Vanishing bile duct syndrome in human immunodeficiency virus infected adults:A report of two cases |
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Authors: | Ana Paula Oppenheimer Christopher Koh Mary McLaughlin John C Williamson Thomas D Norton Jennifer Laudadio Theo Heller David E Kleiner Kevin P High Caryn G Morse |
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Institution: | Section of Infectious Diseases,Department of Medicine,Wake Forest School of Medicine,Winston-Salem,NC 27109-7205,United States;Liver Diseases Branch,National Institute of Diabetes,Digestive and Kidney Diseases,National Institutes of Health Clinical Center,Bethesda,MD 20892-1403,United States;Laboratory of Immunoregulation,National Institute of Allergy and Infectious Diseases,National Institutes of Health Clinical Center,Bethesda,MD 20892-1403,United States;Division of Infectious Diseases and Immunology,Department of Medicine,New York University School of Medicine,New York,NY 10016,United States;Laboratory of Pathology,Wake Forest School of Medicine,Winston-Salem,NC 27109-7205,United States;Laboratory of Pathology,National Cancer Institute,National Institutes of Health Clinical Center,Bethesda,MD 20892-1403,United States;Department of Critical Care Medicine,National Institutes of Health Clinical Center,Bethesda,MD 20892-1403,United States |
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Abstract: | Vanishing bile duct syndrome (VBDS) is a group of rare disorders characterized by ductopenia, the progressive destruction and disappearance of intrahepatic bile ducts leading to cholestasis. Described in association with medications, autoimmune disorders, cancer, transplantation, and infections, the specific mechanisms of disease are not known. To date, only 4 cases of VBDS have been reported in human immunodeficiency virus (HIV) infected patients. We report 2 additional cases of HIV-associated VBDS and review the features common to the HIV-associated cases. Presentation includes hyperbilirubinemia, normal liver imaging, and negative viral and autoimmune hepatitis studies. In HIV-infected subjects, VBDS occurred at a range of CD4+ T-cell counts, in some cases following initiation or change in antiretroviral therapy. Lymphoma was associated with two cases; nevirapine, antibiotics, and viral co-infection were suggested as etiologies in the other cases. In HIV-positive patients with progressive cholestasis, early identification of VBDS and referral for transplantation may improve outcomes. |
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Keywords: | Human immunodeficiency virus Antiretroviral therapy Vanishing bile duct syndrome Ductopenia Liver biopsy |
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