{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,16]],"date-time":"2026-06-16T19:52:11Z","timestamp":1781639531106,"version":"3.54.5"},"reference-count":58,"publisher":"Springer Science and Business Media LLC","issue":"4","license":[{"start":{"date-parts":[[2026,2,26]],"date-time":"2026-02-26T00:00:00Z","timestamp":1772064000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2026,2,26]],"date-time":"2026-02-26T00:00:00Z","timestamp":1772064000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Ann Hematol"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Vanishing bile duct syndrome (VBDS) is a rare, and often fatal complication of Hodgkin lymphoma (HL), characterized by progressive intrahepatic bile duct loss and severe cholestasis. Management remains ill-defined, particularly in patients with refractory HL and significant hepatic dysfunction. We present a case of a young woman with biopsy-proven VBDS who experienced disease progression and worsening cholestasis despite second-line chemotherapy, corticosteroids, and brentuximab vedotin. Salvage therapy with pembrolizumab was initiated, resulting in a complete metabolic remission of HL and normalization of liver function. Notably, the patient did not experience immune-related hepatic adverse events. To our knowledge, this is the first report of HL-related VBDS successfully treated with programmed death-1 blockade. This case suggests that immune checkpoint inhibitors may be a viable therapeutic option for patients with HL-related VBDS, even in the setting of severe hepatic dysfunction.<\/jats:p>","DOI":"10.1007\/s00277-026-06745-3","type":"journal-article","created":{"date-parts":[[2026,2,26]],"date-time":"2026-02-26T08:12:18Z","timestamp":1772093538000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Resolution of vanishing bile duct syndrome in a patient associated with refractory hodgkin lymphoma following Anti\u2013PD-1 therapy: a case report and literature review"],"prefix":"10.1007","volume":"105","author":[{"given":"Yi-Ching","family":"Lin","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wan-Chen","family":"Hsieh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yu-Hsuan","family":"Tuan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hsu-Hua","family":"Tseng","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jia-Huei","family":"Tsai","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tung-Hung","family":"Su","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tai-Chung","family":"Huang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,2,26]]},"reference":[{"key":"6745_CR1","doi-asserted-by":"publisher","first-page":"99","DOI":"10.1016\/0002-9343(61)90067-5","volume":"30","author":"R Levitan","year":"1961","unstructured":"Levitan R, Diamond HD, Craver LF (1961) Jaundice in hodgkin\u2019s disease. Am J Med 30:99\u2013111. https:\/\/doi.org\/10.1016\/0002-9343(61)90067-5","journal-title":"Am J Med"},{"key":"6745_CR2","doi-asserted-by":"publisher","first-page":"872","DOI":"10.1001\/archinte.1962.03620240054009","volume":"110","author":"BA Bouroncle","year":"1962","unstructured":"Bouroncle BA, Old JW Jr., Vazques AG (1962) Pathogenesis of jaundice in Hodgkin\u2019s disease. Arch Intern Med 110:872\u201383. https:\/\/doi.org\/10.1001\/archinte.1962.03620240054009","journal-title":"Arch Intern Med"},{"issue":"1","key":"6745_CR3","doi-asserted-by":"publisher","first-page":"70","DOI":"10.1002\/hep.1840170114","volume":"17","author":"SG Hubscher","year":"1993","unstructured":"Hubscher SG, Lumley MA, Elias E (1993) Vanishing bile duct syndrome: a possible mechanism for intrahepatic cholestasis in hodgkin\u2019s lymphoma. Hepatology 