Objective To explore the adverse drug event (ADE) signals of isavuconazole using data from the US FDA Adverse Event Reporting System (FAERS) database, providing a basis for rational drug use in clinical settings. Methods Data on adverse events in the FAERS database from the first quarter of 2004 to the third quarter of 2023 (79 quarters) were extracted. Signal mining was conducted using the reporting odds ratio (ROR) combined with the proportional reporting ratio (PRR). Results A total of 2,065 cases of adverse events with isavuconazole as the primary suspected drug were identified. Analysis revealed 76 valid preferred term (PT) signals involving 14 system organ classifications (SOCs). The most common ADEs included hepatotoxicity and infections and infestations, which may be related to fungal breakthrough infections. Conclusion ADE signals of isavuconazole can be mined and analyzed based on the FAERS database, providing a basis for safe and rational clinical drug use.