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Mocchegiani F, Vincenzi P, Conte G, Nicolini D, Rossi R, Cacciaguerra AB, Vivarelli M. Intraductal papillary neoplasm of the bile duct: The new frontier of biliary pathology. World J Gastroenterol 2023; 29:5361-5373. [PMID: 37900587 PMCID: PMC10600795 DOI: 10.3748/wjg.v29.i38.5361] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/31/2023] [Revised: 08/07/2023] [Accepted: 08/31/2023] [Indexed: 10/12/2023] Open
Abstract
Intraductal papillary neoplasms of the bile duct (IPNBs) represent a rare variant of biliary tumors characterized by a papillary growth within the bile duct lumen. Since their first description in 2001, several classifications have been proposed, mainly based on histopathological, radiological and clinical features, although no specific guidelines addressing their management have been developed. Bile duct neoplasms generally develop through a multistep process, involving different precursor pathways, ranging from the initial lesion, detectable only microscopically, i.e. biliary intraepithelial neoplasia, to the distinctive grades of IPNB until the final stage represented by invasive cholangiocarcinoma. Complex and advanced investigations, mainly relying on magnetic resonance imaging (MRI) and cholangioscopy, are required to reach a correct diagnosis and to define an adequate bile duct mapping, which supports proper treatment. The recently introduced subclassifications of types 1 and 2 highlight the histopathological and clinical aspects of IPNB, as well as their natural evolution with a particular focus on prognosis and survival. Aggressive surgical resection, including hepatectomy, pancreaticoduodenectomy or both, represents the treatment of choice, yielding optimal results in terms of survival, although several endoscopic approaches have been described. IPNBs are newly recognized preinvasive neoplasms of the bile duct with high malignant potential. The novel subclassification of types 1 and 2 defines the histological and clinical aspects, prognosis and survival. Diagnosis is mainly based on MRI and cholangioscopy. Surgical resection represents the mainstay of treatment, although endoscopic resection is currently applied to nonsurgically fit patients. New frontiers in genetic research have identified the processes underlying the carcinogenesis of IPNB, to identify targeted therapies.
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Affiliation(s)
- Federico Mocchegiani
- Department of Experimental and Clinical Medicine, Polytechnic University of Marche, Ancona 60126, Italy
| | - Paolo Vincenzi
- Department of Gastroenterology and Transplant, United Hospital of Marche, Ancona 60126, Italy
| | - Grazia Conte
- Department of Gastroenterology and Transplant, United Hospital of Marche, Ancona 60126, Italy
| | - Daniele Nicolini
- Department of Gastroenterology and Transplant, United Hospital of Marche, Ancona 60126, Italy
| | - Roberta Rossi
- Department of Gastroenterology and Transplant, United Hospital of Marche, Ancona 60126, Italy
| | | | - Marco Vivarelli
- Department of Experimental and Clinical Medicine, Polytechnic University of Marche, Ancona 60126, Italy
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Xiao G, Xia T, Mou YP, Zhou YC. Reoperation for heterochronic intraductal papillary mucinous neoplasm of the pancreas after bile duct neoplasm resection: A case report. World J Gastrointest Surg 2023; 15:1542-1548. [PMID: 37555129 PMCID: PMC10405119 DOI: 10.4240/wjgs.v15.i7.1542] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/24/2023] [Revised: 04/28/2023] [Accepted: 05/11/2023] [Indexed: 07/21/2023] Open
Abstract
BACKGROUND Intraductal papillary neoplasm of the bile duct (IPNB) and intraductal papillary mucinous neoplasm (IPMN) of the pancreas have similar pathological manifestations. However, they often develop separately and it is rare for both to occur together. Patients presenting with heterochronic IPMN after IPNB are prone to be misdiagnosed with tumor recurrence. CASE SUMMARY A 67-year-old male patient was admitted 8.5 years after IPNB carcinoma and 4 years after the discovery of a pancreatic tumor. A left hepatic bile duct tumor with distal bile duct dilatation was found 8.5 years ago by the computed tomography; therefore, a left hepatectomy was performed. The postoperative pathological diagnosis was malignant IPNB with negative cutting edge and pathological stage T1N0M0. Magnetic resonance imaging 4 years ago showed cystic lesions in the pancreatic head with pancreatic duct dilatation, and carcinoembryonic antigen continued to increase. Positron emission tomography showed a maximum standard uptake value of 11.8 in the soft tissue mass in the pancreatic head, and a malignant tumor was considered. Radical pancreatoduodenectomy was performed. Postoperative pathological diagnosis was pancreatic head IPMN with negative cutting edge, pancreaticobiliary type, stage T3N0M0. He was discharged 15 d after the operation. Follow-up for 6 mo showed no tumor recurrence, and quality of life was good. CONCLUSION IPNB and IPMN are precancerous lesions with similar pathological characteristics and require active surgery and long-term follow-up.