17(1):70\u201377","journal-title":"Hepatology"},{"issue":"11","key":"6745_CR4","doi-asserted-by":"publisher","first-page":"636","DOI":"10.1016\/j.kjms.2013.05.002","volume":"29","author":"KM Wong","year":"2013","unstructured":"Wong KM, Chang CS, Wu CC, Yin HL (2013) Hodgkin\u2019s lymphoma-related vanishing bile duct syndrome: a case report and literature review. Kaohsiung J Med Sci 29(11):636\u2013641. https:\/\/doi.org\/10.1016\/j.kjms.2013.05.002","journal-title":"Kaohsiung J Med Sci"},{"issue":"4","key":"6745_CR5","doi-asserted-by":"publisher","first-page":"512","DOI":"10.1016\/S1470-2045(21)00005-X","volume":"22","author":"J Kuruvilla","year":"2021","unstructured":"Kuruvilla J, Ramchandren R, Santoro A, Paszkiewicz-Kozik E, Gasiorowski R, Johnson NA et al (2021) Pembrolizumab versus brentuximab vedotin in relapsed or refractory classical Hodgkin lymphoma (KEYNOTE-204): an interim analysis of a multicentre, randomised, open-label, phase 3 study. Lancet Oncol 22(4):512\u201324. https:\/\/doi.org\/10.1016\/S1470-2045(21)00005-X","journal-title":"Lancet Oncol"},{"issue":"31","key":"6745_CR6","doi-asserted-by":"publisher","first-page":"3733","DOI":"10.1200\/JCO.2016.67.3467","volume":"34","author":"P Armand","year":"2016","unstructured":"Armand P, Shipp MA, Ribrag V, Michot JM, Zinzani PL, Kuruvilla J et al (2016) Programmed death-1 blockade with pembrolizumab in patients with classical Hodgkin lymphoma after brentuximab vedotin failure. J Clin Oncol 34(31):3733\u20139. https:\/\/doi.org\/10.1200\/JCO.2016.67.3467","journal-title":"J Clin Oncol"},{"issue":"4","key":"6745_CR7","doi-asserted-by":"publisher","first-page":"303","DOI":"10.1007\/s005340170002","volume":"8","author":"Y Nakanuma","year":"2001","unstructured":"Nakanuma Y, Tsuneyama K, Harada K (2001) Pathology and pathogenesis of intrahepatic bile duct loss. J Hepatobiliary Pancreat Surg 8(4):303\u2013315. https:\/\/doi.org\/10.1007\/s005340170002","journal-title":"J Hepatobiliary Pancreat Surg"},{"issue":"4 Suppl 2","key":"6745_CR8","doi-asserted-by":"publisher","first-page":"S90","DOI":"10.1097\/01.mcg.0000155549.29643.ad","volume":"39","author":"M Strazzabosco","year":"2005","unstructured":"Strazzabosco M, Fabris L, Spirli C (2005) Pathophysiology of cholangiopathies. J Clin Gastroenterol 39(4 Suppl 2):S90\u2013S102. https:\/\/doi.org\/10.1097\/01.mcg.0000155549.29643.ad","journal-title":"J Clin Gastroenterol"},{"issue":"7","key":"6745_CR9","doi-asserted-by":"publisher","first-page":"141","DOI":"10.1080\/21548331.1990.11703976","volume":"25","author":"JM Vierling","year":"1990","unstructured":"Vierling JM, Howell CD (1990) Disappearing bile ducts: immunologic mechanisms. Hosp Pract (Off Ed) 25(7):141\u2013150. https:\/\/doi.org\/10.1080\/21548331.1990.11703976","journal-title":"Hosp Pract (Off Ed)"},{"issue":"3","key":"6745_CR10","doi-asserted-by":"publisher","first-page":"261","DOI":"10.1055\/s-2007-1007354","volume":"13","author":"GM Woolf","year":"1993","unstructured":"Woolf GM, Vierling JM (1993) Disappearing intrahepatic bile ducts: the syndromes and their mechanisms. Semin Liver Dis 13(3):261\u2013275. https:\/\/doi.org\/10.1055\/s-2007-1007354","journal-title":"Semin Liver Dis"},{"issue":"6","key":"6745_CR11","doi-asserted-by":"publisher","first-page":"571","DOI":"10.1038\/sj.bmt.1701390","volume":"22","author":"M Picardi","year":"1998","unstructured":"Picardi M, De Rosa G, Muretto P, Pezzullo L, Raiola A, Rotoli B (1998) Ultrasound-guided fine needle cutting biopsy for the characterization of diffuse liver diseases in bone marrow transplant patients. Bone Marrow Transplant 22(6):571\u20133. https:\/\/doi.org\/10.1038\/sj.bmt.1701390","journal-title":"Bone Marrow Transplant"},{"issue":"6","key":"6745_CR12","first-page":"652","volume":"87","author":"M