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A Comparison of Hepatic Mucinous Cystic Neoplasms with Biliary Intraductal Papillary Neoplasms

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A Comparison of Hepatic Mucinous Cystic Neoplasms with Biliary Intraductal Papillary Neoplasms 中国科技论文在线 http://www.paper.edu.cn CLINICAL GASTROENTEROLOGY AND HEPATOLOGY 2009;7:586–593 A Comparison of Hepatic Mucinous Cystic Neoplasms With Biliary Intraductal Papillary Neoplasms TAO LI,* YUAN JI,‡ XU–TING ZHI,* LU WANG,§ XIN–RONG YANG,§ GUO–MING SHI,§ WEI ZHANG,§ and ZHAO–YOU TANG§ *Department of General Surgery, Qilu Hospital, Shandong University, Jinan, China; ‡Department of Pathology and §Liver Cancer Institute, Zhongshan Hospital, Fudan University, Shanghai, China Background & Aims:: There is controversy regarding pattern, commonly an overproduction of mucin and an asso- the term biliary intraductal papillary neoplasms (IPN-B) ciation with invasive adenocarcinoma.7–10 Therefore, some in- and their pathology, which frequently are confused with vestigators recommended the term biliary intraductal papillary hepatic mucinous cystic neoplasms (MCN). We aimed to neoplasms to be adopted in the study of biliary cystic tu- summarize the clinicopathologic features of IPN-B and dif- mors,7,11 in analogy to their pancreatic counterpart, to avoid ferentiate them from MCN. Methods: From January 1998 confusion with hepatic MCN. As in the pancreas, the presence to December 2007, there were 19 patients with intrahepatic of ovarian-like stroma is required to establish the diagnosis of IPN-B and 13 patients with MCN who underwent surgical hepatic MCN and its distinction from IPN-B.10 That is, biliary treatment at Zhongshan Hospital. Multiple demographic cystadenoma or cystadenocarcinoma should be restricted to and clinicopathologic parameters were reviewed retrospec- true cystic neoplasms with ovarian-like stroma, and most cases tively and compared between the groups. Results: The previously reported as biliary cystadenoma or cystadenocarci- mean ages of patients with IPN-B and MCN were 59.5 ؎ noma without ovarian-like stroma,12 which is considered to .the arise from bile ducts,13 are now thought to be IPN-B ;(0004. ؍ and 44.4 ؎ 9.7 years, respectively (P 11.1 ,Currently, great attention is being drawn to IPN-B. However .(028. ؍ male:female ratios also differed (11:8 vs 2:11; P with only a few cases of IPN-B reported, there continues to be (006. ؍ Tumors were significantly smaller (6.0 vs 11.2 cm; P in patients with IPN-B than in those with MCN. More inadequate knowledge available to understand the tumor and patients with IPN-B also had hepatolithiasis (47.4% vs 0%, to distinguish its intrahepatic form from other biliary cystic cholangiectasis and communication between tumors, especially MCN. In the current study we present a ;(004. ؍ P the cyst and main bile duct were more frequent in pa- single-institution, retrospective analysis of the clinical and ra- tients with IPN-B than in those with MCN (P < .001). diologic presentation, treatment, pathologic features, and long- The IPN-B consisted of 4 subtypes—the gastric subtype term outcomes of patients with intrahepatic IPN-B. was the least invasive. Malignant lesions were more com- mon in patients with IPN-B than in those with MCN Patients and Methods The overall 5-year survival rates A retrospective review was made of the medical records .(03. ؍ vs 38.5%; P 78.9%) of patients with IPN-B and MCN were 82% and 100%, of all patients seen from January 1998 to December 2007 with respectively. Conclusions: Intrahepatic IPN-B repre- histologically proven IPN-B in Zhongshan Hospital, Fudan sents a distinct clinicopathologic entity that differs clini- University. IPN-B was defined microscopically as an intraductal cally, histologically, and radiologically from MCN. Curative papillary growth of neoplastic biliary epithelia with fine fibro- resection has a favorable prognosis for patients with IPN-B, vascular cores in the lumen of the biliary tree,8 and the absence but further studies of its subtype are required. of an ovarian-type stroma.10 Noninvasive tumors were classified as adenoma, borderline, or carcinoma in situ depending on the de- gree of epithelial dysplasia within the specimen. Invasive tumors ntraductal papillary neoplasms (adenocarcinoma/adenoma) were classified as tubular, colloid, or mixed-type carcinoma. Iand mucinous cystic neoplasms (MCNs) (cystadenocarcinoma/ Information including demographics, clinical history and cystadenoma) are 2 types of mucin-producing neoplasms of the presentation, diagnostic work-up, type of surgical procedure, bile duct. In contrast to the well-known MCN, biliary intraduc- details of histology, hospital course, and follow-up evaluation tal papillary neoplasm (IPN-B), which has been reported previ- were obtained and compared with those of 13 concurrent pa- ously in the literature under various terms, such as biliary papillomatosis,1 bile duct papillomatosis,2 mucin-hypersecret- ing