Summary: | Antifungal agents account for approximately 3% of Drug-Induced Liver Injury (DILI) cases. Isavuconazole is a novel triazole, and experience with long-term use of it is lacking. We report a case of late-onset DILI occurring after 11 months of isavuconazole therapy in a 55-year old man of Angolan descent on long-term antifungal therapy for the management of chronic pulmonary aspergillosis complicating previously treated pulmonary tuberculosis. The DILI could be described as idiosyncratic as it was not associated with high isavuconazole serum levels and his liver function tests returned to normal following treatment discontinuation. Keywords: Isavuconazole, Chronic pulmonary aspergillosis, Drug-induced liver injury, Late-onset
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