Picardi","year":"2002","unstructured":"Picardi M, Muretto P, De Rosa G, Selleri C, De Renzo A, Persico M et al (2002) Color ultrasound-guided fine needle cutting biopsy for the characterization of diffuse liver damage in critical bone marrow transplanted patients. Haematologica 87(6):652\u2013657","journal-title":"Haematologica"},{"issue":"1","key":"6745_CR13","doi-asserted-by":"publisher","first-page":"262","DOI":"10.1148\/radiol.14132154","volume":"272","author":"M Picardi","year":"2014","unstructured":"Picardi M, Pugliese N, Cirillo M, Zeppa P, Cozzolino I, Ciancia G et al (2014) Advanced-stage Hodgkin lymphoma: US\/chest radiography for detection of relapse in patients in first complete remission\u2013a randomized trial of routine surveillance imaging procedures. Radiology 272(1):262\u201374. https:\/\/doi.org\/10.1148\/radiol.14132154","journal-title":"Radiology"},{"issue":"1","key":"6745_CR14","doi-asserted-by":"publisher","first-page":"5","DOI":"10.1002\/hep.510260101","volume":"26","author":"OM Crosbie","year":"1997","unstructured":"Crosbie OM, Crown JP, Nolan NP, Murray R, Hegarty JE (1997) Resolution of paraneoplastic bile duct paucity following successful treatment of Hodgkin\u2019s disease. Hepatology 26(1):5\u20138. https:\/\/doi.org\/10.1002\/hep.510260101","journal-title":"Hepatology"},{"issue":"4","key":"6745_CR15","doi-asserted-by":"publisher","first-page":"209","DOI":"10.1177\/2040620712443076","volume":"3","author":"C Vaklavas","year":"2012","unstructured":"Vaklavas C, Forero-Torres A (2012) Safety and efficacy of Brentuximab Vedotin in patients with Hodgkin lymphoma or systemic anaplastic large cell lymphoma. Ther Adv Hematol 3(4):209\u2013225. https:\/\/doi.org\/10.1177\/2040620712443076","journal-title":"Ther Adv Hematol"},{"issue":"2","key":"6745_CR16","doi-asserted-by":"publisher","first-page":"e11","DOI":"10.1016\/j.clml.2015.11.015","volume":"16","author":"A Gupta","year":"2016","unstructured":"Gupta A, Petrasek J, Sen S, Gopal P, Naina HV (2016) Single-agent Brentuximab as bridging therapy for Hodgkin lymphoma patients with hepatic impairment. Clin Lymphoma Myeloma Leuk 16(2):e11\u2013e14. https:\/\/doi.org\/10.1016\/j.clml.2015.11.015","journal-title":"Clin Lymphoma Myeloma Leuk"},{"key":"6745_CR17","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2018-227676","author":"M Fong","year":"2019","unstructured":"Fong M, Boyle S, Gutta N (2019) Brentuximab vedotin in combination with sequential procarbazine, cyclophosphamide and prednisolone for the management of Hodgkin\u2019s lymphoma-associated vanishing bile duct syndrome (VBDS) with severe obstructive liver failure. BMJ Case Rep. https:\/\/doi.org\/10.1136\/bcr-2018-227676","journal-title":"BMJ Case Rep"},{"issue":"4","key":"6745_CR18","doi-asserted-by":"publisher","first-page":"1951","DOI":"10.21873\/invivo.12462","volume":"35","author":"I Papakonstantinou","year":"2021","unstructured":"Papakonstantinou I, Kosmidou M, Papathanasiou K, Koumpis E, Kapsali E, Milionis H et al (2021) Paraneoplastic intrahepatic cholestasis in supradiaphragmatic classical Hodgkin lymphoma successfully treated with brentuximab vedotin: a case report and review of the literature. In Vivo 35(4):1951\u20137. https:\/\/doi.org\/10.21873\/invivo.12462","journal-title":"In Vivo"},{"issue":"3","key":"6745_CR19","doi-asserted-by":"publisher","first-page":"154","DOI":"10.3960\/jslrt.21035","volume":"62","author":"K Ishitsuka","year":"2022","unstructured":"Ishitsuka K, Yokoyama Y, Baba N, Matsuoka R, Sakamoto N, Sakamoto T et al (2022) Administration of brentuximab vedotin to a Hodgkin lymphoma patient with liver dysfunction due to vanishing bile duct syndrome