A Case of Vanishing Bile Duct Syndrome after Drug-Induced Liver Injury Caused by Pelubiprofen

Bak, Haein; Kim, Hayeon; Lee, Sieun; Lee, Yoonseok; Bang, Soo-Min; Lee, Young‐Sun

doi:10.3349/ymj.2020.61.12.1060

Cited by 4 publications

(3 citation statements)

References 19 publications

(21 reference statements)

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“…To the best of our knowledge, this is the fifteenth report of VBDS associated with SJS or TEN ( Table 1 ). The prognoses varied greatly, with two patients succumbing to death during hospital treatment ( 15 ), seven patients requiring liver transplantation ( 8 , 11 , 13 , 16 , 18 , 20 , 21 ), and the remaining six patients showing clinical resolution ( 9 , 10 , 12 , 14 , 17 , 19 ). Further studies are necessary to identify the possible risk factors, prognostic indicators, and the standard of care for VBDS with SJS or TEN.…”

Section: Discussionmentioning

confidence: 99%

Case report: Development of vanishing bile duct syndrome in Stevens-Johnson syndrome complicated by hemophagocytic lymphohistiocytosis

2022

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BackgroundVanishing bile duct syndrome is a rare drug-induced disease characterized by cholestasis and ensuing ductopenia. Dermatological manifestations of drug hypersensitivity such as Stevens-Johnson syndrome and toxic epidermal necrolysis may also present in such cases. Hemophagocytic lymphohistiocytosis is a hyperimmune response caused by unchecked stimulation of macrophages, natural killer cells, and cytotoxic T lymphocytes.Case presentationWe report a severe case who presented with concurrent Stevens-Johnson syndrome and vanishing bile duct syndrome complicated by hemophagocytic lymphohistiocytosis after the ingestion of non-steroidal anti-inflammatory drugs. Despite the fact that improvements in vanishing bile duct syndrome can be assumed when combining the clinical lab data clues, as well as repeated liver biopsies showing recovering ductopenia, the patient developed hypovolemic shock combined with septic shock episodes and died on day 236.ConclusionTo our knowledge, this is the fifteenth report of vanishing bile duct syndrome associated with Stevens-Johnson disease or toxic epidermal necrolysis. Mortality rate remains high without treatment guidelines established due to the rarity and heterogenicity of the population. Further studies are needed to identify possible risk factors, prognostic indicators, and the standard of care for vanishing bile duct syndrome associated with Stevens-Johnson disease or toxic epidermal necrolysis.

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Section: Discussionmentioning

confidence: 99%

Case report: Development of vanishing bile duct syndrome in Stevens-Johnson syndrome complicated by hemophagocytic lymphohistiocytosis

2022

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“…Pelubiprofen is a COX-2 selective nonsteroidal anti-inflammatory drugs that is primarily used in South Korea for the treatment of osteoarthritis and is touted for its lower incidence of gastrointestinal side effects [80]. Bak et al reported a 29-year-old female patient who was hospitalized 2 days after ingesting pelibuprofen and found to have markedly elevated liver enzymes, including ALT 6479 IU/L and AST 4254 IU/L on day 2 of admission [81]. Interestingly, this patient had a Tbili of 3.12 mg/dL on the day of admission, which continued to uptrend to a peak of 11.81 mg/dL on day 23.…”

Section: Quantitative Systems Pharmacology and Dilimentioning

confidence: 99%

“…Immunostaining for CK19 confirmed bile duct loss and bile ductular proliferation. Therefore, a diagnosis of vanishing bile duct syndrome was made with a RUCAM score of 6 indicating a low "probable" causation by pelubiprofen, although the height of the aminotransferases is atypical of other cases of vanishing bile duct syndrome [81].…”

Section: Quantitative Systems Pharmacology and Dilimentioning

confidence: 99%

Drug-Induced Liver Injury: Highlights and Controversies in the Recent Literature

et al. 2021

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Drug-induced liver injury (DILI) remains an important, yet challenging diagnosis for physicians. Each year, additional drugs are implicated in DILI and this year was no different, with more than 1400 articles published on the subject. This review examines some of the most significant highlights and controversies in DILI-related research over the past year and their implications for clinical practice. Several new drugs were approved by the US Food and Drug Administration including a number of drugs implicated in causing DILI, particularly among the chemotherapeutic classes. The COVID-19 pandemic was also a major focus of attention in 2020 and we discuss some of the notable aspects of COVID-19-related liver injury and its implications for diagnosing DILI. Updates in diagnostic and causality assessments related to DILI such as the Roussel Uclaf Causality Assessment Method are included, mindful that there is still no single biomarker or diagnostic tool to unequivocally diagnose DILI. Glutamate dehydrogenase received renewed attention as being more specific than alanine aminotransferase. There were a few new reports of previously unrecognized hepatotoxins, including immune modulators and novel gene therapy drugs that we highlight. Updates and new developments of previously described hepatotoxins, such as immune checkpoint inhibitors and anti-tuberculosis drugs are reviewed. Finally, novel technologies such as organoid culture systems to better predict DILI preclinically may be coming of age and determinants of hepatocyte loss, such as calculating P ALT are poised to improve our current means of estimating DILI severity and the risk of acute liver failure.

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Pelubiprofen

2021

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A Case of Vanishing Bile Duct Syndrome after Drug-Induced Liver Injury Caused by Pelubiprofen

Cited by 4 publications

References 19 publications

Case report: Development of vanishing bile duct syndrome in Stevens-Johnson syndrome complicated by hemophagocytic lymphohistiocytosis

Case report: Development of vanishing bile duct syndrome in Stevens-Johnson syndrome complicated by hemophagocytic lymphohistiocytosis

Drug-Induced Liver Injury: Highlights and Controversies in the Recent Literature

Pelubiprofen

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