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Affiliation(s)
- Gang Xiao
- Department of Hepatopancreatobiliary Surgery, Shaoxing People’s Hospital, Shaoxing 312000, Zhejiang Province, China
| | - Tao Xia
- Division of Gastrointestinal and Pancreatic Surgery, Department of General Surgery, Zhejiang Provincial People’s Hospital (People’s Hospital of Hangzhou Medical College), Key Laboratory of Gastroenterology of Zhejiang Province, Hangzhou 310014, Zhejiang Province, China
| | - Yi-Ping Mou
- Division of Gastrointestinal and Pancreatic Surgery, Department of General Surgery, Zhejiang Provincial People’s Hospital (People’s Hospital of Hangzhou Medical College), Key Laboratory of Gastroenterology of Zhejiang Province, Hangzhou 310014, Zhejiang Province, China
| | - Yu-Cheng Zhou
- Division of Gastrointestinal and Pancreatic Surgery, Department of General Surgery, Zhejiang Provincial People’s Hospital (People’s Hospital of Hangzhou Medical College), Key Laboratory of Gastroenterology of Zhejiang Province, Hangzhou 310014, Zhejiang Province, China
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Fukuya H, Kuwano A, Nagasawa S, Morita Y, Tanaka K, Yada M, Masumoto A, Motomura K. Multicentric recurrence of intraductal papillary neoplasm of bile duct after spontaneous detachment of primary tumor: A case report. World J Clin Cases 2022; 10:1000-1007. [PMID: 35127913 PMCID: PMC8790441 DOI: 10.12998/wjcc.v10.i3.1000] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/22/2021] [Revised: 07/07/2021] [Accepted: 12/23/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Intraductal papillary neoplasm of the bile duct (IPNB) rarely recurs in a multicentric manner. We encountered a patient with multiple recurrences of the gastric subtype of IPNB one year after spontaneous detachment of the primary tumor during peroral cholangioscopy (POCS).
CASE SUMMARY A 68-year-old woman on maintenance hemodialysis because of lupus nephritis had several cardiovascular diseases and a pancreatic intraductal papillary mucinous neoplasm (IPMN). She was referred to our department for dilation of the common bile duct (CBD) and a tumor in the lumen, detected using ultrasonography. She had no complaints, and blood tests of hepatobiliary enzymes were normal. Magnetic resonance cholangiopancreatography (MRCP) showed a papillary tumor in the CBD with a filling defect detected using endoscopic retrograde cholangiography (ERC). Intraductal ultrasonography revealed a papillary tumor and stalk at the CBD. During POCS, the tumor spontaneously detached with its stalk into the CBD. Pathology showed low-intermediate nuclear atypia of the gastric subtype of IPNB. After 1 year, follow-up MRCP showed multiple tumors distributed from the left hepatic duct to the CBD. ERC and POCS showed multicentric tumors. She was alive without hepatobiliary symptoms at least two years after initial diagnosis of IPNB.
CONCLUSION The patient experienced gastric subtype of IPNB without curative resection. Observation may be reasonable for patients with this subtype.