resulting in a partial response without any severe adverse events. J Clin Exp Hematop 62(3):154\u20137. https:\/\/doi.org\/10.3960\/jslrt.21035","journal-title":"J Clin Exp Hematop"},{"key":"6745_CR20","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2023-257211","author":"JD Morgan","year":"2023","unstructured":"Morgan JD, Weishar RC, Kwok RM, Lieuw KM (2023) Hodgkin lymphoma associated vanishing bile duct syndrome treated successfully with a brentuximab based regimen. BMJ Case Rep. https:\/\/doi.org\/10.1136\/bcr-2023-257211","journal-title":"BMJ Case Rep"},{"key":"6745_CR21","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2025.1561110","volume":"16","author":"S Ma","year":"2025","unstructured":"Ma S, Cai D, Miao Y, Deng B, Yan X, Gao R (2025) Case report: vanishing bile duct syndrome in Hodgkin\u2019s lymphoma: a case highlighting jaundice and lymphadenopathy as early clues. Front Immunol 16:1561110. https:\/\/doi.org\/10.3389\/fimmu.2025.1561110","journal-title":"Front Immunol"},{"issue":"2","key":"6745_CR22","doi-asserted-by":"publisher","first-page":"659","DOI":"10.1159\/000507695","volume":"13","author":"T Thorsteinsdottir","year":"2020","unstructured":"Thorsteinsdottir T, Loitegard T, Reims HM, Porojnicu AC (2020) Fatal cholestatic liver injury during treatment with PD1 immune checkpoint inhibitor for malignant melanoma: a case report. Case Rep Oncol 13(2):659\u2013663. https:\/\/doi.org\/10.1159\/000507695","journal-title":"Case Rep Oncol"},{"issue":"3","key":"6745_CR23","doi-asserted-by":"publisher","first-page":"906","DOI":"10.1007\/s42399-021-00803-9","volume":"3","author":"C Masetti","year":"2021","unstructured":"Masetti C, Pugliese N, Rimassa L, Finocchiaro G, Di Tommaso L, Lancellotti C et al (2021) Pembrolizumab-induced vanishing bile duct syndrome: a case report. SN Comprehensive Clinical Medicine 3(3):906\u20138. https:\/\/doi.org\/10.1007\/s42399-021-00803-9","journal-title":"SN Comprehensive Clinical Medicine"},{"issue":"4","key":"6745_CR24","doi-asserted-by":"publisher","first-page":"175","DOI":"10.2217\/imt-2021-0078","volume":"14","author":"M Gemelli","year":"2022","unstructured":"Gemelli M, Carbone M, Abbate MI, Mancin M, Zucchini N, Colonese F et al (2022) Vanishing bile duct syndrome following pembrolizumab infusion: case report and review of the literature. Immunotherapy 14(4):175\u201381. https:\/\/doi.org\/10.2217\/imt-2021-0078","journal-title":"Immunotherapy"},{"key":"6745_CR25","doi-asserted-by":"publisher","first-page":"199","DOI":"10.1016\/S0016-5085(63)80013-X","volume":"44","author":"K Juniper","year":"1963","unstructured":"Juniper K (1963) Jr. Prolonged severe obstructive jaundice in hodgkin\u2019s disease. Gastroenterology 44:199\u2013204","journal-title":"Gastroenterology"},{"issue":"2","key":"6745_CR26","first-page":"186","volume":"138","author":"CG Groth","year":"1972","unstructured":"Groth CG, Hellstr\u00f6m K, Hofvendahl S, Nordenstam H, Wengle B (1972) Diagnosis of malignant lymphoma at laparotomy disclosing intrahepatic cholestasis. Acta Chir Scand 138(2):186\u2013189","journal-title":"Acta Chir Scand"},{"issue":"4","key":"6745_CR27","doi-asserted-by":"publisher","first-page":"680","DOI":"10.1016\/S0016-5085(19)32794-5","volume":"67","author":"DR Perera","year":"1974","unstructured":"Perera DR, Greene ML, Fenster LF (1974) Cholestasis associated with extrabiliary Hodgkin\u2019s disease. Report of three cases and review of four others. Gastroenterology 67(4):680\u2013685","journal-title":"Gastroenterology"},{"issue":"1","key":"6745_CR28","doi-asserted-by":"publisher","first-page":"145","DOI":"10.1016\/S0016-5085(79)80026-8","volume":"77","author":"EP Piken","year":"1979","unstructured":"Piken