Voriconazole is the standard treatment for invasive aspergillosis but requires therapeutic drug monitoring for optimizing therapy. We report two cases of central nervous system aspergillosis treated with voriconazole. Because of low trough plasma concentrations, we identified gain-of-function mutations of CYP2C19 that were partially responsible for therapeutic failure of voriconazole. We suggest systematic voriconazole pharmacogenomic investigation in cerebral aspergillosis to avoid effective therapy delay in this life-threatening disease.
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