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Affiliation(s)
- Hiroki Fukuya
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
| | - Akifumi Kuwano
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
| | - Shigehiro Nagasawa
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
| | - Yusuke Morita
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
| | - Kosuke Tanaka
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
| | - Masayoshi Yada
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
| | - Akihide Masumoto
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
| | - Kenta Motomura
- Department of Hepatology, Iizuka Hospital, Iizuka 820-8505, Fukuoka, Japan
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Kitahama T, Yamane H, Mohri K, Fukuoka E, Yoshida T, Yamagishi T, Goto H, Furutani A, Otsubo D, Matsumoto T, Tanaka M, Fujino Y, Tominaga M. A case of intraductal papillary neoplasm of the bile duct accompanied by intraductal papillary mucinous neoplasm of the pancreas and hepatocellular carcinoma. Clin J Gastroenterol 2021; 14:1536-1543. [PMID: 34106396 DOI: 10.1007/s12328-021-01461-z] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/09/2021] [Accepted: 06/03/2021] [Indexed: 12/20/2022]
Abstract
A 73-year-old man with mixed-type intraductal papillary mucinous neoplasm of the pancreas body was followed up for 14 years. Based on imaging findings, the intraductal papillary mucinous neoplasm of the pancreas met the high-risk stigmata, and new hepatic masses were suspected to be intraductal papillary neoplasms of the bile duct. With a diagnosis of intraductal papillary mucinous neoplasm of the pancreas and intraductal papillary neoplasm of the bile duct, the patient had undergone left lateral hepatectomy and distal pancreatectomy. Based on pathology, the pancreatic specimen was diagnosed as a high-grade intraductal papillary mucinous neoplasm of the pancreas, and the hepatic specimen was diagnosed as an intraductal papillary neoplasm of the bile duct and hepatocellular carcinoma. The intraductal papillary neoplasms of the bile duct and hepatocellular carcinoma were adjacent to each other. Fifteen months after surgery, recurrence in the remnant pancreas was detected. The patient had undergone residual total pancreatectomy, with no recurrence thirty months after the second resection. This case demonstrates that second surgery for metachronous high-risk lesions in the remnant pancreas of patients with intraductal papillary mucinous neoplasm of the pancreas and intraductal papillary neoplasm of the bile duct may also be considered to improve survival.
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Affiliation(s)
- Takumi Kitahama
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan.
| | - Hisoka Yamane
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Koichi Mohri
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Eiji Fukuoka
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Toshihiko Yoshida
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Takashi Yamagishi
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Hironobu Goto
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Akinobu Furutani
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Dai Otsubo
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Taku Matsumoto
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Motofumi Tanaka
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Yasuhiro Fujino
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
| | - Masahiro Tominaga
- Division of Gastroenterological Surgery, Hyogo Cancer Center, 13-70 Kitaoji-cho, Akashi, Hyogo, 673-8558, Japan
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Ren X, Zhu CL, Qin XF, Jiang H, Xia T, Qu YP. Co-occurrence of IPMN and malignant IPNB complicated by a pancreatobiliary fistula: A case report and review of the literature. World J Clin Cases 2019; 7:102-108. [PMID: 30637259 PMCID: PMC6327132 DOI: 10.12998/wjcc.v7.i1.102] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/27/2018] [Revised: 12/06/2018] [Accepted: 12/08/2018] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Intraductal papillary neoplasm of the bile duct (IPNB) is pathologically similar to intraductal papillary mucinous neoplasm (IPMN). However, there are several significant differences between them. The rate of IPMN associated with extrapancreatic malignancies has been reported to range from 10%-40%, and it may occasionally be complicated with the presence of fistulas. IPMN associated with malignant IPNB is extremely rare and only nine cases have been reported in the literature.
CASE SUMMARY We report a 52-year-old man who presented with recurrent cholangitis for nine months. Computed tomography and magnetic resonance cholangiopancreatography showed the common bile duct stricture with dilated pancreatobiliary duct without other abnormal findings. The underlying pathogenesis could not be identified based on the radiologic images. Endoscopic retrograde cholangiopancreatography revealed a pancreatobiliary fistula with dilated main pancreatic duct, biliary stricture with dilated biliary tree, and mucus discharge from the enlarged orifice of the major papilla. The patient underwent SpyGlass cholangiopancreatoscopy due to a suspected mucin-producing biliary neoplasm and indeterminate main pancreatic duct dilatation. Multiple papillary growing neoplasms with vascular images, with the extent of lesions spreading in the biliopancreatic ductal lumens, were identified by SpyGlass. In addition, the presence of a pancreatobiliary fistula was also identified. The patient was diagnosed as having benign IPMN and malignant IPNB with focal invasion by postoperative pathology. Furthermore, varying histological subtypes were present in both IPMN and IPNB. Pylorus-preserving pancreaticoduodenectomy was performed on the patient with excellent results during the 52 month follow-up period.
CONCLUSION We deemed that pancreatography and SpyGlass allowed for an efficient diagnosis of IPMN with pancreatobiliary fistula, whereas the etiology could not be identified by radiologic imaging.