EP, Abraham GE, Hepner GW (1979) Investigation of a patient with Hodgkin\u2019s disease and cholestasis. Gastroenterology 77(1):145\u2013147","journal-title":"Gastroenterology"},{"issue":"189","key":"6745_CR29","first-page":"137","volume":"48","author":"PN Trewby","year":"1979","unstructured":"Trewby PN, Portmann B, Brinkley DM, Williams R (1979) Liver disease as presenting manifestation of Hodgkin\u2019s disease. Q J Med 48(189):137\u2013150","journal-title":"Q J Med"},{"issue":"3 Pt 1","key":"6745_CR30","doi-asserted-by":"publisher","first-page":"304","DOI":"10.1097\/00004836-198606000-00022","volume":"8","author":"DA Lieberman","year":"1986","unstructured":"Lieberman DA (1986) Intrahepatic cholestasis due to Hodgkin\u2019s disease. An elusive diagnosis. J Clin Gastroenterol 8(3 Pt 1):304\u20137. https:\/\/doi.org\/10.1097\/00004836-198606000-00022","journal-title":"J Clin Gastroenterol"},{"issue":"3","key":"6745_CR31","doi-asserted-by":"publisher","first-page":"269","DOI":"10.1055\/s-2008-1040582","volume":"7","author":"MJ Birrer","year":"1987","unstructured":"Birrer MJ, Young RC (1987) Differential diagnosis of jaundice in lymphoma patients. Semin Liver Dis 7(3):269\u2013277. https:\/\/doi.org\/10.1055\/s-2008-1040582","journal-title":"Semin Liver Dis"},{"issue":"3","key":"6745_CR32","first-page":"99","volume":"44","author":"PL Jansen","year":"1994","unstructured":"Jansen PL, van der Lelie H (1994) Intrahepatic cholestasis and biliary cirrhosis associated with extrahepatic Hodgkin\u2019s disease. Neth J Med 44(3):99\u2013102","journal-title":"Neth J Med"},{"issue":"6","key":"6745_CR33","first-page":"940","volume":"89","author":"AS Warner","year":"1994","unstructured":"Warner AS, Whitcomb FF (1994) Extrahepatic Hodgkin\u2019s disease and cholestasis. Am J Gastroenterol 89(6):940\u2013941","journal-title":"Am J Gastroenterol"},{"issue":"4","key":"6745_CR34","doi-asserted-by":"publisher","first-page":"430","DOI":"10.1097\/00005176-199704000-00013","volume":"24","author":"F Gottrand","year":"1997","unstructured":"Gottrand F, Cullu F, Mazingue F, Nelken B, Lecomte-Houcke M, Farriaux JP (1997) Intrahepatic cholestasis related to vanishing bile duct syndrome in Hodgkin\u2019s disease. J Pediatr Gastroenterol Nutr 24(4):430\u2013433. https:\/\/doi.org\/10.1097\/00005176-199704000-00013","journal-title":"J Pediatr Gastroenterol Nutr"},{"issue":"11","key":"6745_CR35","first-page":"1038","volume":"83","author":"BC de Meiros","year":"1998","unstructured":"de Meiros BC, Lacerda MA, Telles JE, da Silva JA, de Meiros CR (1998) Cholestasis secondary to Hodgkin\u2019s disease: report of 2 cases of vanishing bile duct syndrome. Haematologica 83(11):1038\u201340","journal-title":"Haematologica"},{"issue":"1","key":"6745_CR36","doi-asserted-by":"publisher","first-page":"83","DOI":"10.1159\/000012005","volume":"57","author":"S Yal\u00e7in","year":"1999","unstructured":"Yal\u00e7in S, Kars A, S\u00f6kmens\u00fcer C, Atahan L (1999) Extrahepatic hodgkin\u2019s disease with intrahepatic cholestasis: report of two cases. Oncology 57(1):83\u201385. https:\/\/doi.org\/10.1159\/000012005","journal-title":"Oncology"},{"issue":"9","key":"6745_CR37","doi-asserted-by":"publisher","first-page":"1055","DOI":"10.1097\/00042737-199909000-00019","volume":"11","author":"SP Dourakis","year":"1999","unstructured":"Dourakis SP, Tzemanakis E, Deutsch M, Kafiri G, Hadziyannis SJ (1999) <article-title update=\"added\">Fulminant hepatic failure as a presenting paraneoplastic manifestation of Hodgkin\u02bcs disease: Eur J Gastroenterol Hepatol 11(9):1055\u20138. https:\/\/doi.org\/10.1097\/00042737-199909000-00019","journal-title":"Eur J Gastroenterol