused with peribiliary gland
hamartoma (PGH), also known as intrahepatic
bile duct adenoma, originating from peribiliary
glands. Microscopic examination of PGH
reveals disordered peribiliary gland acini
embedded in fibrotic tissue which causes
chronic inflammation [8, 30]. Microscopical
examination of BHs shows small irregular and
dilated bile ducts embedded in fibrocollagenous stroma without inflammation [8, 10, 31,
32]. Part of the dilated bile ducts turn into small
Int J Clin Exp Med 2015;8(8):13145-13153
Bile duct hamartomas
cysts and maybe contain inspissated bile in
them [8, 30, 33]. However, it is difficult to distinguish BHs from PGH by macroscopical examination or imaging methods. The only difference
is that PGH has been considered to have
delayed or prolonged enhancement on contrast
enhanced CT and MRI [8]. The most precise
identifying method to distinguish BH and PGH
should be immunohistochemistry. The serous
and mucous cells comprised in acini and
tubules of PGH express D10 and 1F6, which
are also secreted by the intrahepatic peribiliary
glands [30, 34, 35]. Another related investigation demonstrates that PGH can be marked
specifically by five foregut antigens (D10, 1F6,
MUC6, MUC5AC, and TFF2) as well as the exocrinosity of acid mucin [35]. Therefore, imaging
examination may help somewhat in indicating
the most likely differential diagnosis of BHs and
PGH, with the final diagnosis depending on
pathology.
Conclusion
BHs are benign liver malformations, with an
extremely low probability of malignant transformation. Recently, they have attracted a lot of
attention because they simulate the performance of liver metastasis on gross observation
and imaging examination. As mentioned, it is
difficult with imaging studies alone to differentiate BH from similar disease. However, when
imaging, we believe MRI is most important for
differential diagnosis. Pathological examination is necessary when a history of primary
malignancy exists or the patient has atypical
appearance on radiological images. In addition,
we present diagnosis of BHs from a small lesion
in the liver of a young patient with hepatitis. We
also present a novel association between BHs
and tumor-feeding vessels that has never been
reported, potentially creating a new link
between benign lesions and tumor vasculature.
Of course, we cannot rule out that our case may
be an individual phenomenon.
Disclosure of conflict of interest
None.
Abbreviations
BHs, Bile duct hamartomas; VMC, Von Meyenburg complex; US, Ultrasound; CEUS, Contrast
enhanced ultrasound; CT, Computerized tomographic scanning; MRI, Magnetic resonance
imaging; T1WI, T1-weighted images; 2WI,
T2-weighted images; DWI, Diffusion-weighted
imaging; CK, Cytokeratin; MRCP, Magnetic resonance
cholangiopancreatography;
PGH,
Peribiliary gland hamartoma.
Address correspondence to: Dr. Lian-Fang Du,
Department of Ultrasound, Shanghai First People’s
Hospital Affiliated to Shanghai Jiao Tong University
School of Medicine, 85 Wu Jin Road, Shanghai
200080, China. Tel: +86-13386259562; Fax:
(0086) 021-37798276; E-mail: du_lianfang2013@
163.com
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Acknowledgements
This project was carried out with financial support from the National Natural Science
Foundation of China (Grant Nos. 30332369/
H1205 and 81171352/H1805) and the
Science and Technology Support Projects of
the Science and Technology Commission of
Shanghai (Grant No. 124119a5300).
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