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Original Report |
1
Department of Radiology, Wonkwang University School of Medicine, 344-2
Singyong-dong, Iksan, Chunbuk 570-180, Korea.
2
Department of Radiology, Asan Medical Center, University of Ulsan College of
Medicine, 388-1, Poongnab-dong, Songpa-ku, Seoul 138-040, Korea.
3
Department of Pathology, Wonkwang University School of Medicine, Chunbuk
570-180, Korea.
4
Department of General Surgery, Wonkwang University School of Medicine, Chunbuk
570-180, Korea.
5
Department of Radiology, Samsung Medical Center, Sunggyunkwan University
School of Medicine, 50 Ilwon-dong, Kangnam-ku, Seoul 135-710, Korea.
Received January 5, 2000;
accepted after revision March 20, 2000.
Supported by Wonkwang University.
Abstract
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CONCLUSION. CT is a useful technique for revealing intraductal lesions, although the findings are nonspecific and variable. When intraductal masses or nodules are seen with localized dilatation of the intrahepatic bile ducts on CT scans, malignant papillary neoplasms of the intrahepatic bile ducts should be included in the differential diagnosis.
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Papillary neoplasms of the bile duct can be histologically classified into papillomas and papillary adenocarcinomas [2]. Additionally, multiple neoplasms, called biliary papillomatosis, also belong to this class of tumors that usually appear as multicentric papillomas involving the intrahepatic or extrahepatic biliary tract [2, 4]. These papillary tumors grow slowly and tend to be less aggressive than traditional cholangiocarcinomas [1]. Although most intrahepatic cholangiocarcinomas have limited resectability and a poor prognosis, this type of biliary neoplasm is of low-grade malignancy and merits consideration for surgery. Therefore, early diagnosis of this disease is important to maximize patient survival.
To detect and characterize abnormalities of the hepatobiliary systems, various imaging techniques have been used, including CT, sonography, direct cholangiography, and MR cholangiopancreatography [6,7,8]. However, the role of these techniques for diagnosing malignant papillary neoplasms of the intrahepatic bile ducts has not been investigated.
We report the CT and pathologic findings in a series of patients with malignant papillary neoplasms of the intrahepatic bile duct.
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Abdominal CT was performed on a Somatom Plus-S scanner (Siemens, Erlangen, Germany) in 10 patients and on a HiSpeed Advantage system (General Electric Medical Systems, Milwaukee, WI) in five. Contrast material (Iopamiro 300 [iopamidol]; Bracco, Milan, Italy) was administered IV as a 100-120 mL bolus with a mechanical power injector (MCT Plus; Medrad, Pittsburgh, PA), and unenhanced and contrast-enhanced CT scans were obtained. The rate of injection was 2.5-3.0 mL/sec. After the start of the infusion, arterial phase (30-35 sec) and portal venous phase (65-70 sec) images were obtained in 13 patients using a helical technique with a pitch of 1.0-1.5. In three patients, only portal venous phase images (65-70 sec) were obtained. Section thickness ranged from 5 to 10 mm and in most cases was 7 mm. For all patients, the time interval between radiologic imaging and surgery ranged from 1 day to 3 weeks.
CT findings were analyzed for the presence of intraductal mass or nodule, appearance of tumoral margin (well defined or poorly defined), attenuation of tumor (hypoattenuating, isoattenuating, or hyperattenuating), presence of bile duct dilatation (localized or diffuse), and ancillary findings such as the presence of hepatolithiasis. The imaging features of the tumor were correlated with the gross features and histopathologic findings of surgical specimens.
For gross specimens, tumors were classified into three types: expansile polypoid masses or nodules located in the dilated intrahepatic bile duct; sessile or plaquelike nodules; or multiple polyposis. Expansile polypoid masses or nodules included intraductal expansile masses (diameter, >2.0 cm) or nodules (diameter, <2.0 cm) that were confined to the dilated bile duct. Sessile or plaquelike nodules included intraductal nodules with sessile and broad-based polyp or plaque-like tumor. Multiple polyposis included multiple or innumerable polypoid nodules present along the bile duct. Mucin-hypersecreting tumors were defined as papillary cholangiocarcinoma with hyperproduction of mucin regardless of whether the gross morphology of the intraductal mass resulted in massive mucin retention and bile stasis in the intrahepatic or extrahepatic bile duct.
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Postoperative follow-up ranged from 3 to 47 months, with a mean of 18.6 months. At follow-up, 12 of 15 patients were alive without tumor recurrence or metastasis on CT. However, three patients died 15, 17, and 26 months after surgery, as a result of biliary sepsis (n = 2) or hepatic failure (n = 1) directly related to tumor recurrence or metastasis.
Pathologic Findings
All 15 patients' conditions were diagnosed as papillary adenocarcinoma. Of
these patients, the malignancy developed from multiple papillomatosis in three
patients. On gross specimens, solitary papillary tumors ranged in size from
1.0 to 4.5 cm (mean, 2.5 cm), and multiple papillomatosis ranged in size from
0.5 to 2.0 cm (most were <1.0 cm). On gross specimens, tumors appeared as
expansile masses or nodules (Fig.
1A,1B,1C)
in the dilated intrahepatic duct of eight patients, as sessile or plaquelike
nodules (Fig.
2A,2B,2C)
in four, and as multiple polypoid nodules (Fig.
3A,3B)
in three. On microscopic examination, distinctive papillary proliferation of
the bile duct epithelial cells around slender fibrovascular stalks, a
characteristic finding in papillary tumors, was seen in all patients (Figs.
1A,1B,1C
and
2A,2B,2C).