Hepatol"},{"issue":"2","key":"6745_CR38","doi-asserted-by":"publisher","first-page":"154","DOI":"10.1097\/00043426-200003000-00014","volume":"22","author":"MA Yusuf","year":"2000","unstructured":"Yusuf MA, Elias E, H\u00fcbscher SG (2000) Jaundice caused by the vanishing bile duct syndrome in a child with hodgkin lymphoma. J Pediatr Hematol Oncol 22(2):154\u2013157. https:\/\/doi.org\/10.1097\/00043426-200003000-00014","journal-title":"J Pediatr Hematol Oncol"},{"issue":"5","key":"6745_CR39","doi-asserted-by":"publisher","first-page":"154","DOI":"10.1590\/s1516-31802000000500008","volume":"118","author":"MS Rossini","year":"2000","unstructured":"Rossini MS, Lorand-Metze I, Oliveira GB, Souza CA (2000) Vanishing bile duct syndrome in hodgkin\u2019s disease: case report. Sao Paulo Med J 118(5):154\u2013157. https:\/\/doi.org\/10.1590\/s1516-31802000000500008","journal-title":"Sao Paulo Med J"},{"issue":"1","key":"6745_CR40","first-page":"52","volume":"20","author":"Y Allory","year":"2000","unstructured":"Allory Y, M\u00e9treau J, Zafrani E (2000) [Paraneoplastic vanishing bile duct syndrome in a case of hodgkin\u2019s disease]. Ann Pathol 20(1):52\u201355","journal-title":"Ann Pathol"},{"issue":"3","key":"6745_CR41","doi-asserted-by":"publisher","first-page":"398","DOI":"10.1002\/mpo.1096","volume":"36","author":"A Ozkan","year":"2001","unstructured":"Ozkan A, Yoruk A, Celkan T, Apak H, Yildiz I, Ozbay G (2001) The vanishing bile duct syndrome in a child with hodgkin disease. Med Pediatr Oncol 36(3):398\u2013399. https:\/\/doi.org\/10.1002\/mpo.1096","journal-title":"Med Pediatr Oncol"},{"issue":"5","key":"6745_CR42","doi-asserted-by":"publisher","first-page":"313","DOI":"10.1016\/s0210-5705(02)79026-2","volume":"25","author":"C Ripoll","year":"2002","unstructured":"Ripoll C, Carretero L, Sabin P, Alvarez E, Marrupe D, Ba\u00f1ares R (2002) [Idiopathic cholestasis associated with progressive ductopenia in two patients with hodgkin\u2019s disease]. Gastroenterol Hepatol 25(5):313\u20135. https:\/\/doi.org\/10.1016\/s0210-5705(02)79026-2","journal-title":"Gastroenterol Hepatol"},{"issue":"1","key":"6745_CR43","doi-asserted-by":"publisher","first-page":"57","DOI":"10.1007\/s00277-001-0406-7","volume":"81","author":"S K\u00f6m\u00fcrc\u00fc","year":"2002","unstructured":"K\u00f6m\u00fcrc\u00fc S, Ozet A, Altundag MK, Arpaci F, Ozt\u00fcrk B, Celasun B et al (2002) Vanishing bile duct syndrome occurring after high-dose chemotherapy and autologous peripheral stem cell transplantation in a patient with Hodgkin\u2019s disease. Ann Hematol 81(1):57\u20138. https:\/\/doi.org\/10.1007\/s00277-001-0406-7","journal-title":"Ann Hematol"},{"issue":"3","key":"6745_CR44","doi-asserted-by":"publisher","first-page":"323","DOI":"10.1097\/00042737-200203000-00019","volume":"14","author":"S Liangpunsakul","year":"2002","unstructured":"Liangpunsakul S, Kwo P, Koukoulis GK (2002) Hodgkin\u2019s disease presenting as cholestatic hepatitis with prominent ductal injury. Eur J Gastroenterol Hepatol 14(3):323\u2013327. https:\/\/doi.org\/10.1097\/00042737-200203000-00019","journal-title":"Eur J Gastroenterol Hepatol"},{"issue":"5","key":"6745_CR45","doi-asserted-by":"publisher","first-page":"517","DOI":"10.1177\/030089160409000516","volume":"90","author":"S Guliter","year":"2004","unstructured":"Guliter S, Erdem O, Isik M, Yamac K, Uluoglu O (2004) Cholestatic liver disease with ductopenia (vanishing bile duct syndrome) in hodgkin\u2019s disease: report of a case. Tumori 90(5):517\u2013520. https:\/\/doi.org\/10.1177\/030089160409000516","journal-title":"Tumori"},{"issue":"5","key":"6745_CR46","doi-asserted-by":"publisher","first-page":"275","DOI":"10.1157\/13074061","volume":"28","author":"A C\u00f3rdoba