Cloxacillin-induced acute vanishing bile duct syndrome: a case study and
literature review
Abstract
Ductopenia is often regarded as a chronic process where ≥50% of portal
tracts lack bile ducts, which is also known as vanishing bile duct
syndrome (VBDS). One etiology is drug-induced liver injury. Cloxacillin,
an anti-staphylococcal penicillin, typically causes “bland”
cholestasis. We present the first case of cloxacillin-induced acute
ductopenia or VBDS and a review of published cloxacillin-induced liver
injuries. A 66-year-old woman with no prior liver disease, but known
penicillin allergy, was treated for post-carotid angioplasty
staphylococcal infection with 6 weeks of cloxacillin. She presented with
a two-week history of weakness and jaundice. Laboratory work-up showed
elevated liver enzymes, hyperbilirubinemia, and eosinophilia. She
required ICU transfer for hypotension and was started empirically on
prednisone. Liver biopsy revealed severe centrilobular cholestasis, mild
necroinflammation, and ductopenia with epithelial injury, but no
ductular reaction. Two-months later, she was discharged on
hydrocortisone and ursodiol with persistently elevated alkaline
phosphatase and bilirubin. She was considered for liver transplantation
but died of liver failure four months later. Four additional articles
were found with histopathologic descriptions of cloxacillin-related
liver injury. These included portal inflammation, cholestasis and mild
necroinflammation. Clinical features were reported in two cases; both
had mild symptoms with cholestatic liver enzymes and hyperbilirubinemia.
Both patients recovered completely within 10-60 days.
Cloxacillin-induced cholestasis can be secondary to acute ductopenia,
which can result in worse clinical outcomes than previously described
“bland” cholestasis. Liver biopsy is recommended to identify cases
with acute VBDS.