Successful liver transplantation for drug-induced vanishing bile duct syndrome

Hashim, Ahmed; Barnabas, Ashley; Miquel, Rosa; Agarwal, Kosh

doi:10.1136/bcr-2019-233052

Cited by 9 publications

(6 citation statements)

References 19 publications

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“…Similarly, anecdotal evidence for plasmapheresis to treat VBDS has been reported with mixed results 14,46,47 . In cases that progress to biliary cirrhosis, supportive management and liver transplantation would be indicated 18 …”

Section: Discussionmentioning

confidence: 99%

“…In certain cases, jaundice and abnormal liver function tests may eventually resolve, and some potential treatments have been reported 13–15 . However, VBDS may also progress to biliary cirrhosis and require liver transplantation 12,16–18 …”

Section: Introductionmentioning

confidence: 99%

“…[13][14][15] However, VBDS may also progress to biliary cirrhosis and require liver transplantation. 12,[16][17][18] Cloxacillin, oxacillin and flucloxacillin belong to a group of penicillinase-resistant isoxazolyl penicillins which are often used for methicillin-sensitive Staphylococcus species, Pneumococcus and betahaemolytic Streptococci. 19 Flucloxacillin is a well-known cause of drug-induced cholestasis, particularly ductopenia.…”

Section: Introductionmentioning

confidence: 99%

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Cloxacillin‐induced acute vanishing bile duct syndrome: A case study and literature review

Faragalla

Lau

Wang

et al. 2022

Brit J Clinical Pharma

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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 aetiology is drug‐induced liver injury. Cloxacillin, an antistaphylococcal 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 postcarotid angioplasty staphylococcal infection with 6 weeks of cloxacillin. She presented with a 2‐week history of weakness and jaundice. Laboratory work‐up showed elevated liver enzymes with a cholestatic pattern, 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 4 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.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Cloxacillin‐induced acute vanishing bile duct syndrome: A case study and literature review

Faragalla

Lau

Wang

et al. 2022

Brit J Clinical Pharma

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“…Plasmapheresis has been used successfully in a case of loxprofen-induced ductopenia in an adolescent female unresponsive to initial treatment measures 14 . In cases that progress to biliary cirrhosis, supportive management and LT would be indicated 18 .…”

Section: Discussionmentioning

confidence: 99%

“…In certain cases, jaundice and abnormal liver function tests may eventually resolve, and successful treatments have been reported [13][14][15] . However, VBDS may also progress to biliary cirrhosis and require liver transplantation (LT) 12,[16][17][18] .…”

Section: Introductionmentioning

confidence: 99%

Cloxacillin-induced acute vanishing bile duct syndrome: a case study and literature review

Faragalla

Lau

Wang

et al. 2022

Preprint

View full text Add to dashboard Cite

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.

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