Minoxidil, an antihypertensive agent used in severe refractory hypertension is metabolized by the liver and excreted by the kidneys. It is not known to be hepatotoxic but dose adjustments are required in renal failure. We report a case of a 73 year old female with elevated bilirubin and unresolving pruritus of 2 months duration. She complained of rash, pruritus, yellow discoloration of eyes and dark urine. She denied abdominal pain, anorexia, nausea, vomiting, fever or confusion. She also denied use of alcohol, illicit drugs, herbals or over-the-counter medications. Patient was on long term treatment with clonidine, losartan-hydrochlorothiazide, metoprolol, and insulin detemir. She started hemodialysis three months prior for end stage renal disease. Minoxidil was initiated two months prior for uncontrolled hypertension, which coincided with the onset of her symptoms. Vital signs were normal except an elevated blood pressure. Physical examination was unremarkable but for sclera icterus and maculopapular rash on her back and arms. Laboratory showed mild normocytic anemia, normal platelets, and white cell count 13,700/cmm, blood urea nitrogen 28 mg/dl, creatinine 4.7 mg/dl, total bilirubin 23.6 mg/dl, direct bilirubin > 15 mg/dl, aspartate aminotransferase 109 U/L, alanine aminotransferase 101 U/L, albumin 2.9 g/dl, and alkaline phosphatase 759 U/L. She was diagnosed with cholestatic hepatitis. Abdominal computed tomography showed cholelithiasis with normal bile ducts. Hepatitis panel, including AMA, ANA, IgG, ACE levels were normal. Antibiotics and supportive treatment were initiated. Magnetic resonance cholangiopancreatography showed no abnormalities. Bilirubin and transaminases remained elevated despite regular hemodialysis. Liver biopsy was attempted, during which she suffered peritoneal bleeding, hypotension and ultimately cardiac arrest. Subsequent mesenteric angiogram and embolization were unsuccessful. Patient expired the next day after multiple cardiac arrests. Liver biopsy at autopsy revealed vanishing biliary duct syndrome (VBDS). VBDS, also known as ductopenia, is associated with destruction and disappearance of intrahepatic bile ducts leading to cholestasis. Patients can present with fatigue, anorexia, pruritus, abnormal liver chemistries with cholestatasis.Imaging and laboratory values aide in diagnosis, however liver biopsy is confirmatory. This case illustrates an importance to consider VBDS as a differential diagnosis of cholestasis.