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Intrahepatic Bile Duct Biliary Intraepithelial Neoplasia

MedGen UID:
474764
•Concept ID:
C3273131
•
Neoplastic Process
Synonyms: Intrahepatic Bile Duct BilIN; Intrahepatic Bile Duct Dysplasia

Definition

A neoplastic, non-invasive lesion that affects the intrahepatic bile duct epithelium. It is characterized by the presence of atypical epithelial cells with an increased nuclear/cytoplasmic ratio, nuclear hyperchromasia, and loss of nuclear polarity. [from NCI]

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  
  • CROGVIntrahepatic Bile Duct Biliary Intraepithelial Neoplasia

Professional guidelines

PubMed

Hepatic Cysts: Reappraisal of the Classification, Terminology, Differential Diagnosis, and Clinicopathologic Characteristics in 258 Cases.
Armutlu A, Quigley B, Choi H, Basturk O, Akkas G, Pehlivanoglu B, Memis B, Jang KT, Erkan M, Erkan B, Balci S, Saka B, Bagci P, Farris AB, Kooby DA, Martin D, Kalb B, Maithel SK, Sarmiento J, Reid MD, Adsay NV
Am J Surg Pathol 2022 Sep 1;46(9):1219-1233. Epub 2022 Jul 4 doi: 10.1097/PAS.0000000000001930. PMID: 35778790
A statement by the Japan-Korea expert pathologists for future clinicopathological and molecular analyses toward consensus building of intraductal papillary neoplasm of the bile duct through several opinions at the present stage.
Nakanuma Y, Jang KT, Fukushima N, Furukawa T, Hong SM, Kim H, Lee KB, Zen Y, Jang JY, Kubota K
J Hepatobiliary Pancreat Sci 2018 Mar;25(3):181-187. Epub 2018 Feb 12 doi: 10.1002/jhbp.532. PMID: 29272078
Pathogenesis of primary sclerosing cholangitis and advances in diagnosis and management.
Eaton JE, Talwalkar JA, Lazaridis KN, Gores GJ, Lindor KD
Gastroenterology 2013 Sep;145(3):521-36. Epub 2013 Jul 1 doi: 10.1053/j.gastro.2013.06.052. PMID: 23827861Free PMC Article

Recent clinical studies

Etiology

Deep learning-based phenotyping reclassifies combined hepatocellular-cholangiocarcinoma.
Calderaro J, Ghaffari Laleh N, Zeng Q, Maille P, Favre L, Pujals A, Klein C, Bazille C, Heij LR, Uguen A, Luedde T, Di Tommaso L, Beaufrère A, Chatain A, Gastineau D, Nguyen CT, Nguyen-Canh H, Thi KN, Gnemmi V, Graham RP, Charlotte F, Wendum D, Vij M, Allende DS, Aucejo F, Diaz A, Rivière B, Herrero A, Evert K, Calvisi DF, Augustin J, Leow WQ, Leung HHW, Boleslawski E, Rela M, François A, Cha AW, Forner A, Reig M, Allaire M, Scatton O, Chatelain D, Boulagnon-Rombi C, Sturm N, Menahem B, Frouin E, Tougeron D, Tournigand C, Kempf E, Kim H, Ningarhari M, Michalak-Provost S, Gopal P, Brustia R, Vibert E, Schulze K, Rüther DF, Weidemann SA, Rhaiem R, Pawlotsky JM, Zhang X, Luciani A, Mulé S, Laurent A, Amaddeo G, Regnault H, De Martin E, Sempoux C, Navale P, Westerhoff M, Lo RC, Bednarsch J, Gouw A, Guettier C, Lequoy M, Harada K, Sripongpun P, Wetwittayaklang P, Loménie N, Tantipisit J, Kaewdech A, Shen J, Paradis V, Caruso S, Kather JN
Nat Commun 2023 Dec 14;14(1):8290. doi: 10.1038/s41467-023-43749-3. PMID: 38092727Free PMC Article
Preneoplastic lesions in the liver: Molecular insights and relevance for clinical practice.
Desjonqueres E, Campani C, Marra F, Zucman-Rossi J, Nault JC
Liver Int 2022 Mar;42(3):492-506. Epub 2022 Jan 17 doi: 10.1111/liv.15152. PMID: 34982503
Impact of Remnant Carcinoma in Situ at the Ductal Stump on Long-Term Outcomes in Patients with Distal Cholangiocarcinoma.
Yasukawa K, Shimizu A, Motoyama H, Kubota K, Notake T, Fukushima K, Ikehara T, Hayashi H, Kobayashi A, Soejima Y
World J Surg 2021 Jan;45(1):291-301. Epub 2020 Sep 28 doi: 10.1007/s00268-020-05799-2. PMID: 32989580
Anatomical, histomorphological and molecular classification of cholangiocarcinoma.
Kendall T, Verheij J, Gaudio E, Evert M, Guido M, Goeppert B, Carpino G
Liver Int 2019 May;39 Suppl 1:7-18. doi: 10.1111/liv.14093. PMID: 30882996
Adult bile duct strictures: differentiating benign biliary stenosis from cholangiocarcinoma.
Nguyen Canh H, Harada K
Med Mol Morphol 2016 Dec;49(4):189-202. Epub 2016 Jun 27 doi: 10.1007/s00795-016-0143-6. PMID: 27350291

