ABSTRACT
Background: Cladophialophora bantiana is a dematiaceous fungus that rarely infects the central nervous system (CNS). It is associated with a mortality rate of over 70% despite treatment. Case Description: An 81-year-old female with a remote history of renal cell carcinoma presented with progressive headache and an expressive aphasia for 3 days. Computed tomography imaging revealed a left frontotemporal mass with surrounding vasogenic edema. A left frontotemporal craniotomy was performed and cultures revealed C. bantiana. The initial management with IV voriconazole was unsuccessful and the patient had a recurrence of the cranial infection and developed pulmonary abscesses. Following the addition of oral flucytosine, the patient showed a significant improvement with a complete radiographic resolution of both the cranial and pulmonary lesions. Conclusion: C. bantiana involving the CNS is a rare and often fatal disease. Surgical management along with standard antifungal treatment may not provide definitive therapy. The addition of flucytosine to IV voriconazole resulted in a positive outcome for this patient who is alive, living independently 1 year from the original diagnosis. In this rare fungal infection, standard antifungal treatment may not provide adequate coverage and the utilization of additional therapy may be required.