Resolution of paraneoplastic bile duct paucity following successful treatment of Hodgkin's disease

Crosbie, Órla; Crown, John; Nolan, Niamh; Murray, R; Hegarty, John E.

doi:10.1002/hep.510260101

Cited by 43 publications

(13 citation statements)

References 8 publications

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“…Only Crosbie et al described a single patient with successfully treated lymphoma who survived with serum bilirubin levels decreasing from 622 to 11 mmol/l. In a liver biopsy sample taken after therapy, ductular proliferation was detected [16]. In this journal Liangpunsakul et al described a patient with a cholestatic liver disease caused by Hodgkin's lymphoma.…”

Section: Discussionmentioning

confidence: 95%

Severe jaundice, due to vanishing bile duct syndrome, as presenting symptom of Hodgkinʼs lymphoma, fully reversible after chemotherapy

Leeuwenburgh¹,

Lugtenburg

Buuren³

et al. 2008

European Journal of Gastroenterology & Hepatology

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Liver involvement in Hodgkin's lymphoma is common and is caused by hepatic infiltration, biliary obstruction by lymphoma, hepatitis, sepsis or complications of chemotherapeutic treatment. Jaundice caused by the vanishing bile duct syndrome related to Hodgkin's lymphoma is very rare. The mechanism is poorly understood but a paraneoplastic effect seems most likely as liver biopsy samples show cholestasis in the absence of lymphoma cells. Despite adequate treatment almost all reported patients died of liver failure or disease progression. Disease progression is explained partly by the difficulties encountered in the administration of potential hepatotoxic chemotherapy in severely cholestatic patients. We describe a 17-year-old man with vanishing bile duct syndrome and Hodgkin's lymphoma who was treated successfully with chemotherapy. The markedly elevated serum bilirubin levels completely normalized. Our case demonstrates that although dosing of chemotherapy in this situation can be very difficult, a good clinical outcome is possible, which makes the attempt at curative treatment worthwhile.

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Section: Discussionmentioning

confidence: 95%

Severe jaundice, due to vanishing bile duct syndrome, as presenting symptom of Hodgkinʼs lymphoma, fully reversible after chemotherapy

Leeuwenburgh¹,

Lugtenburg

Buuren³

et al. 2008

European Journal of Gastroenterology & Hepatology

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“…Indeed, in some of the previous reports of HL-related VBDS or IC other potential hepatotoxic etiologies were present including ketoconazole [1], medroxyprogesterone [17], pregnancy [10], myeloablative chemotherapy [21], and erythromycin [15]; leading one to question the exact etiology of liver injury in these cases. However, most of the reported cases of hepatic dysfunction occurred in the absence of any other identifiable cause, implicating HL as the inciting factor.…”

Section: Identification Of Hl-related Vbds and Icmentioning

confidence: 99%

“…As shown in our analysis of all published cases, 32% (12/37) of patients with HL-related VBDS/IC were alive with stable and near-normal LFT's at last follow-up, indicating this disease is reversible in at least a subgroup of patients. Indeed, two patients actually had repeat post-therapy biopsies demonstrating regeneration of the intrahepatic bile ducts after complete remission of HL [15,17].…”

Section: Clinical Coursementioning

confidence: 99%

Hodgkin lymphoma-related vanishing bile duct syndrome and idiopathic cholestasis: Statistical analysis of all published cases and literature review

et al. 2008

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“…[17]1YesFullLateHepatic failureWarner A.S. [18]1YesFullNoCureGottrand F. [19]1NoNoNoHepatic failureCrosbie O.M. [20]1YesFullYesCureDe Medeiros B.C. [21]2YesFullNoHepatic failureFullYalcin S. [22]2NoNoNoHepatic failureYesFullNoCureDourakis S.P.…”

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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Resolution of paraneoplastic bile duct paucity following successful treatment of Hodgkin's disease

Cited by 43 publications

References 8 publications

Severe jaundice, due to vanishing bile duct syndrome, as presenting symptom of Hodgkinʼs lymphoma, fully reversible after chemotherapy

Severe jaundice, due to vanishing bile duct syndrome, as presenting symptom of Hodgkinʼs lymphoma, fully reversible after chemotherapy

Hodgkin lymphoma-related vanishing bile duct syndrome and idiopathic cholestasis: Statistical analysis of all published cases and literature review

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

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