Diagnosis

Deep learning-based phenotyping reclassifies combined hepatocellular-cholangiocarcinoma.
Calderaro J, Ghaffari Laleh N, Zeng Q, Maille P, Favre L, Pujals A, Klein C, Bazille C, Heij LR, Uguen A, Luedde T, Di Tommaso L, Beaufrère A, Chatain A, Gastineau D, Nguyen CT, Nguyen-Canh H, Thi KN, Gnemmi V, Graham RP, Charlotte F, Wendum D, Vij M, Allende DS, Aucejo F, Diaz A, Rivière B, Herrero A, Evert K, Calvisi DF, Augustin J, Leow WQ, Leung HHW, Boleslawski E, Rela M, François A, Cha AW, Forner A, Reig M, Allaire M, Scatton O, Chatelain D, Boulagnon-Rombi C, Sturm N, Menahem B, Frouin E, Tougeron D, Tournigand C, Kempf E, Kim H, Ningarhari M, Michalak-Provost S, Gopal P, Brustia R, Vibert E, Schulze K, Rüther DF, Weidemann SA, Rhaiem R, Pawlotsky JM, Zhang X, Luciani A, Mulé S, Laurent A, Amaddeo G, Regnault H, De Martin E, Sempoux C, Navale P, Westerhoff M, Lo RC, Bednarsch J, Gouw A, Guettier C, Lequoy M, Harada K, Sripongpun P, Wetwittayaklang P, Loménie N, Tantipisit J, Kaewdech A, Shen J, Paradis V, Caruso S, Kather JN
Nat Commun 2023 Dec 14;14(1):8290. doi: 10.1038/s41467-023-43749-3. PMID: 38092727Free PMC Article
Intraductal papillary neoplasm of the bile duct: The new frontier of biliary pathology.
Mocchegiani F, Vincenzi P, Conte G, Nicolini D, Rossi R, Cacciaguerra AB, Vivarelli M
World J Gastroenterol 2023 Oct 14;29(38):5361-5373. doi: 10.3748/wjg.v29.i38.5361. PMID: 37900587Free PMC Article
Bile duct dysplasia and associated invasive carcinoma: clinicopathological features, diagnosis, and practical challenges.
Liu Y, Yeh MM
Hum Pathol 2023 Feb;132:158-168. Epub 2022 Jun 14 doi: 10.1016/j.humpath.2022.06.012. PMID: 35714833
Carcinoma in situ of the perihilar bile duct diagnosed by peroral cholangioscopy.
Iwano K, Kurita A, Yazumi S
J Hepatobiliary Pancreat Sci 2022 Sep;29(9):e84-e85. Epub 2022 May 11 doi: 10.1002/jhbp.1160. PMID: 35546273
Anatomical, histomorphological and molecular classification of cholangiocarcinoma.
Kendall T, Verheij J, Gaudio E, Evert M, Guido M, Goeppert B, Carpino G
Liver Int 2019 May;39 Suppl 1:7-18. doi: 10.1111/liv.14093. PMID: 30882996

