DOI: 10.1148/rg.241035002
(Radiographics. 2004;24:53-66.)
© RSNA, 2004
Intraductal Papillary Mucinous Tumor of the Bile Ducts1
Jae Hoon Lim, MD,
Kwon-Ha Yoon, MD,
Seong Hyun Kim, MD,
Ha Young Kim, MD,
Hyo Keun Lim, MD,
Sang Yong Song, MD and
Kyung Jin Nam, MD
1 From the Departments of Radiology (J.H.L., S.H.K., H.Y.K., H.K.L.) and Pathology (S.Y.S.), Samsung Medical Center, Sungkyunkwan University School of Medicine, 50 Ilwon-dong, Kangnam-ku, Seoul 135\N710, Korea; Department of Radiology, Wonkwang University School of Medicine, Chunbuk, Korea (K.H.Y.); and Department of Radiology, Dong-A University Hospital, Busan, Korea (K.J.N.). Presented as an education exhibit at the 2002 RSNA scientific assembly. Received January 3, 2003; revision requested March 10 and received March 28; accepted May 27. Supported in part by Sungkyunkwan University and Wonkwang University. All authors have no financial relationships to disclose. Address correspondence to J.H.L. (e-mail: jhlim@smc.samsung.co.kr).
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Abstract
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Papillary tumors of the bile ducts are intraductal tumors with numerous minute frondlike papillary projections. Some intraductal papillary tumors of the bile ducts produce a large amount of mucin that disturbs bile flow and causes severe biliary dilatation. In the presence of a tumor of this subgroup, the entire biliary tree is dilated; segmental or lobar bile ducts are dilated disproportionately, and aneurysmal dilatation may occur. Mucin is depicted at cholangiography as multiple elongated or cordlike filling defects, and the tumor is depicted on cross-sectional images as a castlike, polypoid, or fungating mass in the dilated biliary tree. Based on these characteristic imaging features—dilatation, mucin, and tumor—correct diagnosis of intraductal papillary mucinous tumor of the bile ducts may be made.
© RSNA, 2004
Index Terms: Bile ducts, CT, 76.1211 • Bile ducts, MR, 76.1214 • Bile ducts, neoplasms, 76.294, 76.3192, 76.321, 76.329 • Bile ducts, stenosis or obstruction, 76.143 • Bile ducts, US, 76.1298 • Pancreatic ducts, 77.312
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LEARNING OBJECTIVES
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After reading this article and taking the test, the reader will be able to:
- Describe the histopathologic characteristics of intraductal papillary mucinous tumor of the bile ducts.
- Recognize the characteristic appearance of intraductal papillary mucinous tumor of the bile ducts at US, CT, cholangiography, and MR cholangiography.
- Identify the pattern of bile duct dilatation that enables the differential diagnosis of intraductal papillary mucinous tumor of the bile ducts.
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Introduction
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Papillary tumors of the bile ducts are characterized by intraluminal papillary masses in association with bile duct obstruction and dilatation (1–7). Some of these tumors secrete an excessive amount of mucin, which may disturb bile flow and cause severe ductal dilatation (6–8). Few reports have been published about mucin-hypersecreting bile duct tumors (6–12), but the concept and nomenclature of intraductal mucin-hypersecreting papillary tumor of the bile ducts have continued to evolve. Various terms have been used to describe this pathologic condition, including intraductal mucin-hypersecreting tumor, mucin-producing cholangiocarcinoma, mucin-hypersecreting carcinoma, ductectatic mucinous cystadenocarcinoma, and mucinous ductal ectasia of the biliary tree (10–14). The condition currently is most commonly described as intraductal papillary mucinous tumor of the bile ducts.
Intraductal papillary mucinous tumor of the pancreas, which is characterized by excessive secretion of mucin by the tumor in the pancreatic duct, is commonly acknowledged to be a distinct disease entity (15–19). Clinical symptoms of the pancreatic tumor resemble those in acute cholangitis and chronic pancreatitis and are attributable to intermittent obstruction of the bile ducts and pancreatic duct by excessive mucin secretion. Intraductal papillary mucinous tumor of the bile ducts bears a striking similarity to intraductal papillary mucinous tumor of the pancreas in its histopathologic features, production of a large amount of mucin, pathophysiologic characteristics, and resultant clinical manifestations (8). Because of the shared origins of the biliary tract and pancreas, the two systems may have a homologous pathologic condition (20). Reports have been published that describe tumors involving both the bile ducts and the pancreatic duct (21–23). In this article, the radiologic features of intraductal papillary mucinous tumor of the bile ducts are described and correlated with clinical, surgical, and histopathologic findings.
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Pathologic Description
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Intraductal papillary mucinous tumor of the bile ducts is characterized by innumerable frondlike papillary infoldings, which consist of columnar epithelial cells surrounding slender fibrovascular stalks that are supported by connective tissue from the lamina propria (Fig 1a, 1b) (12). The tumor grows slowly and tends to spread along the mucosal surface. In the late phase of development, it may invade the ductal wall and penetrate to its exterior (2,6). In the condition known as papillomatosis, tumors are multiple (1,2,4,6–8) and often extend along a lengthy segment of the intra- and extrahepatic bile ducts (12). At histopathologic analysis, intraductal papillary adenocarcinoma, adenoma, and dysplasia typically are found, frequently in coexistence (2,3,6,8). The tumor is friable and tends to slough, causing intermittent biliary obstruction that may simulate biliary stone and recurrent pyogenic cholangitis (8,9,12). A definitive diagnosis is most often made at surgery.