cholangiocarcinoma,3 or mucin-hypersecreting bile duct Abbreviations used in this paper: CA 19-9, carbohydrate antigen tumor,4 is a newly recognized entity and its intrahepatic form 19-9; CT, computed tomography; ERCP, endoscopic retrograde cholan- usually is confused with MCN. giopancreatography; IDUS, intraductal ultrasonography; IPN-B, biliary Recent studies have revealed that IPN-B has striking similar- intraductal papillary neoplasm; IPMN-P, pancreatic intraductal papil- lary mucinous neoplasms; MCN, mucinous cystic neoplasm; MRCP, ities with pancreatic intraductal papillary mucinous neoplasms magnetic resonance cholangiopancreatography; MRI, magnetic reso- (IPMN-P), which can be distinguished from pancreatic MCN by nance imaging; US, ultrasound. the absence of ovarian-like stroma and communication with © 2009 by the AGA Institute the pancreatic duct.5,6 Both IPN-B and IPMN-P arise within the 1542-3565/09/$36.00 duct system and show a predominantly intraductal growth doi:10.1016/j.cgh.2009.02.019 转载 中国科技论文在线 http://www.paper.edu.cn May 2009 BILIARY INTRADUCTAL PAPILLARY NEOPLASM 587 Table 1. Demographics and Clinical Characteristics of IPN-B Results and MCN Clinical Characteristics Characteristics IPN-B (n ϭ 19) MCN (n ϭ 13) P value There were 19 cases of intrahepatic IPN-B and 13 cases of Demographic data MCN. Clinical characteristics are described in Table 1. The mean Mean age, y 59.5 Ϯ 11.1 44.4 Ϯ 9.7 .0004 age of patients with IPN-B was 59.5 Ϯ 11.1 years (median, 60 y; Median age, y 60 43 range, 30–76 y) and the male:female ratio was 11:8, which was Sex, male:female 11:8 2:11 .028 quite different from those patients with MCN, for whom the mean Clinical manifestations age was 44.4 Ϯ 9.7 years (median, 43 y; range, 28–60 y; P ϭ .0004), Abdominal pain 6 (31.6%) 5 (38.5%) NS and female predominance was marked (M:F ratio, 2:11; P ϭ .028). Jaundice 1 (5.3%) 0 NS Overall, more than half of the patients of both groups were Weight loss 1 (5.3%) 0 NS asymptomatic and the neoplasms were discovered incidentally. Acute cholangitis 1 (5.3%) 0 NS There were no significant differences between IPN-B and MCN No symptoms 10 (52.6%) 8 (61.5%) NS ϭ Associated diseases with respect to clinical presentation (P NS), and the most Hepatolithiasis 9 (47.4%) 0 .004 common clinical manifestation at admission was epigastric Clonorchiasis 1 (5.3%) 0 NS pain. But IPN-B was associated more commonly with hepato- Hepatitis 0 2 (15.4%) NS lithiasis than MCN (47.4% vs 0%; P ϭ .004). Cirrhosis 1 (5.3%) 1 (7.7%) NS Both IPN-B and MCN seldom happened in patients with None 8 (42.1%) 10 (76.9%) NS hepatitis or cirrhosis, and serum carcinoembryonic antigen and Mean serum CA 19-9 50.2 (2.8–159) 251.3 (0.6–1434) NS ␣-fetoprotein levels were normal, except that the serum carbo- level, U/mL (range) hydrate antigen 19-9 (CA 19-9) level was increased in 42.1% of IPN-B patients and in 46.2% of MCN patients. However, the increase of CA 19-9 level was correlated to neither the progres- ϭ tients with MCN. All patients had follow-up evaluations con- sion nor the prognosis of both tumors (P NS). sisting of a clinical examination, serologic assessment of tumor markers and imaging, which included abdominal ultrasound Radiologic Findings (US), computed tomography (CT), magnetic resonance imaging US was performed in all IPN-B and MCN patients and (MRI), or magnetic resonance cholangiopancreatography (MRCP). revealed multilocular or unilocular lesions. Papillary projections or In all patients, recurrences were confirmed histologically. masses along the walls were detected in 18 IPN-B patients (Figure All continuous data are presented at mean Ϯ standard devia- 1A), including 3 hypoechoic, 9 hyperechoic, and 6 hyperechoic and tion. Categoric variables were compared using the Pearson chi- hypoechoic mixed masses, but were not detected in most MCN square test and the Fisher exact test when cell counts were less patients (94.7% vs 30.8%; P ϭ .0002), which usually were misdiag- than 5. The Student t test was used for all comparisons among nosed as simple cysts. Marked dilatation of the intrahepatic bile continuous variables. Significance was accepted at the 5% level. duct was detected in 14 IPN-B patients (Figure 1B), but in none of Figure 1. US findings of IPN-B. (A) Cystic lesion with echogenic projec- tions along the cyst walls on US. (B) US showed multilocular cystic lesions with marked dilatation of the bile duct (arrows). (C) US showed unilocular cystic lesion communicated with bile ducts (arrows). (D) On cholangiogra- phy, the dilated right bile ducts re- vealed multiple, amorphous filling de- fects (arrows) and ragged irregularity of the bile duct wall. (E) Choledocho- scope showed pinkish or red multiple papillary masses scattered within the bile duct. Panels D and E are from the same IPN-B patient. 中国科技论文在线 http://www.paper.edu.cn 588 LI ET AL CLINICAL GASTROENTEROLOGY AND HEPATOLOGY Vol. 7, No. 5 Figure 2. CT findings of IPN-B.
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