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Affiliation(s)
- Xu Ren
- Digestive Disease Hospital of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Province, China
| | - Chun-Lan Zhu
- The Second Digestive Department, Digestive Disease Hospital of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Provice, China
| | - Xu-Fu Qin
- The Second Digestive Department, Digestive Disease Hospital of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Provice, China
| | - Hong Jiang
- Department of Pathology of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Provice, China
| | - Tian Xia
- Digestive Disease Hospital of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Province, China
| | - Yong-Ping Qu
- Digestive Endoscopy Center, Digestive Disease Hospital of Heilongjiang Provincial Hospital, Harbin 150001, Heilongjiang Provice, China
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Tong A, Veillette G, Budhai A, Gilet A. Intraductal oncocytic papillary neoplasm: a benign hepatic cystic neoplasm. BMJ Case Rep 2017; 2017:bcr-2016-218139. [PMID: 28438794 DOI: 10.1136/bcr-2016-218139] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
A 61-year-old Asian man presented with severe right upper quadrant pain which had been worsening for several months. Laboratory results indicated elevated aspartate aminotransferase and alanine aminotransferase. The subsequent ultrasound, CT and MRI showed a large cystic mass with solid components and severe intrahepatic and extrahepatic biliary ductal dilatation. The mass was resected and pathology showed a well-demarcated mucinous cystic lesion with polygonal cells containing ovoid nuclei and abundant pink oncocytic cytoplasm. These findings are characteristic of intraductal oncocytic papillary neoplasm (IOPN), a rare subtype of intraductal papillary neoplasm of the liver. IOPN occurs most frequently in the Asian regions endemic to clonorchiasis and hepatolithiasis; however, cases have been reported in the Western world as well. Patients with IOPN are male or female and typically middle-aged. Treatment is typically resection. Prognosis is favourable and recurrence is rare. The 5-year survival rate is upwards of 80%.
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Affiliation(s)
- Angela Tong
- Radiology, Westchester Medical Center, New York Medical Center, Valhalla, New York, USA
| | - Gregory Veillette
- Transplant Surgery, Westchester Medical Center, New York Medical Center, Valhalla, New York, USA
| | | | - Anthony Gilet
- Radiology, Westchester Medical Center, Valhalla, New York, USA
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Jang KT, Ahn S. Tumoral Versus Flat Intraepithelial Neoplasia of Pancreatobiliary Tract, Gallbladder, and Ampulla of Vater. Arch Pathol Lab Med 2016; 140:429-36. [DOI: 10.5858/arpa.2015-0319-ra] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Context.—The identification of a precursor lesion is important to understanding the histopathologic and genetic alterations in carcinogenesis. There are a plethora of terminologies that describe precursor lesions of the pancreatobiliary tract, ampulla of Vater, and gallbladder. The current terminologies for precursor lesions may make it difficult to understand the tumor biology. Here, we propose the concept of tumoral and flat intraepithelial neoplasia to improve our understanding of precursor lesions of many epithelial organs, including the pancreatobiliary tract, ampulla of Vater, and gallbladder.
Objective.—To understand the dichotomous pattern of tumoral and flat intraepithelial neoplasia in carcinogenesis of pancreatobiliary tract, ampulla of Vater, and gallbladder.
Data Sources.—Review of relevant literatures indexed in PubMed.
Conclusions.—Tumoral intraepithelial neoplasia presents as an intraluminal or intraductal, mass-forming, polypoid lesion or a macroscopic, visible, cystic lesion without intracystic papillae. Microscopically, tumoral intraepithelial neoplasia shows various proportions of papillary and tubular architecture, often with a mixed pattern, such as papillary, tubular, and papillary-tubular. The malignant potential depends on the degree of dysplasia and the cell phenotype of the epithelium. Flat intraepithelial neoplasia presents as a flat or superficial, spreading, mucosal lesion that is frequently accompanied by an invasive carcinoma. Tumoral and flat intraepithelial neoplasias are not homogeneous entities and may exhibit histopathologic spectrum changes and different genetic profiles. Although intraepithelial neoplasia showed a dichotomous pattern in the tumoral versus flat types, they can coexist. Tumoral and flat intraepithelial neoplasia can be interpreted as part of a spectrum of changes in the carcinogenesis pathway of each organ.
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Affiliation(s)
| | - Sangjeong Ahn
- From the Department of Pathology and Translational Genomics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea (Dr Jang); and the Department of Pathology, Pusan National University Hospital and the Pusan National University School of Medicine, and the Biomedical Research Institute, Pusan National University Hospital, Pusan, Korea (Dr Ahn)
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Bansal A, Thung SN, Zhu H, Schwartz M, Lewis S. Synchronous pancreatic adenocarcinoma and intrahepatic cholangiocarcinoma arising in the context of intraductal papillary neoplasms. Clin Imaging 2016; 40:897-901. [PMID: 27183137 DOI: 10.1016/j.clinimag.2015.12.019] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/05/2015] [Accepted: 12/04/2015] [Indexed: 02/07/2023]
Abstract
Field cancerization theory provides rationale for the development of multiple pancreatic ductal and biliary lesions in a single patient through the development and accumulation of multiple genetic changes. Genetic alterations result in the development of precursor lesions including intraductal papillary mucinous neoplasms of the pancreas (IPMN), intraductal papillary neoplasm of the bile duct (IPN-B), and their malignant counterparts, pancreatic adenocarcinoma and cholangiocarcinoma. Although IPMN are frequently encountered, IPN-B are uncommon and the synchronous occurrence of both lesions is rare. We present a case of synchronous pancreatic adenocarcinoma and intrahepatic cholangiocarcinoma with histopathologic evidence of underlying precursor lesions, IPMN-P and IPN-B.