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Figure 1a. Gross, histopathologic, and endoscopic findings in intraductal papillary mucinous tumor of the bile ducts. (a) Photograph shows characteristic myriad frondlike papillary infoldings. (b) Photomicrograph (original magnification, x100; hematoxylin-eosin stain) shows multiple ductules with tall columnar mucinous epithelial cells and fibrovascular cores (arrows). (c) Endoscopic image of the duodenum shows greenish mucin (arrows) draining from a patulous papillary orifice.
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Figure 1b. Gross, histopathologic, and endoscopic findings in intraductal papillary mucinous tumor of the bile ducts. (a) Photograph shows characteristic myriad frondlike papillary infoldings. (b) Photomicrograph (original magnification, x100; hematoxylin-eosin stain) shows multiple ductules with tall columnar mucinous epithelial cells and fibrovascular cores (arrows). (c) Endoscopic image of the duodenum shows greenish mucin (arrows) draining from a patulous papillary orifice.
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Figure 1c. Gross, histopathologic, and endoscopic findings in intraductal papillary mucinous tumor of the bile ducts. (a) Photograph shows characteristic myriad frondlike papillary infoldings. (b) Photomicrograph (original magnification, x100; hematoxylin-eosin stain) shows multiple ductules with tall columnar mucinous epithelial cells and fibrovascular cores (arrows). (c) Endoscopic image of the duodenum shows greenish mucin (arrows) draining from a patulous papillary orifice.
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Pathophysiology of Mucin Hypersecretion
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Intraductal papillary tumor produces a variable amount of mucin that in most cases is retained in the tumor (8,13). Some papillary tumors of the bile ducts (about 24%, in our experience) produce a large amount of mucin (1,6–10,12) that is discharged into the bile duct. Excessive mucin discharge may intermittently and partially impede bile flow, whereupon obstructive jaundice occurs, sometimes complicated by cholangitis or stone formation (13). At endoscopy, a large amount of mucin may be seen draining from the patulous orifice of the duodenal papilla (Fig 1c) (8). The extrusion of mucin from the hepatopancreatic ampulla is diagnostic of a mucin-producing tumor of the bile duct or pancreas. When the flow of mucin obstructs the papilla of Vater, bile ducts both proximal and distal to the tumor become diffusely dilated. Dilatation may affect only the bile ducts in a particular hepatic segment or lobe or involve the entire biliary tree, depending on the location of the tumor. Dilatation in bile duct branches that contain tumors may be disproportionate with that in the surrounding biliary tree. In some cases, the marked dilatation of tumor-containing bile ducts may resemble that in an aneurysm (Fig 2b). Bile duct stone may occur in association with dilatation because bile stasis facilitates stone formation.
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Figure 2a. Patterns of biliary dilatation caused by intraductal papillary mucinous tumor of the bile ducts. Schematics show segmental or lobar dilatation produced by tumor-induced stenosis (a), aneurysmal dilatation caused by multiple fungating tumors (b), generalized dilatation without any visible tumor (c), and generalized dilatation with disproportionately greater dilatation of the hepatic segmental bile ducts containing a tumor (d).
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Figure 2b. Patterns of biliary dilatation caused by intraductal papillary mucinous tumor of the bile ducts. Schematics show segmental or lobar dilatation produced by tumor-induced stenosis (a), aneurysmal dilatation caused by multiple fungating tumors (b), generalized dilatation without any visible tumor (c), and generalized dilatation with disproportionately greater dilatation of the hepatic segmental bile ducts containing a tumor (d).
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Figure 2c. Patterns of biliary dilatation caused by intraductal papillary mucinous tumor of the bile ducts. Schematics show segmental or lobar dilatation produced by tumor-induced stenosis (a), aneurysmal dilatation caused by multiple fungating tumors (b), generalized dilatation without any visible tumor (c), and generalized dilatation with disproportionately greater dilatation of the hepatic segmental bile ducts containing a tumor (d).
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Figure 2d. Patterns of biliary dilatation caused by intraductal papillary mucinous tumor of the bile ducts. Schematics show segmental or lobar dilatation produced by tumor-induced stenosis (a), aneurysmal dilatation caused by multiple fungating tumors (b), generalized dilatation without any visible tumor (c), and generalized dilatation with disproportionately greater dilatation of the hepatic segmental bile ducts containing a tumor (d).
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Clinical Manifestations
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Compared with other types of cholangiocarcinoma, intraductal papillary mucinous tumor is a low-grade malignancy that is generally limited to the mucosa, although it may invade the ductal wall in the late phase. Long-term survival can be expected after surgery (2,6,7). Because the disease has a protracted course, clinical symptoms are recurrent. These may include upper abdominal pain, episodic biliary pain, fever, chills, and jaundice. These symptoms are related to the recurrent obstruction of the bile ducts by excessive mucin or by tumor-induced stenosis. Most reported cases of intraductal papillary mucinous tumors of the bile ducts have occurred in East Asian countries (6–13).
