Kaposi sarcoma is one of the acquired immunodeficiency syndrome (AIDS) defining diseases. AIDS-associated Kaposi sarcoma affects primarily the skin and the lungs. Although gastrointestinal involvement is relatively common, biliary tract involvement has rarely been reported. It has been associated mostly with extension from liver disease. We describe an uncommon presentation of disseminated Kaposi sarcoma causing extrahepatic cholestasis due to extrahepatic biliary tract involvement that resolved after sphincterotomy with biliary stenting. We present a case of a 35-year-old African American male diagnosed with human immunodeficiency virus (HIV) infection in 2005. He presented with AIDS after discontinuation of antiretroviral therapy for one year, subsequently being diagnosed with systemic Kaposi sarcoma. He presented with signs and symptoms of obstructive biliary disease, including jaundice, abdominal pain, fatigue, and fever. We encountered a rare presentation of malignant single extrahepatic biliary stenosis secondary to biliary Kaposi sarcoma. The biochemical pattern markedly improved after endoscopic retrograde cholangiopancreatography with sphincterotomy and stenting. However, and despite the resumption of combined antiretroviral therapy, deep immunosuppression caused worsening clinical condition and death five months after initial presentation. Certainly, among the multiple etiologies of biliary obstruction in AIDS, Kaposi sarcoma is one to consider.
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