Iturriagagoitia","year":"2005","unstructured":"C\u00f3rdoba Iturriagagoitia A, I\u00f1arrairaegui Bastarrica M, de P\u00e9rez Equiza E, Zozaya Urmeneta JM, Mart\u00ednez-Pe\u00f1uela JM, Beloqui P\u00e9rez R (2005) [Ductal regeneration in vanishing bile duct syndrome in Hodgkin\u2019s lymphoma]. Gastroenterol Hepatol 28(5):275\u20138. https:\/\/doi.org\/10.1157\/13074061","journal-title":"Gastroenterol Hepatol"},{"issue":"2","key":"6745_CR47","first-page":"164","volume":"11","author":"WS Han","year":"2005","unstructured":"Han WS, Jung ES, Kim YH, Kim CH, Park SC, Lee JY et al (2005) [Spontaneous resolution of vanishing bile duct syndrome in hodgkin\u2019s lymphoma]. Korean J Hepatol 11(2):164\u2013168","journal-title":"Korean J Hepatol"},{"issue":"5","key":"6745_CR48","doi-asserted-by":"publisher","first-page":"509","DOI":"10.1056\/NEJMcps051762","volume":"354","author":"A Schmitt","year":"2006","unstructured":"Schmitt A, Gilden DJ, Saint S, Moseley RH (2006) Clinical problem-solving. Empirically incorrect. N Engl J Med 354(5):509\u2013514. https:\/\/doi.org\/10.1056\/NEJMcps051762","journal-title":"N Engl J Med"},{"issue":"1","key":"6745_CR49","doi-asserted-by":"publisher","first-page":"77","DOI":"10.3816\/CLM.2006.n.044","volume":"7","author":"SK Barta","year":"2006","unstructured":"Barta SK, Yahalom J, Shia J, Hamlin PA (2006) Idiopathic cholestasis as a paraneoplastic phenomenon in hodgkin\u2019s lymphoma. Clin Lymphoma Myeloma 7(1):77\u201382. https:\/\/doi.org\/10.3816\/CLM.2006.n.044","journal-title":"Clin Lymphoma Myeloma"},{"issue":"1","key":"6745_CR50","doi-asserted-by":"publisher","first-page":"49","DOI":"10.1038\/ncpgasthep1001","volume":"5","author":"AT DeBenedet","year":"2008","unstructured":"DeBenedet AT, Berg CL, Enfield KB, Woodford RL, Bennett AK, Northup PG (2008) A case of vanishing bile duct syndrome and IBD secondary to hodgkin\u2019s lymphoma. Nat Clin Pract Gastroenterol Hepatol 5(1):49\u201353. https:\/\/doi.org\/10.1038\/ncpgasthep1001","journal-title":"Nat Clin Pract Gastroenterol Hepatol"},{"issue":"2","key":"6745_CR51","doi-asserted-by":"publisher","first-page":"145","DOI":"10.1097\/MEG.0b013e3282b9e6c2","volume":"20","author":"I Leeuwenburgh","year":"2008","unstructured":"Leeuwenburgh I, Lugtenburg EP, van Buuren HR, Zondervan PE, de Man RA (2008) Severe jaundice, due to vanishing bile duct syndrome, as presenting symptom of Hodgkin\u2019s lymphoma, fully reversible after chemotherapy. Eur J Gastroenterol Hepatol 20(2):145\u20137. https:\/\/doi.org\/10.1097\/MEG.0b013e3282b9e6c2","journal-title":"Eur J Gastroenterol Hepatol"},{"issue":"5","key":"6745_CR52","doi-asserted-by":"publisher","first-page":"962","DOI":"10.1080\/02841860701644078","volume":"47","author":"A Ballonoff","year":"2008","unstructured":"Ballonoff A, Kavanagh B, Nash R, Drabkin H, Trotter J, Costa L et al (2008) Hodgkin lymphoma-related vanishing bile duct syndrome and idiopathic cholestasis: statistical analysis of all published cases and literature review. Acta Oncol 47(5):962\u201370. https:\/\/doi.org\/10.1080\/02841860701644078","journal-title":"Acta Oncol"},{"issue":"12","key":"6745_CR53","doi-asserted-by":"publisher","first-page":"976","DOI":"10.1097\/MPH.0b013e31818b37c4","volume":"30","author":"AK Pass","year":"2008","unstructured":"Pass AK, McLin VA, Rushton JR, Kearney DL, Hastings CA, Margolin JF (2008) Vanishing bile duct syndrome and Hodgkin disease: a case series and review of the literature. J Pediatr Hematol Oncol 30(12):976\u2013980. https:\/\/doi.org\/10.1097\/MPH.0b013e31818b37c4","journal-title":"J Pediatr Hematol Oncol"},{"issue":"2","key":"6745_CR54","first-page":"277","volume":"72","author":"H Umit","year":"2009","unstructured":"Umit