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Radiologic Findings
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A review of hospital records from two institutions for 1995–2002 disclosed 61 patients with pathologically proved papillary tumors of the bile ducts. Among these, tumors in 15 patients were diagnosed on the basis of clinical and histopathologic findings as intraductal papillary mucinous tumor of the bile ducts. The records of these 15 patients were retrospectively analyzed. In all 15 patients, excessive mucin in the biliary tree was depicted at cholangiography and endoscopy and confirmed at surgery. The clinical, radiologic, surgical, and histopathologic findings are summarized in the Table. In two patients, intrahepatic bile duct stones were associated with the condition.
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Clinical, Radiologic, Surgical, and Histopathologic Findings in Patients with Intraductal Papillary Mucinous Tumor of the Bile Ducts
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Tumor
On imaging studies, the intraductal papillary mucinous tumor may appear as a small and flat or fungating mass in the dilated bile ducts (Figs 3–5) (6,7). Large tumors may be depicted at US, computed tomography (CT), ERCP or tube cholangiography, or magnetic resonance (MR) cholangiopancreatography (6,7); however, tumors that are relatively small or sessile or that spread along the mucosal surface are difficult or impossible to visualize with imaging of any modality (6, 10,11) (Figs 6–8). Because tumor localization is essential for surgical planning, thorough examination of the dilated ducts is necessary. Cholangioscopy may be performed for this purpose when a suspected tumor is not depicted radiologically (Fig 7) (2,8). If the tumor is not visible at either imaging or cholangioscopy, the hepatic segment or portion containing the bile ducts that are disproportionately more dilated may be resected.
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Figure 3a. Papillary adenocarcinoma in a 62-year-old man who presented with epigastric pain of 6 months’ duration. Transverse contrast-enhanced CT image (a) and sonogram (b) show a nodular mass (arrow) and severe dilatation of the lateral segmental bile ducts of the left hepatic lobe. Slight dilatation of the right intrahepatic bile ducts, also visible, is caused by clonorchiasis. (c) ERCP image shows complete obstruction of the lateral segmental bile duct (arrow), normal extrahepatic bile ducts and right hepatic duct, and mild dilatation of the right intrahepatic bile ducts because of clonorchiasis. (d) Photomicrograph (original magnification, x40; hematoxylin-eosin stain) shows a papillary tumor (arrow) obstructing a segmental bile duct. (Reprinted, with permission, from reference 12.)
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Figure 3b. Papillary adenocarcinoma in a 62-year-old man who presented with epigastric pain of 6 months’ duration. Transverse contrast-enhanced CT image (a) and sonogram (b) show a nodular mass (arrow) and severe dilatation of the lateral segmental bile ducts of the left hepatic lobe. Slight dilatation of the right intrahepatic bile ducts, also visible, is caused by clonorchiasis. (c) ERCP image shows complete obstruction of the lateral segmental bile duct (arrow), normal extrahepatic bile ducts and right hepatic duct, and mild dilatation of the right intrahepatic bile ducts because of clonorchiasis. (d) Photomicrograph (original magnification, x40; hematoxylin-eosin stain) shows a papillary tumor (arrow) obstructing a segmental bile duct. (Reprinted, with permission, from reference 12.)
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Figure 3c. Papillary adenocarcinoma in a 62-year-old man who presented with epigastric pain of 6 months’ duration. Transverse contrast-enhanced CT image (a) and sonogram (b) show a nodular mass (arrow) and severe dilatation of the lateral segmental bile ducts of the left hepatic lobe. Slight dilatation of the right intrahepatic bile ducts, also visible, is caused by clonorchiasis. (c) ERCP image shows complete obstruction of the lateral segmental bile duct (arrow), normal extrahepatic bile ducts and right hepatic duct, and mild dilatation of the right intrahepatic bile ducts because of clonorchiasis. (d) Photomicrograph (original magnification, x40; hematoxylin-eosin stain) shows a papillary tumor (arrow) obstructing a segmental bile duct. (Reprinted, with permission, from reference 12.)
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Figure 3d. Papillary adenocarcinoma in a 62-year-old man who presented with epigastric pain of 6 months’ duration. Transverse contrast-enhanced CT image (a) and sonogram (b) show a nodular mass (arrow) and severe dilatation of the lateral segmental bile ducts of the left hepatic lobe. Slight dilatation of the right intrahepatic bile ducts, also visible, is caused by clonorchiasis. (c) ERCP image shows complete obstruction of the lateral segmental bile duct (arrow), normal extrahepatic bile ducts and right hepatic duct, and mild dilatation of the right intrahepatic bile ducts because of clonorchiasis. (d) Photomicrograph (original magnification, x40; hematoxylin-eosin stain) shows a papillary tumor (arrow) obstructing a segmental bile duct. (Reprinted, with permission, from reference 12.)
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Figure 4a. Papillary adenocarcinoma in a 63-year-old man who presented with epigastric pain. (a, b) Transverse contrast-enhanced CT images (a at a level higher than b) show aneurysmal dilatation of the lateral segmental bile ducts of the left hepatic lobe containing multiple fungating masses and associated peripheral bile duct dilatation (arrow in a). (c) Photograph of the resected specimen shows fungating tumors. Histopathologic findings confirmed the diagnosis.