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Affiliation(s)
- Anmol Bansal
- Department of Radiology, Icahn School of Medicine at Mount Sinai, One Gustave Levy Place, New York, NY 10029
| | - Swan N Thung
- The Lillian and Henry Stratton-Hans Popper Department of Pathology, Icahn School of Medicine at Mount Sinai, One Gustave Levy Place, New York, NY 10029
| | - Hongfa Zhu
- The Lillian and Henry Stratton-Hans Popper Department of Pathology, Icahn School of Medicine at Mount Sinai, One Gustave Levy Place, New York, NY 10029
| | - Myron Schwartz
- Department of Surgery, RMTI Liver Surgery Program, Icahn School of Medicine at Mount Sinai, One Gustave Levy Place, New York, NY 10029
| | - Sara Lewis
- Department of Radiology, Icahn School of Medicine at Mount Sinai, One Gustave Levy Place, New York, NY 10029.
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Simultaneous Extensive Intraductal Papillary Neoplasm of the Bile Duct and Pancreas: A Very Rare Entity. Case Rep Surg 2016; 2016:1518707. [PMID: 26925284 PMCID: PMC4746402 DOI: 10.1155/2016/1518707] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/25/2015] [Revised: 01/07/2016] [Accepted: 01/10/2016] [Indexed: 12/11/2022] Open
Abstract
Intraductal papillary neoplasm of the bile duct (IPNB) is a specific type of bile duct tumor. It has been proposed that it could be the biliary counterpart of the intraductal papillary neoplasm of the pancreas (IPMN-P). This hypothesis is supported by the presence of simultaneous intraductal tumors of both the bile duct and pancreas. There have been five reports of patients with simultaneous IPNB and IPMN-P. In all of these cases, biliary involvement was limited to the intrahepatic and perihilar bile duct, which had characteristics similar to IPMN-P and usually had slow progression in nature. Herein, we present the first case of extensive intraductal neoplasm involving the extrahepatic bile duct, intrahepatic bile duct, and entire length of the pancreas with a poor outcome, even after being treated aggressively with radical surgery and adjuvant chemotherapy. Additionally, we summarize previous case reports of simultaneous intraductal lesions of the bile duct and pancreas.
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Grillo F, Guadagno A, Rimini E, Minetti G, Mastracci L. Pancreatobiliary intraductal papillary neoplasms: what is in a name? Int J Surg Pathol 2015; 23:290-291. [PMID: 25782732 DOI: 10.1177/1066896915576960] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Affiliation(s)
- Federica Grillo
- University of Genoa, Genoa, Italy IRCCS AUO San Martino IST, Genoa, Italy
| | - Antonio Guadagno
- University of Genoa, Genoa, Italy IRCCS AUO San Martino IST, Genoa, Italy
| | | | | | - Luca Mastracci
- University of Genoa, Genoa, Italy IRCCS AUO San Martino IST, Genoa, Italy
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Moon DB, Lee SG, Jung DH, Park GC, Park YH, Park HW, Kim MH, Lee SK, Yu ES, Kim JH. Synchronous Malignant Intraductal Papillary Mucinous Neoplasms of the Bile Duct and Pancreas Requiring Left Hepatectomy and Total Pancreatectomy. THE KOREAN JOURNAL OF GASTROENTEROLOGY 2014; 63:129-33. [DOI: 10.4166/kjg.2014.63.2.129] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/27/2022]
Affiliation(s)
- Deok-Bog Moon
- Hepatobiliary Surgery and Liver Transplantation, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Sung-Gyu Lee
- Hepatobiliary Surgery and Liver Transplantation, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Dong-Hwan Jung
- Hepatobiliary Surgery and Liver Transplantation, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Gil-Chun Park
- Hepatobiliary Surgery and Liver Transplantation, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Yo-Han Park
- Hepatobiliary Surgery and Liver Transplantation, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Hyung-Woo Park
- Hepatobiliary Surgery and Liver Transplantation, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Myung-Hwan Kim
- Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Sung-Koo Lee
- Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Eun-Sil Yu
- Department of Diagnostic Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Ji-Hoon Kim