H, Unsal G, Tezel A, Soylu AR, Pamuk GE, Turgut B et al (2009) Vanishing bile duct syndrome in a patient with hodgkin\u2019s lymphoma and asymptomatic hepatitis B virus infection. Acta Gastroenterol Belg 72(2):277\u2013278","journal-title":"Acta Gastroenterol Belg"},{"issue":"6","key":"6745_CR55","doi-asserted-by":"publisher","first-page":"286","DOI":"10.4103\/1319-3767.121037","volume":"19","author":"A Aleem","year":"2013","unstructured":"Aleem A, Al-Katari M, Alsaleh K, AlSwat K, Al-Sheikh A (2013) Vanishing bile duct syndrome in a Hodgkin\u2019s lymphoma patient with fatal outcome despite lymphoma remission. Saudi J Gastroenterol 19(6):286\u2013289. https:\/\/doi.org\/10.4103\/1319-3767.121037","journal-title":"Saudi J Gastroenterol"},{"issue":"4","key":"6745_CR56","doi-asserted-by":"publisher","first-page":"e164","DOI":"10.1177\/030089161309900426","volume":"99","author":"K Nader","year":"2013","unstructured":"Nader K, Mok S, Kalra A, Harb A, Schwarting R, Ferber A (2013) Vanishing bile duct syndrome as a manifestation of Hodgkin\u2019s lymphoma: a case report and review of the literature. Tumori 99(4):e164\u2013e168. https:\/\/doi.org\/10.1177\/030089161309900426","journal-title":"Tumori"},{"key":"6745_CR57","doi-asserted-by":"publisher","first-page":"529","DOI":"10.1186\/1756-0500-7-529","volume":"7","author":"D Rota Scalabrini","year":"2014","unstructured":"Rota Scalabrini D, Caravelli D, Carnevale Schianca F, D\u2019Ambrosio L, Tolomeo F, Boccone P et al (2014) Complete remission of paraneoplastic vanishing bile duct syndrome after the successful treatment of hodgkin\u2019s lymphoma: a case report and review of the literature. BMC Res Notes 7:529. https:\/\/doi.org\/10.1186\/1756-0500-7-529","journal-title":"BMC Res Notes"},{"issue":"2","key":"6745_CR58","doi-asserted-by":"publisher","first-page":"366","DOI":"10.3748\/wjg.v23.i2.366","volume":"23","author":"M Bakhit","year":"2017","unstructured":"Bakhit M, McCarty TR, Park S, Njei B, Cho M, Karagozian R et al (2017) Vanishing bile duct syndrome in Hodgkin\u2019s lymphoma: a case report and literature review. World J Gastroenterol 23(2):366\u201372. https:\/\/doi.org\/10.3748\/wjg.v23.i2.366","journal-title":"World J Gastroenterol"}],"container-title":["Annals of Hematology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00277-026-06745-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s00277-026-06745-3","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s00277-026-06745-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,16]],"date-time":"2026-06-16T18:54:47Z","timestamp":1781636087000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s00277-026-06745-3"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,2,26]]},"references-count":58,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2026,4]]}},"alternative-id":["6745"],"URL":"https:\/\/doi.org\/10.1007\/s00277-026-06745-3","relation":{},"ISSN":["1432-0584"],"issn-type":[{"value":"1432-0584","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,2,26]]},"assertion":[{"value":"4 November 2025","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"22 December 2025","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"26 February 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"This case report was reviewed and approved by the Research Ethics Committee B of National Taiwan University Hospital (approval No. 202504144\u00a0W; date of approval: May 14, 2025). The study was conducted in accordance with the Declaration of Helsinki.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent for publication of clinical details and images was obtained from the patient.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"144"}}