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Figure 4b. Papillary adenocarcinoma in a 63-year-old man who presented with epigastric pain. (a, b) Transverse contrast-enhanced CT images (a at a level higher than b) show aneurysmal dilatation of the lateral segmental bile ducts of the left hepatic lobe containing multiple fungating masses and associated peripheral bile duct dilatation (arrow in a). (c) Photograph of the resected specimen shows fungating tumors. Histopathologic findings confirmed the diagnosis.
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Figure 4c. Papillary adenocarcinoma in a 63-year-old man who presented with epigastric pain. (a, b) Transverse contrast-enhanced CT images (a at a level higher than b) show aneurysmal dilatation of the lateral segmental bile ducts of the left hepatic lobe containing multiple fungating masses and associated peripheral bile duct dilatation (arrow in a). (c) Photograph of the resected specimen shows fungating tumors. Histopathologic findings confirmed the diagnosis.
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Figure 5a. Papillary adenocarcinoma in a 71-year-old man with no abdominal symptoms. (a) Transverse contrast-enhanced portal phase CT image shows aneurysmally dilated bile ducts in the lateral segment of the left hepatic lobe and small intraductal masses (arrow). Bile ducts in the medial segments of the left and right hepatic lobes, as well as the extrahepatic ducts, are not dilated. (b) Sonogram of the left hepatic lobe, obtained during work-up for lung cancer staging, shows a large complex solid mass (arrows) in the aneurysmally dilated bile ducts. Histopathologic findings confirmed the diagnosis.
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Figure 5b. Papillary adenocarcinoma in a 71-year-old man with no abdominal symptoms. (a) Transverse contrast-enhanced portal phase CT image shows aneurysmally dilated bile ducts in the lateral segment of the left hepatic lobe and small intraductal masses (arrow). Bile ducts in the medial segments of the left and right hepatic lobes, as well as the extrahepatic ducts, are not dilated. (b) Sonogram of the left hepatic lobe, obtained during work-up for lung cancer staging, shows a large complex solid mass (arrows) in the aneurysmally dilated bile ducts. Histopathologic findings confirmed the diagnosis.
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Figure 6a. Papillary tumor of borderline malignancy in a 48-year-old woman who presented with abdominal pain and intermittent fever and chills. (a) Transverse contrast-enhanced portal phase CT image shows severe dilatation of the left and right intrahepatic bile ducts and marked dilatation of the extrahepatic bile ducts but no mucin or tumor. Note the disproportionately greater dilatation of the left hepatic bile ducts. (b) Sonogram shows severe dilatation of the intra- and extrahepatic bile ducts (CBD), which have diameters of 1.5-2.5 cm, but no evidence of mucin or tumor in the dilated ducts. (c) ERCP image shows severe dilatation of the biliary tree, especially the left hepatic ducts, and multiple cordlike filling defects (straight arrow) that represent mucin in the extrahepatic bile ducts. These defects were only minimally altered by forceful injection of contrast material into the ducts. Note also the oval filling defect (curved arrow), which indicates the presence of a nodule in the lateral segmental bile duct of the left hepatic lobe. At surgery, a papillary tumor with a volume of 1.2 x 1.0 x 1.0 cm was found in the left intrahepatic duct, and a large amount of mucin was found in the dilated extrahepatic bile ducts.
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Figure 6b. Papillary tumor of borderline malignancy in a 48-year-old woman who presented with abdominal pain and intermittent fever and chills. (a) Transverse contrast-enhanced portal phase CT image shows severe dilatation of the left and right intrahepatic bile ducts and marked dilatation of the extrahepatic bile ducts but no mucin or tumor. Note the disproportionately greater dilatation of the left hepatic bile ducts. (b) Sonogram shows severe dilatation of the intra- and extrahepatic bile ducts (CBD), which have diameters of 1.5-2.5 cm, but no evidence of mucin or tumor in the dilated ducts. (c) ERCP image shows severe dilatation of the biliary tree, especially the left hepatic ducts, and multiple cordlike filling defects (straight arrow) that represent mucin in the extrahepatic bile ducts. These defects were only minimally altered by forceful injection of contrast material into the ducts. Note also the oval filling defect (curved arrow), which indicates the presence of a nodule in the lateral segmental bile duct of the left hepatic lobe. At surgery, a papillary tumor with a volume of 1.2 x 1.0 x 1.0 cm was found in the left intrahepatic duct, and a large amount of mucin was found in the dilated extrahepatic bile ducts.
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Figure 6c. Papillary tumor of borderline malignancy in a 48-year-old woman who presented with abdominal pain and intermittent fever and chills. (a) Transverse contrast-enhanced portal phase CT image shows severe dilatation of the left and right intrahepatic bile ducts and marked dilatation of the extrahepatic bile ducts but no mucin or tumor. Note the disproportionately greater dilatation of the left hepatic bile ducts. (b) Sonogram shows severe dilatation of the intra- and extrahepatic bile ducts (CBD), which have diameters of 1.5-2.5 cm, but no evidence of mucin or tumor in the dilated ducts. (c) ERCP image shows severe dilatation of the biliary tree, especially the left hepatic ducts, and multiple cordlike filling defects (straight arrow) that represent mucin in the extrahepatic bile ducts. These defects were only minimally altered by forceful injection of contrast material into the ducts. Note also the oval filling defect (curved arrow), which indicates the presence of a nodule in the lateral segmental bile duct of the left hepatic lobe. At surgery, a papillary tumor with a volume of 1.2 x 1.0 x 1.0 cm was found in the left intrahepatic duct, and a large amount of mucin was found in the dilated extrahepatic bile ducts.
