Hodgkin's lymphoma-related vanishing bile duct syndrome: A case report and literature review

Abstract We report the case of a 38-year-old man who developed vanishing bile duct syndrome in association with Hodgkin's lymphoma. He was noted to have cervical lymphadenopathy and marked elevation of total serum bilirubin at diagnosis. He achieved complete remission with normalization of seru...

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Bibliographic Details
Published in:The Kaohsiung journal of medical sciences 2013-11, Vol.29 (11), p.636-641
Main Authors: Wong, Kiong-Ming, Chang, Chao-Sung, Wu, Chun-Chieh, Yin, Hsin-Ling
Format: Article
Language:English
Subjects:
Adult
Bile Duct Diseases - complications
Bile Duct Diseases - pathology
Bilirubin
Cancer
Chemotherapy
Diagnosis
Ductopenia
Hematopoietic stem cells
Hodgkin Disease - complications
Hodgkin Disease - pathology
Hodgkin's lymphoma
Humans
Internal Medicine
Liver
Lymphomas
Male
Transplantation
Vanishing bile duct syndrome
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Summary:Abstract We report the case of a 38-year-old man who developed vanishing bile duct syndrome in association with Hodgkin's lymphoma. He was noted to have cervical lymphadenopathy and marked elevation of total serum bilirubin at diagnosis. He achieved complete remission with normalization of serum bilirubin after eight courses of Adriamycin, bleomycin, vinblastine, and dacarbazine chemotherapy followed with autologous hematopoietic cell transplantation. Consecutive liver biopsies performed at diagnosis and at the stage of complete remission revealed the disappearance and regeneration of interlobular bile ducts, respectively. Our case provides pathological evidence that Hodgkin's lymphoma-related vanishing bile duct syndrome is a reversible bile duct injury disease. Bilirubin is a reliable serum marker to monitor the treatment response of these cases. The mechanism to develop hyperbilirubinemia with vanishing bile duct in such a case of Hodgkin's lymphoma remains to be studied. A literature review was carried out.
ISSN:1607-551X
2410-8650
DOI:10.1016/j.kjms.2013.05.002