Therapy

Laparoscopic Right Hemi-hepatectomy of the Bile Duct-Obstructed Area Guided by Real-Time Fluorescence Imaging.
Li X, Zeng X, Zhu W, Tao H, Yang J
Ann Surg Oncol 2024 Nov;31(12):7894-7895. Epub 2024 Aug 13 doi: 10.1245/s10434-024-16040-y. PMID: 39138774
Comparison of clinicopathological characteristics between patients with occupational and non-occupational intrahepatic cholangiocarcinoma.
Hamano G, Kubo S, Takemura S, Tanaka S, Shinkawa H, Kinoshita M, Ito T, Yamamoto T, Wakasa K, Shibata T
J Hepatobiliary Pancreat Sci 2016 Jul;23(7):389-96. Epub 2016 May 3 doi: 10.1002/jhbp.353. PMID: 27062258
Characteristics of printing company workers newly diagnosed with occupational cholangiocarcinoma.
Kubo S, Kinoshita M, Takemura S, Tanaka S, Shinkawa H, Nishioka T, Hamano G, Ito T, Abue M, Aoki M, Nakagawa K, Unno M, Hijioka S, Fujiyoshi T, Shimizu Y, Mizuguchi T, Shirabe K, Nishie A, Oda Y, Takenaka K, Kobarai T, Hisano T, Saiura A, Numao H, Toda M, Kuwae Y, Nakanuma Y, Endo G
J Hepatobiliary Pancreat Sci 2014 Nov;21(11):809-17. Epub 2014 Aug 3 doi: 10.1002/jhbp.137. PMID: 25088751
Pathogenesis of primary sclerosing cholangitis and advances in diagnosis and management.
Eaton JE, Talwalkar JA, Lazaridis KN, Gores GJ, Lindor KD
Gastroenterology 2013 Sep;145(3):521-36. Epub 2013 Jul 1 doi: 10.1053/j.gastro.2013.06.052. PMID: 23827861Free PMC Article
Precancerous bile duct pathology in end-stage primary sclerosing cholangitis, with and without cholangiocarcinoma.
Lewis JT, Talwalkar JA, Rosen CB, Smyrk TC, Abraham SC
Am J Surg Pathol 2010 Jan;34(1):27-34. doi: 10.1097/PAS.0b013e3181bc96f9. PMID: 19898228

Prognosis

Deep learning-based phenotyping reclassifies combined hepatocellular-cholangiocarcinoma.
Calderaro J, Ghaffari Laleh N, Zeng Q, Maille P, Favre L, Pujals A, Klein C, Bazille C, Heij LR, Uguen A, Luedde T, Di Tommaso L, Beaufrère A, Chatain A, Gastineau D, Nguyen CT, Nguyen-Canh H, Thi KN, Gnemmi V, Graham RP, Charlotte F, Wendum D, Vij M, Allende DS, Aucejo F, Diaz A, Rivière B, Herrero A, Evert K, Calvisi DF, Augustin J, Leow WQ, Leung HHW, Boleslawski E, Rela M, François A, Cha AW, Forner A, Reig M, Allaire M, Scatton O, Chatelain D, Boulagnon-Rombi C, Sturm N, Menahem B, Frouin E, Tougeron D, Tournigand C, Kempf E, Kim H, Ningarhari M, Michalak-Provost S, Gopal P, Brustia R, Vibert E, Schulze K, Rüther DF, Weidemann SA, Rhaiem R, Pawlotsky JM, Zhang X, Luciani A, Mulé S, Laurent A, Amaddeo G, Regnault H, De Martin E, Sempoux C, Navale P, Westerhoff M, Lo RC, Bednarsch J, Gouw A, Guettier C, Lequoy M, Harada K, Sripongpun P, Wetwittayaklang P, Loménie N, Tantipisit J, Kaewdech A, Shen J, Paradis V, Caruso S, Kather JN
Nat Commun 2023 Dec 14;14(1):8290. doi: 10.1038/s41467-023-43749-3. PMID: 38092727Free PMC Article
Intraductal papillary neoplasm of the bile duct: The new frontier of biliary pathology.
Mocchegiani F, Vincenzi P, Conte G, Nicolini D, Rossi R, Cacciaguerra AB, Vivarelli M
World J Gastroenterol 2023 Oct 14;29(38):5361-5373. doi: 10.3748/wjg.v29.i38.5361. PMID: 37900587Free PMC Article
Impact of Remnant Carcinoma in Situ at the Ductal Stump on Long-Term Outcomes in Patients with Distal Cholangiocarcinoma.
Yasukawa K, Shimizu A, Motoyama H, Kubota K, Notake T, Fukushima K, Ikehara T, Hayashi H, Kobayashi A, Soejima Y
World J Surg 2021 Jan;45(1):291-301. Epub 2020 Sep 28 doi: 10.1007/s00268-020-05799-2. PMID: 32989580
Cholangiocarcinoma: Classification, Histopathology and Molecular Carcinogenesis.
Lendvai G, Szekerczés T, Illyés I, Dóra R, Kontsek E, Gógl A, Kiss A, Werling K, Kovalszky I, Schaff Z, Borka K
Pathol Oncol Res 2020 Jan;26(1):3-15. Epub 2018 Nov 17 doi: 10.1007/s12253-018-0491-8. PMID: 30448973
Hepatobiliary and Pancreatic: Biliary intra-epithelial neoplasia.
Ho CM, Wu CH, Liau JY
J Gastroenterol Hepatol 2017 Aug;32(8):1424. doi: 10.1111/jgh.13792. PMID: 28752688

