To describe a rare case of cerebral phaeohyphomycosis.

Central nervous system (CNS) fungal infections are unusual. Curvularia is a filamentous, dematiaceous fungus ubiquitously found in soil around the world, which rarely affects humans.

Here we present a case of a patient with fulminant phaeohyphomycosis in the setting of immunosuppression.

A 42-year-old woman, immunosuppressed after cardiac transplant 1-month prior, was initially hospitalized to the Surgical ICU with decompensated heart failure. On day 63 of admission, neurology team was consulted due to an acute decline in mentation. Examination at that time revealed left-gaze deviation and right-sided hemiparesis. CT scan of the brain and subsequent imaging visualized a rapidly expanding left frontoparietal mass lesion.  Empiric steroids, antibiotics and antifungal agents were started empirically, however patient continue to deteriorate and died on day 80 of her admission due to a herniation syndrome. Biopsy of the lesion was diagnostic for fulminant cerebral phaeohyphomycosis due to Curvularia species, a ubiquitous dematiaceous fungus, with less than 10 cases of brain involvement reported. To our knowledge, this is the first transplanted patient to have such opportunistic infection.

We reported an unusual central nervous system fungal infection following cardiac transplant. Clinical recognition and initiation of aggressive antibiotic therapy are critical.