- Department of Diagnostic Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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Xu XW, Li RH, Zhou W, Wang J, Zhang RC, Chen K, Mou YP. Laparoscopic resection of synchronous intraductal papillary mucinous neoplasms: A case report. World J Gastroenterol 2012; 18:6510-6514. [PMID: 23197900 PMCID: PMC3508649 DOI: 10.3748/wjg.v18.i44.6510] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/25/2012] [Accepted: 09/28/2012] [Indexed: 02/06/2023] Open
Abstract
We describe herein a 68-year-old woman who was diagnosed with a quite rare entity of intraductal papillary mucinous neoplasms (IPMNs) occurring simultaneously in the left lateral lobe of liver and the tail of pancreas. Abdominal computed tomography and magnetic resonance cholangiopancreatography showed a cystic dilatation of the pancreatic duct in the pancreatic tail, which suggested an IPMN, and multiple intrahepatic duct stones in the left lateral lobe. The patient underwent a laparoscopic left lateral hepatolobectomy and spleen-preserving distal pancreatectomy. Intra-operative finding of massive mucin in the dilated bile duct implied an intraductal mucinous tumor in the liver. The diagnosis of synchronous IPMNs in the liver and pancreas was confirmed by pathological examination. The patient was followed up for 6 mo without signs of recurrence. Although several cases of IPMN of liver without any pancreatic association have been reported, the simultaneous occurrence of IPMNs in the liver and pancreas is very rare. To the best of our knowledge, it is the first reported case treated by laparoscopic resection.
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MESH Headings
- Aged
- Biopsy
- Carcinoma, Pancreatic Ductal/pathology
- Carcinoma, Pancreatic Ductal/surgery
- Cholangiopancreatography, Magnetic Resonance
- Female
- Hepatectomy/methods
- Humans
- Laparoscopy
- Liver Neoplasms/pathology
- Liver Neoplasms/surgery
- Neoplasms, Cystic, Mucinous, and Serous/pathology
- Neoplasms, Cystic, Mucinous, and Serous/surgery
- Neoplasms, Multiple Primary/pathology
- Neoplasms, Multiple Primary/surgery
- Pancreatectomy/methods
- Pancreatic Neoplasms/pathology
- Pancreatic Neoplasms/surgery
- Tomography, X-Ray Computed
- Treatment Outcome
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Intracholecystic papillary-tubular neoplasms (ICPN) of the gallbladder (neoplastic polyps, adenomas, and papillary neoplasms that are ≥1.0 cm): clinicopathologic and immunohistochemical analysis of 123 cases. Am J Surg Pathol 2012; 36:1279-301. [PMID: 22895264 DOI: 10.1097/pas.0b013e318262787c] [Citation(s) in RCA: 163] [Impact Index Per Article: 12.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
The literature on the clinicopathologic characteristics of tumoral intraepithelial neoplasms (neoplastic polyps) of the gallbladder (GB) is fairly limited, due in part to the variability in definition and terminology. Most reported adenomas (pyloric gland type and others) were microscopic and thus regarded as clinically inconsequential, whereas papillary in situ carcinomas have been largely considered a type of invasive adenocarcinoma under the heading of "papillary adenocarcinomas." In this study, 123 GB cases that have a well-defined exophytic preinvasive neoplasm measuring ≥1 cm were analyzed. The patients were predominantly female (F/M=2:1) with a mean age of 61 y and a median tumor size of 2.2 cm. Half of the patients presented with pain, and in the other half the neoplasm was detected incidentally. Other neoplasms, most being gastrointestinal tract malignancies, were present in 22% of cases. Gallstones were identified in only 20% of cases. Radiologically, almost half were diagnosed as "cancer," roughly half with polypoid tumor, and in 10% the lesion was missed. Pathologic findings: (1) The predominant configuration was papillary in 43%, tubulopapillary in 31%, tubular in 26%. (2) Each case was assigned a final lineage type on the basis of the predominant pattern (>75% of the lesion) on morphology, and supported with specific immunohistochemical cell lineage markers. The predominant cell lineage could be identified as biliary in 50% (66% of which were MUC1), gastric foveolar in 16% (all were MUC5AC), gastric pyloric in 20% (92% MUC6), intestinal in 8% (100% CK20; 75% CDX2; 50%, MUC2), and oncocytic in 6% (17% HepPar and 17% MUC6); however, 90% of cases had some amount of secondary or unclassifiable pattern and hybrid