Clinical prediction guides

Clinical implications of pediatric biliary intraepithelial neoplasia diagnosed from a choledochal cyst specimen.
Gang S, Kwon H, Song IH, Namgoong JM
World J Surg Oncol 2024 Apr 20;22(1):105. doi: 10.1186/s12957-024-03384-8. PMID: 38643155Free PMC Article
Deep learning-based phenotyping reclassifies combined hepatocellular-cholangiocarcinoma.
Calderaro J, Ghaffari Laleh N, Zeng Q, Maille P, Favre L, Pujals A, Klein C, Bazille C, Heij LR, Uguen A, Luedde T, Di Tommaso L, Beaufrère A, Chatain A, Gastineau D, Nguyen CT, Nguyen-Canh H, Thi KN, Gnemmi V, Graham RP, Charlotte F, Wendum D, Vij M, Allende DS, Aucejo F, Diaz A, Rivière B, Herrero A, Evert K, Calvisi DF, Augustin J, Leow WQ, Leung HHW, Boleslawski E, Rela M, François A, Cha AW, Forner A, Reig M, Allaire M, Scatton O, Chatelain D, Boulagnon-Rombi C, Sturm N, Menahem B, Frouin E, Tougeron D, Tournigand C, Kempf E, Kim H, Ningarhari M, Michalak-Provost S, Gopal P, Brustia R, Vibert E, Schulze K, Rüther DF, Weidemann SA, Rhaiem R, Pawlotsky JM, Zhang X, Luciani A, Mulé S, Laurent A, Amaddeo G, Regnault H, De Martin E, Sempoux C, Navale P, Westerhoff M, Lo RC, Bednarsch J, Gouw A, Guettier C, Lequoy M, Harada K, Sripongpun P, Wetwittayaklang P, Loménie N, Tantipisit J, Kaewdech A, Shen J, Paradis V, Caruso S, Kather JN
Nat Commun 2023 Dec 14;14(1):8290. doi: 10.1038/s41467-023-43749-3. PMID: 38092727Free PMC Article
Simultaneous activation of Kras-Akt and Notch pathways induces extrahepatic biliary cancer via the mTORC1 pathway.
Namikawa M, Fukuda A, Mizukoshi K, Iwane K, Kawai M, Yamakawa G, Omatsu M, Sono M, Masuda T, Araki O, Nagao M, Yoshikawa T, Ogawa S, Hiramatsu Y, Muta Y, Tsuda M, Maruno T, Nakanishi Y, Tsuruyama T, Taura K, Hatano E, Seno H
J Pathol 2023 Aug;260(4):478-492. Epub 2023 Jun 13 doi: 10.1002/path.6139. PMID: 37310065
Imaging bile duct tumors: pathologic concepts, classification, and early tumor detection.
Joo I, Lee JM
Abdom Imaging 2013 Dec;38(6):1334-50. doi: 10.1007/s00261-013-0027-3. PMID: 23925840
Histological characteristics of biliary intraepithelial neoplasia-3 and intraepithelial spread of cholangiocarcinoma.
Sato Y, Harada K, Sasaki M, Nakanuma Y
Virchows Arch 2013 Apr;462(4):421-7. Epub 2013 Feb 28 doi: 10.1007/s00428-013-1384-6. PMID: 23446751

Recent systematic reviews

Pathological aspects of mucinous cholangiocarcinoma: A single-center experience and systematic review.
Laohawetwanit T, Klaikaew N
Pathol Int 2020 Sep;70(9):661-670. Epub 2020 Jul 7 doi: 10.1111/pin.12983. PMID: 32638458

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