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Figure 7a. Papillary adenocarcinomatosis in a 70-year-old woman who presented with intermittent abdominal pain, fever, chills, and jaundice. (a) Sagittal single shot fast spin-echo MR cholangiopancreatographic image shows marked dilatation of the extrahepatic ducts and the posterior segmental bile ducts in the right hepatic lobe and mild dilatation of the anterior segmental ducts of the right hepatic lobe and the left hepatic ducts. (b) Transverse T2-weighted fast spin-echo MR cholangiopancreatographic image shows the dilated posterior segmental bile ducts of the right hepatic lobe with their serrated margins (arrows) but no definite mass. (c) Transverse contrast-enhanced CT image shows markedly dilated posterior segmental bile ducts of the right hepatic lobe with some irregularity along the dilated ducts (arrow) but no tumor or mucin. (d) Sonogram of the right hepatic lobe shows no tumor or mucin, but the irregular contours of the dilated posterior segmental duct (arrow) indicate the presence of sessile tumors attached to the bile duct wall. (e) ERCP image shows large elongated filling defects (arrows) that represent mucin in the extrahepatic ducts but does not show the posterior segmental bile ducts of the right hepatic lobe, probably because of obstruction by the mucin plug. (f) Endoscopic image of the papilla of Vater in the descending duodenum shows a patulous papillary orifice from which flows a large amount of greenish-yellow mucin (arrows). Papillary tumors were found in the posterior segmental bile ducts at cholangioscopy. (g) The resected liver specimen shows a sessile papillary tumor (arrow) in the markedly dilated posterior segmental bile ducts of the right hepatic lobe. The intra- and extrahepatic bile ducts were dilated and filled with mucin.
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Figure 7b. Papillary adenocarcinomatosis in a 70-year-old woman who presented with intermittent abdominal pain, fever, chills, and jaundice. (a) Sagittal single shot fast spin-echo MR cholangiopancreatographic image shows marked dilatation of the extrahepatic ducts and the posterior segmental bile ducts in the right hepatic lobe and mild dilatation of the anterior segmental ducts of the right hepatic lobe and the left hepatic ducts. (b) Transverse T2-weighted fast spin-echo MR cholangiopancreatographic image shows the dilated posterior segmental bile ducts of the right hepatic lobe with their serrated margins (arrows) but no definite mass. (c) Transverse contrast-enhanced CT image shows markedly dilated posterior segmental bile ducts of the right hepatic lobe with some irregularity along the dilated ducts (arrow) but no tumor or mucin. (d) Sonogram of the right hepatic lobe shows no tumor or mucin, but the irregular contours of the dilated posterior segmental duct (arrow) indicate the presence of sessile tumors attached to the bile duct wall. (e) ERCP image shows large elongated filling defects (arrows) that represent mucin in the extrahepatic ducts but does not show the posterior segmental bile ducts of the right hepatic lobe, probably because of obstruction by the mucin plug. (f) Endoscopic image of the papilla of Vater in the descending duodenum shows a patulous papillary orifice from which flows a large amount of greenish-yellow mucin (arrows). Papillary tumors were found in the posterior segmental bile ducts at cholangioscopy. (g) The resected liver specimen shows a sessile papillary tumor (arrow) in the markedly dilated posterior segmental bile ducts of the right hepatic lobe. The intra- and extrahepatic bile ducts were dilated and filled with mucin.
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Figure 7c. Papillary adenocarcinomatosis in a 70-year-old woman who presented with intermittent abdominal pain, fever, chills, and jaundice. (a) Sagittal single shot fast spin-echo MR cholangiopancreatographic image shows marked dilatation of the extrahepatic ducts and the posterior segmental bile ducts in the right hepatic lobe and mild dilatation of the anterior segmental ducts of the right hepatic lobe and the left hepatic ducts. (b) Transverse T2-weighted fast spin-echo MR cholangiopancreatographic image shows the dilated posterior segmental bile ducts of the right hepatic lobe with their serrated margins (arrows) but no definite mass. (c) Transverse contrast-enhanced CT image shows markedly dilated posterior segmental bile ducts of the right hepatic lobe with some irregularity along the dilated ducts (arrow) but no tumor or mucin. (d) Sonogram of the right hepatic lobe shows no tumor or mucin, but the irregular contours of the dilated posterior segmental duct (arrow) indicate the presence of sessile tumors attached to the bile duct wall. (e) ERCP image shows large elongated filling defects (arrows) that represent mucin in the extrahepatic ducts but does not show the posterior segmental bile ducts of the right hepatic lobe, probably because of obstruction by the mucin plug. (f) Endoscopic image of the papilla of Vater in the descending duodenum shows a patulous papillary orifice from which flows a large amount of greenish-yellow mucin (arrows). Papillary tumors were found in the posterior segmental bile ducts at cholangioscopy. (g) The resected liver specimen shows a sessile papillary tumor (arrow) in the markedly dilated posterior segmental bile ducts of the right hepatic lobe. The intra- and extrahepatic bile ducts were dilated and filled with mucin.