immunophenotypes. (3) Of the cases that would have qualified as "pyloric gland adenoma," 21/24 (88%) had at least focal high-grade dysplasia and 18% had associated invasive carcinoma. Conversely, 8 of 47 "papillary adenocarcinoma"-type cases displayed some foci of low-grade dysplasia, and 15/47 (32%) had no identifiable invasion. (4) Overall, 55% of the cases had an associated invasive carcinoma (pancreatobiliary type, 58; others, 10). Factors associated significantly with invasion were the extent of high-grade dysplasia, cell type (biliary or foveolar), and papilla formation. Among systematically analyzed invasive carcinomas, tumoral intraepithelial neoplasia was detected in 6.4% (39/606). (5) The 3-year actuarial survival was 90% for cases without invasion and 60% for those associated with invasion. In contrast, those associated with invasion had a far better clinical outcome compared with pancreatobiliary-type GB carcinomas (3-yr survival, 27%), and this survival advantage persisted even with stage-matched comparison. Death occurred in long-term follow-up even in a few noninvasive cases (4/55; median 73.5 mo) emphasizing the importance of long-term follow-up. In conclusion, tumoral preinvasive neoplasms (≥1 cm) in the GB are analogous to their pancreatic and biliary counterparts (biliary intraductal papillary neoplasms, pancreatic intraductal papillary mucinous neoplasms, and intraductal tubulopapillary neoplasms). They show variable cellular lineages, a spectrum of dysplasia, and a mixture of papillary or tubular growth patterns, often with significant overlap, warranting their classification under 1 unified parallel category, intracholecystic papillary-tubular neoplasm. Intracholecystic papillary-tubular neoplasms are relatively indolent neoplasia with significantly better prognosis compared with pancreatobiliary-type GB carcinomas. In contrast, even seemingly innocuous examples such as those referred to as "pyloric gland adenomas" can progress to carcinoma and be associated with invasion and fatal outcome.
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Synchronous extrapancreatic malignant papillary mucinous neoplasms in a patient with intraductal papillary mucinous neoplasm of the pancreas: a rare case of simultaneous pancreatic, hepatic, and pulmonary involvement. Pancreas 2012; 41:501-2. [PMID: 22415674 DOI: 10.1097/mpa.0b013e3182320bf7] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/10/2022]
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Valente R, Capurso G, Pierantognetti P, Iannicelli E, Piciucchi M, Romiti A, Mercantini P, Larghi A, Federici GF, Barucca V, Osti MF, Di Giulio E, Ziparo V, Delle Fave G. Simultaneous intraductal papillary neoplasms of the bile duct and pancreas treated with chemoradiotherapy. World J Gastrointest Oncol 2012; 4:22-5. [PMID: 22403738 PMCID: PMC3296805 DOI: 10.4251/wjgo.v4.i2.22] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/30/2011] [Revised: 12/16/2011] [Accepted: 12/23/2011] [Indexed: 02/05/2023] Open
Abstract
Some authors have suggested that intraductal papillary mucinous neoplasms of the bile duct (IPMN-B) could be the the biliary counterpart of IPMN of the pancreas (IPMN-P) since they share several clinical-pathological features. These include prominent intraductal papillary proliferation pattern, a gastrointestinal phenotype, frequent mucin hyper-secretion and progression to mucinous carcinoma. To date there are just four reported cases of patients with synchronous IPMN-B and IPMN-P all of which were treated surgically. We hereby report the case of a 76-year-old woman who was incidentally diagnosed with both an asymptomatic 3 cm bulky fluid lesion obstructing the bile duct lumen, diagnosed as a malignant IPMN-B, and synchronous multiple pancreatic cystic lesions (10-13 mm) communicating with an irregular Wirsung, diagnosed as branch duct IPMN-P. Since surgery was ruled-out because of the patient’s age and preferences, she underwent a conservative management regimen comprising both chemotherapy and radiotherapy. This was effective in decreasing the mass size and in resolving subsequent jaundice. This is also the first reported case of IPMN-B successfully treated with chemoradiotherapy. Clinicians should consider medical treatment as an option in this clinical scenario, in patients who may be unfit for surgery.
