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Temozolomide (TMZ)-levetiracetam (LEV) combination therapy in glioblastoma management is gradually becoming a mainstay treatment given its superior effect compared with TMZ monotherapy. While there have been previous cases of hepatotoxicity, there are no prior reports of vanishing bile duct syndrome (VBDS) associated with TMZ-LEV combination use. This case report details a male in his 50s who had recently completed TMZ and LEV for right frontal lobe glioblastoma.

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