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Figure 7d. Papillary adenocarcinomatosis in a 70-year-old woman who presented with intermittent abdominal pain, fever, chills, and jaundice. (a) Sagittal single shot fast spin-echo MR cholangiopancreatographic image shows marked dilatation of the extrahepatic ducts and the posterior segmental bile ducts in the right hepatic lobe and mild dilatation of the anterior segmental ducts of the right hepatic lobe and the left hepatic ducts. (b) Transverse T2-weighted fast spin-echo MR cholangiopancreatographic image shows the dilated posterior segmental bile ducts of the right hepatic lobe with their serrated margins (arrows) but no definite mass. (c) Transverse contrast-enhanced CT image shows markedly dilated posterior segmental bile ducts of the right hepatic lobe with some irregularity along the dilated ducts (arrow) but no tumor or mucin. (d) Sonogram of the right hepatic lobe shows no tumor or mucin, but the irregular contours of the dilated posterior segmental duct (arrow) indicate the presence of sessile tumors attached to the bile duct wall. (e) ERCP image shows large elongated filling defects (arrows) that represent mucin in the extrahepatic ducts but does not show the posterior segmental bile ducts of the right hepatic lobe, probably because of obstruction by the mucin plug. (f) Endoscopic image of the papilla of Vater in the descending duodenum shows a patulous papillary orifice from which flows a large amount of greenish-yellow mucin (arrows). Papillary tumors were found in the posterior segmental bile ducts at cholangioscopy. (g) The resected liver specimen shows a sessile papillary tumor (arrow) in the markedly dilated posterior segmental bile ducts of the right hepatic lobe. The intra- and extrahepatic bile ducts were dilated and filled with mucin.
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Figure 7e. Papillary adenocarcinomatosis in a 70-year-old woman who presented with intermittent abdominal pain, fever, chills, and jaundice. (a) Sagittal single shot fast spin-echo MR cholangiopancreatographic image shows marked dilatation of the extrahepatic ducts and the posterior segmental bile ducts in the right hepatic lobe and mild dilatation of the anterior segmental ducts of the right hepatic lobe and the left hepatic ducts. (b) Transverse T2-weighted fast spin-echo MR cholangiopancreatographic image shows the dilated posterior segmental bile ducts of the right hepatic lobe with their serrated margins (arrows) but no definite mass. (c) Transverse contrast-enhanced CT image shows markedly dilated posterior segmental bile ducts of the right hepatic lobe with some irregularity along the dilated ducts (arrow) but no tumor or mucin. (d) Sonogram of the right hepatic lobe shows no tumor or mucin, but the irregular contours of the dilated posterior segmental duct (arrow) indicate the presence of sessile tumors attached to the bile duct wall. (e) ERCP image shows large elongated filling defects (arrows) that represent mucin in the extrahepatic ducts but does not show the posterior segmental bile ducts of the right hepatic lobe, probably because of obstruction by the mucin plug. (f) Endoscopic image of the papilla of Vater in the descending duodenum shows a patulous papillary orifice from which flows a large amount of greenish-yellow mucin (arrows). Papillary tumors were found in the posterior segmental bile ducts at cholangioscopy. (g) The resected liver specimen shows a sessile papillary tumor (arrow) in the markedly dilated posterior segmental bile ducts of the right hepatic lobe. The intra- and extrahepatic bile ducts were dilated and filled with mucin.
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Figure 7f. Papillary adenocarcinomatosis in a 70-year-old woman who presented with intermittent abdominal pain, fever, chills, and jaundice. (a) Sagittal single shot fast spin-echo MR cholangiopancreatographic image shows marked dilatation of the extrahepatic ducts and the posterior segmental bile ducts in the right hepatic lobe and mild dilatation of the anterior segmental ducts of the right hepatic lobe and the left hepatic ducts. (b) Transverse T2-weighted fast spin-echo MR cholangiopancreatographic image shows the dilated posterior segmental bile ducts of the right hepatic lobe with their serrated margins (arrows) but no definite mass. (c) Transverse contrast-enhanced CT image shows markedly dilated posterior segmental bile ducts of the right hepatic lobe with some irregularity along the dilated ducts (arrow) but no tumor or mucin. (d) Sonogram of the right hepatic lobe shows no tumor or mucin, but the irregular contours of the dilated posterior segmental duct (arrow) indicate the presence of sessile tumors attached to the bile duct wall. (e) ERCP image shows large elongated filling defects (arrows) that represent mucin in the extrahepatic ducts but does not show the posterior segmental bile ducts of the right hepatic lobe, probably because of obstruction by the mucin plug. (f) Endoscopic image of the papilla of Vater in the descending duodenum shows a patulous papillary orifice from which flows a large amount of greenish-yellow mucin (arrows). Papillary tumors were found in the posterior segmental bile ducts at cholangioscopy. (g) The resected liver specimen shows a sessile papillary tumor (arrow) in the markedly dilated posterior segmental bile ducts of the right hepatic lobe. The intra- and extrahepatic bile ducts were dilated and filled with mucin.
