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

Wong, Kiong-Ming; Chang, Chao-Sung; Wu, Chun‐Chieh; Yin, Hsin‐Ling

doi:10.1016/j.kjms.2013.05.002

Cited by 21 publications

(29 citation statements)

References 18 publications

(27 reference statements)

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“…[38]1YesFullNoCureUmit H. [39]1UnknownUnknownUnknownUnknownGill R.M. [40]1YesReducedNoHepatic failureForamiti S. [41]1YesFullYesHepatic failureGagnon MF [42]1YesFullNoCureWong KM [43]1YesFullNoCureAleem A1 [44]1YesFullNoHepatic failureNader K [45]1YesFullNoHepatic failureOur patient1YesFullYesCure*either chemotherapy or radiotherapy.…”

Section: Resultsmentioning

confidence: 99%

Complete remission of paraneoplastic vanishing bile duct syndrome after the successful treatment of Hodgkin’s lymphoma: a case report and review of the literature

et al. 2014

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BackgroundVanishing bile duct syndrome has been associated with different pathologic conditions (adverse drug reactions, autoimmune diseases, graft versus host disease, and cancer). Though its causes are unknown, an immune-related pathogenesis is the most likely one. Vanishing bile duct syndrome can evolve to hepatic failure and, eventually, to death. The treatment is uncertain, but it needs the resolution of the underlying pathologic condition.Case presentationWe describe the association of Hodgkin’s lymphoma with a syndrome characterized by cholestasis, aminotransferase elevation and an histological picture of bile duct loss. All other causes of hepatic function impairment were excluded (in particular, drugs, viral and autoimmune related diseases) eventually leading to the diagnosis of vanishing bile duct syndrome. Despite the fact that the dysfunction is not caused by hepatic Hodgkin’s lymphoma involvement, liver impairment can limit the optimal therapy of Hodgkin’s lymphoma. A treatment consisting of ursodeoxycholic acid, prednisone, and full dose chemotherapy restored hepatic function and achieved complete and long-lasting remission of Hodgkin’s lymphoma.ConclusionWe reviewed all case reports showing that vanishing bile duct syndrome is a dismal paraneoplastic syndrome being fatal in a high proportion of patients if not adequately treated. Indeed, this syndrome requires both an early recognition and an appropriate aggressive treatment consisting of full dose upfront chemotherapy which is the only way to achieve a resolution of the vanishing bile duct syndrome. Delayed or reduced intensity treatments unfavorably correlate with survival.

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Section: Resultsmentioning

confidence: 99%

Complete remission of paraneoplastic vanishing bile duct syndrome after the successful treatment of Hodgkin’s lymphoma: a case report and review of the literature

et al. 2014

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“…Not only lymphoma responded to 8 cycles of ABVD regimen followed by autologous hematopoietic stem cell transplantation, but also VBDS disappeared. The repeated liver biopsy at the moment of lymphoma complete remission found interlobular bile ducts regeneration, an argument for the reversibility of bile duct injury in VBDS [58]. Such isolated cases do not allow us to draw any definite conclusion on the general reversibility of VBDS.…”

Section: Hodgkin's Lymphoma and Vanishing Bile Duct Syndromementioning

confidence: 92%

Liver Involvement in Hodgkin's Lymphoma: Types of Injuries and Therapeutic Implications

Mihăilă

2017

IJPPE

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Abstract. The hepatocytolysis raises questions on following therapeutic conduct when it occurs during chemotherapy for Hodgkin's lymphoma, expression of its liver toxicity. But the onset of primary liver Hodgkin's lymphoma, including the form manifested by acute liver failure, poses even greater problems, as in the case of occurrence of vanishing bile duct syndrome -expression of a paraneoplastic syndrome, hemophagocytic lymphohistiocytosis, peliosis hepatis or association of lymphoma with infection with hepatitis viruses or human immunodeficiency virus or different autoimmune diseases. This review summarizes the clinical experience acquired on the relationship between Hodgkin's lymphoma and liver, from the point of view of clinical manifestations, used treatments and clinical evolution. Suggestions on the course of treatment in patients with Hodgkin's lymphoma and liver damage have been formulated starting from the metabolism and elimination of chemotherapy drugs and taking into account the clinical experience of published clinical trials and cases. This review is a synthesis of knowledge obtained in this field, during the time, of therapeutic possibilities and limits, and formulates potential future milestones for research.

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“…30 A second theory includes direct bile duct damage by paraneoplastic toxic cytokines released by the lymphoma cells. 31 Without treatment, the natural progression is hepatic failure. In cases of VBDS that are not related to HL, the bile ducts disappear irreversibly; however, as a paraneoplastic phenomenon, full resolution of symptomatology, laboratory, and histologic findings has been described with appropriate HL therapy in some case reports.…”

Section: Idiopathic Cholestasismentioning

confidence: 99%

“…In cases of VBDS that are not related to HL, the bile ducts disappear irreversibly; however, as a paraneoplastic phenomenon, full resolution of symptomatology, laboratory, and histologic findings has been described with appropriate HL therapy in some case reports. 5,30,31 Two reviews of HL and VBDS in both adults and children reported severe complications, notably liver failure and sepsis, and high mortality rates. The addition of steroid therapy to standard chemotherapy and radiotherapy improved hepatic function and was more likely to lead to complete remission.…”

Section: Idiopathic Cholestasismentioning

confidence: 99%

Paraneoplastic Syndromes in Children with Hodgkin Lymphoma

Betcherman¹,

Punnett²

2017

Oncology &Amp; Hematology Review (US)

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Paraneoplastic syndromes (PNS) refer to a phenomenon whereby certain malignancies manifest as symptoms not directly related to the tumor itself. PNS has been described in association with Hodgkin lymphoma (HL) in adults and children and may affect a number of organ systems. The pathophysiology is variable and in many cases is not well understood. Specific paraneoplastic antibodies have been isolated in some syndromes, though are not required for the diagnosis. The two best described for HL are the anti-Tr and anti-mGluR5 antibodies identified in some cases of limbic encephalitis and cerebellar degeneration respectively. A high index of suspicion for underlying malignancy is necessary when recognizing these clinical syndromes to avoid a diagnostic delay. Successful treatment of the HL often reverses the manifestations of PNS although organ function at diagnosis may limit therapeutic options. Some patients suffer devastating complications of their PNS.

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

Cited by 21 publications

References 18 publications

Complete remission of paraneoplastic vanishing bile duct syndrome after the successful treatment of Hodgkin’s lymphoma: a case report and review of the literature

Complete remission of paraneoplastic vanishing bile duct syndrome after the successful treatment of Hodgkin’s lymphoma: a case report and review of the literature

Liver Involvement in Hodgkin's Lymphoma: Types of Injuries and Therapeutic Implications

Paraneoplastic Syndromes in Children with Hodgkin Lymphoma

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