A 37-year-old patient with pulmonary mucormycosis who developed epileptic-like seizures during treatment with isavuconazole. The patient had multiple comorbidities, including hypoalbuminemia, hepatic dysfunction, and renal insufficiency and displayed normal mental status before treatment. However, after 27 days of isavuconazole therapy, the patient experienced a sudden onset of unresponsiveness, absence seizures, and body rigidity. Therapeutic drug monitoring revealed a plasma trough concentration of isavuconazole at 6.4 mg·L^-1, the epileptic-like seizures were suspected to be caused by neurotoxicity due to elevated isavuconazole plasma levels. The drug was immediately discontinued, and no further seizures occurred. The Naranjo’s Assessment Scale was used to evaluate the association between adverse reactions and isavuconazole, and the result was "probably relevant". This case highlights the need for caution regarding the potential adverse reaction of seizures when using isavuconazole in clinical practice. This case suggests that clinical use of isavuconazole should be auompanied by vigilance epileptic-like adverse events. Pharmaceutical care and timely analysis of medication use should be carried out, and appropriate symptomatic management should be provided promptly in case of any adverse events.

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