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Figure 7g. Papillary adenocarcinomatosis in a 70-year-old woman who presented with intermittent abdominal pain, fever, chills, and jaundice. (a) Sagittal single shot fast spin-echo MR cholangiopancreatographic image shows marked dilatation of the extrahepatic ducts and the posterior segmental bile ducts in the right hepatic lobe and mild dilatation of the anterior segmental ducts of the right hepatic lobe and the left hepatic ducts. (b) Transverse T2-weighted fast spin-echo MR cholangiopancreatographic image shows the dilated posterior segmental bile ducts of the right hepatic lobe with their serrated margins (arrows) but no definite mass. (c) Transverse contrast-enhanced CT image shows markedly dilated posterior segmental bile ducts of the right hepatic lobe with some irregularity along the dilated ducts (arrow) but no tumor or mucin. (d) Sonogram of the right hepatic lobe shows no tumor or mucin, but the irregular contours of the dilated posterior segmental duct (arrow) indicate the presence of sessile tumors attached to the bile duct wall. (e) ERCP image shows large elongated filling defects (arrows) that represent mucin in the extrahepatic ducts but does not show the posterior segmental bile ducts of the right hepatic lobe, probably because of obstruction by the mucin plug. (f) Endoscopic image of the papilla of Vater in the descending duodenum shows a patulous papillary orifice from which flows a large amount of greenish-yellow mucin (arrows). Papillary tumors were found in the posterior segmental bile ducts at cholangioscopy. (g) The resected liver specimen shows a sessile papillary tumor (arrow) in the markedly dilated posterior segmental bile ducts of the right hepatic lobe. The intra- and extrahepatic bile ducts were dilated and filled with mucin.
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Figure 8a. Papillary adenocarcinoma with bile duct rupture in a 54-year-old woman who presented with indigestion and pain in the right upper abdominal quadrant 2 years after undergoing left lateral hepatic segmentectomy for cholangitis and intrahepatic bile duct stones. (a) Transverse contrast-enhanced CT image shows aneurysmal dilatation of the medial segmental bile ducts (black arrows) and a circular fluid collection anterior to the stomach (white arrow). (b) Sonogram of the liver shows severe dilatation of the bile ducts in the left hepatic lobe (arrow). (c) Coronal MR cholangiopancreatographic image shows diffuse dilatation of the biliary tree and aneurysmal dilatation (curved arrow) of bile ducts in the medial segment of the left hepatic lobe, as well as a large oval fluid collection (straight arrow) in the left upper abdominal quadrant. At surgery, a collection of mucin was found that had formed anterior to the stomach as a result of rupture of the dilated left intrahepatic duct. The histopathologic diagnosis was mucin-hypersecreting well-differentiated adenocarcinoma.
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Figure 8b. Papillary adenocarcinoma with bile duct rupture in a 54-year-old woman who presented with indigestion and pain in the right upper abdominal quadrant 2 years after undergoing left lateral hepatic segmentectomy for cholangitis and intrahepatic bile duct stones. (a) Transverse contrast-enhanced CT image shows aneurysmal dilatation of the medial segmental bile ducts (black arrows) and a circular fluid collection anterior to the stomach (white arrow). (b) Sonogram of the liver shows severe dilatation of the bile ducts in the left hepatic lobe (arrow). (c) Coronal MR cholangiopancreatographic image shows diffuse dilatation of the biliary tree and aneurysmal dilatation (curved arrow) of bile ducts in the medial segment of the left hepatic lobe, as well as a large oval fluid collection (straight arrow) in the left upper abdominal quadrant. At surgery, a collection of mucin was found that had formed anterior to the stomach as a result of rupture of the dilated left intrahepatic duct. The histopathologic diagnosis was mucin-hypersecreting well-differentiated adenocarcinoma.
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Figure 8c. Papillary adenocarcinoma with bile duct rupture in a 54-year-old woman who presented with indigestion and pain in the right upper abdominal quadrant 2 years after undergoing left lateral hepatic segmentectomy for cholangitis and intrahepatic bile duct stones. (a) Transverse contrast-enhanced CT image shows aneurysmal dilatation of the medial segmental bile ducts (black arrows) and a circular fluid collection anterior to the stomach (white arrow). (b) Sonogram of the liver shows severe dilatation of the bile ducts in the left hepatic lobe (arrow). (c) Coronal MR cholangiopancreatographic image shows diffuse dilatation of the biliary tree and aneurysmal dilatation (curved arrow) of bile ducts in the medial segment of the left hepatic lobe, as well as a large oval fluid collection (straight arrow) in the left upper abdominal quadrant. At surgery, a collection of mucin was found that had formed anterior to the stomach as a result of rupture of the dilated left intrahepatic duct. The histopathologic diagnosis was mucin-hypersecreting well-differentiated adenocarcinoma.
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Mucin
At US, mucin is usually anechoic, like bile (Figs 6, 7). Previous investigators have reported echogenic mucin indicative of mucin-producing bile duct tumor (10), but such reports are not supported by our experience. The attenuation of mucin at CT is the same as that of water, and therefore the presence of mucin cannot be determined with CT (Figs 6–8). The signal intensity of mucin at MR imaging also is the same as that of water, making mucin impossible to detect on MR images (Figs 7, 8). At ERCP or percutaneous transhepatic cholangiography, however, mucin lumps may be depicted as multiple elongated linear, ovoid, or amorphous filling defects in the dilated bile ducts (Figs 5–7) (6–8).