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Affiliation(s)
- Roberto Valente
- Roberto Valente, Gabriele Capurso, Matteo Piciucchi, Emilio Di Giulio, Gianfranco Delle Fave, Digestive and Liver Disease Unit, Second Medical School, "La Sapienza" University, S. Andrea Hospital, 00189 Rome, Italy
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Intra-ampullary papillary-tubular neoplasm (IAPN): characterization of tumoral intraepithelial neoplasia occurring within the ampulla: a clinicopathologic analysis of 82 cases. Am J Surg Pathol 2010; 34:1731-48. [PMID: 21084962 DOI: 10.1097/pas.0b013e3181f8ff05] [Citation(s) in RCA: 64] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
BACKGROUND There has been no uniform terminology for systematic analysis of mass-forming preinvasive neoplasms (which we term tumoral intraepithelial neoplasia) that occur specifically within the ampulla. Here, we provide a detailed analysis of these neoplasms, which we propose to refer to as intra-ampullary papillary-tubular neoplasm (IAPN). MATERIALS AND METHODS Three hundred and seventeen glandular neoplasms involving the ampulla were identified through a review of 1469 pancreatoduodenectomies and 11 ampullectomies. Eighty-two neoplasms characterized by substantial preinvasive exophytic component that grew almost exclusively (>75%) within the ampulla (in the ampullary channel or intra-ampullary portions of the very distal segments of the common bile duct or pancreatic duct) were analyzed. RESULTS (1) Clinical: The mean age was 64 years, male/female ratio was 2.4, and mean tumor size was 2.7 cm. (2) Pathology: The tumors had a mixture of both papillary and tubular growth (each constituting at least 25% of the lesion) in 57%; predominantly (>75%) papillary in 23%, and predominantly (>75%) tubular in 20%. High-grade dysplasia was present in 94% of cases, of which 39% showed focal (<25% of the lesion), 28% showed substantial (25% to 75%), and 27% showed extensive (>75%) high-grade dysplasia. In terms of cell-lineage morphology, 45% had a mixture of patterns. However, when evaluated with a forced-binary approach as intestinal (INT) versus gastric/pancreatobiliary (GPB) based on the predominant pattern, 74% were classified as INT and 26% as GPB. (3) Immunohistochemistry: Percent sensitivity/specificity of cell-lineage markers were, for INT phenotype: MUC2 85/78 and CDX2 94/61; and for GBP: MUC1 89/79, MUC5AC 95/69, and MUC6 83/76, respectively. Cytokeratin 7 and 20 were coexpressed in more than half. (4) Invasive carcinoma: In 64 cases (78%), there was an associated invasive carcinoma. Size of the tumor and amount of dysplasia correlated with the incidence of invasion. Invasive carcinoma was of INT-type in 58% and of pancreatobiliary-type in 42%. Cell lineage in the invasive component was the same as that of the preinvasive component in 84%. All discrepant cases were pancreatobiliary-type invasions, which occurred in INT-type preinvasive lesions. (5) OUTCOME: The overall survival of invasive cases were significantly worse than that of noninvasive ones (57% vs. 93%; P=0.01); and 3 years, 69% versus 100% (P=0.08); and 5 years, 45% versus 100% (P=0.07), respectively. When compared with 166 conventional invasive carcinomas of the ampullary region, invasive IAPNs had significantly better prognosis with a mean survival of 51 versus 31 months (P<0.001) and the 3-year survival of 69% versus 44% (P<0.01). CONCLUSIONS Tumoral intraepithelial neoplasia occurring within the ampulla are highly analogous to pancreatic or biliary intraductal papillary and tubular neoplasms as evidenced by their papillary and/or tubular growth, variable cell lineage, and spectrum of dysplastic change (adenoma-carcinoma sequence), and thus we propose to refer to these as IAPN. IAPNs are biologically indolent; noninvasive examples show an excellent prognosis, whereas those with invasion exhibit a malignant but nevertheless significantly better prognosis than typical invasive ampullary carcinomas unaccompanied by IAPNs. Twenty eight percent (64 of 230) of invasive carcinomas within the ampulla arise in association with IAPNs.
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An uncommon etiology of cholestatic jaundice: intraductal mucinous papillary tumor of the bile duct. South Med J 2009; 102:422-4. [PMID: 19279543 DOI: 10.1097/smj.0b013e318195b239] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
Intraductal papillary mucinous tumors (IPMT) are known to occur in the pancreas but rarely can occur in the bile ducts. These tumors secrete mucin that may form mucous plugs resulting in biliary stasis, biliary duct obstruction and dilation. We report a case of IPMT of the biliary tract that presented with fatigue and a persistent cholestatic pattern of liver enzyme elevation and required a high index of suspicion to make the final diagnosis. The early recognition and treatment of these tumors is important, as unlike other bile duct tumors, these have a good prognosis.
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