Of our 15 patients, three had mucin-hypersecreting tumors, two had polypoid
masses measuring 4.5 cm with cystic dilatation of the intrahepatic bile duct
(Fig.
4A,4B),
and one had an intraductal sessile polyp in the intrahepatic bile duct.
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The degree of tumor in the 15 patients was well differentiated in 11 and moderately differentiated in four. The depth of invasion was limited to the mucosal epithelium in six patients, restricted to the fibromuscular layer in two, involved the periductal connective tissue in two, and extended into the hepatic parenchyma in five. On surgical specimens, we noted no vascular invasion or intrahepatic or lymph node metastasis.
CT Findings
The CT findings in the 15 patients are given in
Table 1. Most of the tumors
were seen as hypo- or isoattenuating masses or nodules on contrast-enhanced
CT. Two tumors appeared on unenhanced CT as hypoattenuating masses; the
remaining tumors did not appear on unenhanced CT. In eight patients with
expansile masses or nodules in the dilated bile duct, tumors appeared as
hypoattenuating (7/8; 87%) or isoattenuating (1/8; 13%) lesions with distinct
margins and localized dilatation of the proximal bile ducts on
contrast-enhanced imaging (Figs.
1A,1B,1C
and
4A,4B).
In eight patients, hypoattenuating masses or nodules showed prolonged
enhancement on portal venous phase images and a thin rim of enhancement at the
periphery of the lesion on arterial phase images. However, isoattenuating
masses showed a similar degree of enhancement in the hepatic parenchyma on two
sequences of contrast-enhanced CT. Of four patients who had sessile or
plaquelike nodules, two showed a hypoattenuating nodule on two phases of
contrast-enhanced imaging; the other two patients exhibited only localized
dilatation of the bile duct without a definite mass (Fig.
2A,2B,2C).
Of three patients with multiple papillomatosis, two showed poorly defined,
hypoattenuating nodular lesions along the intrahepatic bile duct (Fig.
3A,3B);
the other patient had poorly defined, hypoattenuating masses at the left main
hepatic duct with marked dilatation of the proximal duct. Of three patients
with mucin-hypersecreting tumors, two had a hypoattenuating mass on two phases
of contrast-enhanced imaging, cystic dilatation of the left intrahepatic duct,
and diffuse dilatation of the extrahepatic bile duct (Fig.
4A,4B);
the other patient had an isoattenuating polypoid nodule with marked dilatation
of the intra- and extrahepatic ducts. Ancillary findings included
hepatolithiasis in one patient.
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The radiologic features of intrahepatic cholangiocarcinoma are closely related to its gross pathologic appearance and modes of extension [6, 7]. Until now, mainly the peripheral and hilar types of cholangiocarcinoma have appeared in the radiology literature [6,7,8], and the imaging features of papillary neoplasm of the intrahepatic bile duct have been published sporadically [2,3,4,5]. Hilar cholangiocarcinoma usually appears on CT and cholangiography as an ill-defined infiltrating tumor at the porta hepatis [7]. However, in some patients, this tumor appears on CT as a well-defined, nodular mass located at the porta hepatis, with findings similar to those of some malignant papillary neoplasms of the intrahepatic bile duct. Therefore, in some tumors located near the porta hepatis, it may be difficult or impossible to differentiate papillary cholangiocarcinoma from a nodular growing type of ductal adenocarcinoma.
Kukubo et al. [9] described five patients with mucin-hypersecreting intrahepatic biliary neoplasms and one with papillary cholangiocarcinoma. According to their report, the most characteristic appearance of this tumor on CT was marked dilatation of the intra- and extrahepatic bile ducts distal to the hepatic tumor. However, these researchers could not identify papillary tumor per se in the duct on CT. Conversely, we found that CT could reveal an intraductal hypoattenuating mass or nodule associated with markedly dilated intra- and extrahepatic bile ducts in our three patients with mucin-hypersecreting papillary cholangiocarcinoma.
Prompt and accurate detection of biliary tract carcinoma is essential for optimal treatment. A potential benefit of early diagnosis is suggested by a report of more favorable outcomes in patients with early bile duct cancer [10]. Our results show that by using CT, tumors were detected in 13 (87%) of 15 patients. On CT, most tumors appeared as intraductal hypoattenuating lesions with the dilated bile duct proximal to the tumor. Kawakatsu et al. [2] described the same CT features we found. However, in limited cases, Kawakatsu et al. reported cholangiographic findings including a filling defect with or without stricture of the intrahepatic bile duct, and a few tumors appeared with irregular, beaded patterns of stricture of the intrahepatic bile duct on cholangiography; these findings are not expected on other imaging techniques such as CT or sonography [2]. In this respect, a combination of CT and cholangiography seems to be the most effective means for detecting this tumor and evaluating the extent of disease. Moreover, recent advances in cholangioscopy enable physicians to make a diagnosis of papillary tumors on the basis of endoscopic findings and biopsy [11, 12].
In summary, understanding the CT and pathologic features of malignant papillary tumors of the intrahepatic bile duct is important because this disease has a better prognosis than other types of intrahepatic cholangiocarcinoma. We propose using CT for revealing intraductal lesions; however, the findings are nonspecific and variable. When intraductal masses or nodules are seen with localized dilatation of the intrahepatic bile duct on CT, malignant papillary tumor of the intrahepatic bile duct should be included in the differential diagnosis.
Acknowledgments
We thank Yuji Itai, Clinical Institute of Medicine, University of Tsukuba,
Ibaraki, Japan, for his critical review of this manuscript and useful
discussion. We also thank Bonnie Hami, Department of Radiology, University
Hospitals of Cleveland, Cleveland, OH, for her editorial assistance in
preparing this manuscript.
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