Bile Duct Dilatation
The biliary tree becomes dilated when either tumor or mucin impedes the bile flow through the bile ducts (Fig 3). In the presence of mucin overproduction, the entire biliary tree (both intra- and extrahepatic ducts) is markedly dilated; both proximal and distal bile ducts are dilated, irrespective of the location of the primary mucin-producing tumor (Figs 6–8) (6,7). Marked general dilatation occurs because the hepatopancreatic ampulla is obstructed, even if only intermittently and incompletely, by a large amount of viscous mucin.
When a tumor develops in a hepatic lobar bile duct, the bile ducts in that lobe are disproportionately more dilated than the ducts in the contralateral lobe (Figs 6–8). Bile ducts that contain a tumor also may show focal dilatation like that of an aneurysm (Figs 4, 5) (6,7,11). We consider aneurysmal dilatation of any branch of the biliary tree a characteristic sign of intraductal papillary mucinous tumor of the bile ducts. The disproportionate or aneurysmal dilatation of the bile duct surrounding a mucin-producing tumor is the result of obstructed bile flow, which in turn may result from direct impingement by the tumor, as well as from excessive mucin secretion. Therefore, even when no mass is visible on images, a tumor may be present in the portion of the biliary tree that is disproportionately more dilated (Fig 7). Dilated peripheral intrahepatic bile ducts, furthermore, have a propensity to rupture, causing the formation of a mucin collection or the implantation of tumor cells in the peritoneal space (pseudomyxoma peritonei) (Fig 8). Rupture probably results from increased pressure in the severely dilated bile ducts.
When diffuse or segmental biliary dilatation is seen with or without a definite mass at US, CT, or MR imaging, the use of duodenal endoscopy and ERCP is recommended to determine the presence and location of the suspected intraductal tumor and the presence and quantity of mucin draining from the major duodenal papilla. If the tumor is not depicted at imaging, cholangioscopy and cholangioscopic biopsy should be performed.
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Differential Diagnosis
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Papillary mucinous tumors can be easily differentiated from other cholangiocarcinomas because the latter manifest as nodular masses or segmental stenoses of the bile ducts with proximal dilatation, whereas papillary mucinous tumors typically manifest with diffuse or segmental dilatation and either with or without a visible bile duct mass or stenosis. In addition, the intraductal masses in papillary mucinous tumor may be either single or multiple (24).
Intraductal papillary mucinous tumor of the bile ducts may be misdiagnosed as recurrent pyogenic cholangitis with bile duct stones because both diseases involve intermittent and incomplete biliary obstruction and evident intraluminal masses or filling defects at imaging (6,25). Mucin plugs or sloughed tumor may be confused with stones at both US and cholangiography. Likewise, recurrent pyogenic cholangitis with focal bile duct stricture may be misdiagnosed as papillary tumor of the intrahepatic bile duct because both diseases may produce segmental or lobar bile duct dilatation without identifiable masses or stones (26). Obstruction of the severely dilated lobar or segmental intrahepatic duct in pyogenic cholangitis may be caused by a cystic mass that mimics hepatic abscess, necrotic tumor, or biliary cystadenoma or cystadenocarcinoma (11,27). Although cystadenoma and cystadenocarcinoma may be mucin hypersecreting, the mucin they produce is confined in the cystic tumor and does not enter the bile ducts (10,25).
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Conclusions
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Intraductal papillary mucinous tumors of the bile ducts secrete a large amount of mucin, which results in intermittent obstruction of the segmental or lobar bile ducts or the entire biliary tree, depending on the location of the tumor and the amount of mucin. When generalized biliary dilatation is evident on images that also show disproportionately more severe or aneurysmal dilatation in part of the biliary tree and excessive mucin in the bile ducts, a diagnosis of intraductal papillary mucinous tumor of the bile ducts should be considered. US, CT, and MR images should be carefully scrutinized for evidence of mucin-producing papillary tumor in the dilated bile ducts, especially in the disproportionately more dilated part of the biliary tree. On the basis of this characteristic pattern of biliary dilatation and intraductal tumor depicted at US, CT, ERCP or tube cholangiography, or MR cholangiopancreatography, one can make a diagnosis of intraductal papillary mucinous tumor of the bile ducts.
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Footnotes
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Editor’s Note.—On occasion and because of the educational nature of the Journal, RadioGraphics receives material that is similar to that published in other journals. During the editorial review process, a decision about the advisability of publication is made by the editor in consultation with peer reviewers and editorial board members. This decision is dependent on factors such as degree of similarity, interest, and venue of prior publication. Portions of this article are similar in content to a previous publication (Lim JH, Yi CA, Lim HK, Lee WJ, Lee SJ, Kim SH, Radiological spectrum of intraductal papillary tumors of the bile ducts, Korean J Radiol 2002; 3:57–63). In this instance, the editor believes that the interest in this subject justifies the current publication in RadioGraphics.
See the commentary by Levy
following this article.
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References
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