Acute vanishing bile duct syndrome after ibuprofen therapy in a child

Taghian, Mehdi; Tran, Tú-Anh; Bresson‐Hadni, Solange; Menget, A.; Félix, Sophie; Jacquemin, Emmanuel

doi:10.1016/j.jpeds.2004.05.027

Cited by 54 publications

(47 citation statements)

References 13 publications

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“…Observations of at least partial resolution of the 'vanishing bile duct syndrome' (VBDS) following allograft rejection 1 or transient exposure to drugs such as carbamazepine [2][3][4] suggests that the loss of bile ducts and their constituent intrahepatic biliary epithelial cells (IBECs) might be reversible. Nevertheless, progressive cholestasis resulting from bile duct loss is conventionally thought to predispose to portal fibrosis and, ultimately, biliary cirrhosis.…”

mentioning

confidence: 99%

Epithelial–mesenchymal transition contributes to portal tract fibrogenesis during human chronic liver disease

Rygiel

Robertson

Marshall

et al. 2008

Laboratory Investigation

214

189

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mentioning

confidence: 99%

Epithelial–mesenchymal transition contributes to portal tract fibrogenesis during human chronic liver disease

Rygiel

Robertson

Marshall

et al. 2008

Laboratory Investigation

214

189

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“…4 Okan et al did not use TNF-a blockade but hypothesized that it could be effective because case reports showed rapid resolution of skin and mucosal damage in patients with SJS/TEN after administration of infliximab, 10,11 together with the theory of a common immunologic mechanism. [1][2][3][4][5][6][7][12][13][14] Infliximab has been reported to increase the incidence of infections, but the majority of reports have been upper respiratory tract illnesses, ,2% of which were pneumonia. 15 We chose infliximab for our patient because of its more favorable side effect profile.…”

Section: Discussionmentioning

confidence: 99%

“…Those cases showed mixed results, with 2 patients showing clinical resolution and 1 patient requiring liver transplant (see Table 1). 1,3,4,7 Longer follow-up will be required to evaluate infliximab' s efficacy. Our case also supports choleresis for VBDS because our patient' s bilirubin levels declined once ursodiol was started.…”

Section: Discussionmentioning

confidence: 99%

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Infliximab/Plasmapheresis in Vanishing Bile Duct Syndrome Secondary to Toxic Epidermal Necrolysis

White¹,

Appleman²

2014

Pediatrics

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Vanishing bile duct syndrome (VBDS) is a rare disorder characterized by loss of interlobular bile ducts and progressive worsening cholestasis. The acute presentation of this disease is typically associated with a drug hypersensitivity and Stevens-Johnson syndrome/toxic epidermal necrolysis (TEN). The mainstay of treatment has been ursodeoxycholic acid with mixed results from immunosuppressive regimens. Anti–tumor necrosis factor-α and plasmapheresis have been speculated to be of potential benefit. It is hoped that early identification and intervention in VBDS secondary to Stevens-Johnson syndrome/TEN with continued reporting will lead to better regimens and outcomes. Our case report details the first reported use of infliximab and plasmapheresis, in addition to steroids, in a patient with VBDS secondary to TEN, as well as a literature review that supports a mechanism for why these modalities could be effective treatments. Unfortunately, our patient died, and the use of these therapies had an unclear benefit on his liver and skin disease. We hope that additional work can be published to confirm or refute their utility in the treatment of these diseases.

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“…Ука-зывается на длительный -до 7 мес. и более -период раз-решения данного гепатита [20,21], а также возможность формирования цирроза печени в исходе такового.…”

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