Cerebral pheohyphomycosis due to curvularia species

Curvularia species are soil dwelling dematiaceous fungi that usually infect plants [1]. Infections in humans are rare and usually involve respiratory tract, skin, and cornea [2]. To date, only five cases of central nervous system infection have been published [2], [3]. We report the first case of cerebral Curvularia infection in Italy.

A 68-year-old white man was admitted to our hospital for a transient episode of headache and confusion. For these symptoms, 7 days before admission, he had undergone a cerebral CT that had shown multiple brain lesions. His clinical history was significant for non-Hodgkin lymphoma treated with chemotherapy and radiotherapy two years before the current hospitalization, and apparently in complete remission. The patient was not taking medications, and he denied use of illicit drugs. The clinical examination was unremarkable, as were the routine laboratory exams. A brain MRI showed numerous supratentorial and subtentorial small lesions with post-contrast enhancing ring and restricted diffusion (Fig. 1).

Fig. 1.

Axial T1-weighted fat suppressed ring-enhancing lesions following administration of intravenous gadolinium.

Serum Cryptococcus, galactomannan and beta-d-glucan antigens were negative, as were the search for Mycobacterium tuberculosis and Toxoplasma gondi DNA with the polymerase chain reaction in the cerebrospinal fluid. No lymphomatous cells were detected in the cytologic exam. A biopsy of one of the brain lesions was performed. Meanwhile the patient conditions worsened, and he experienced low-grade fever, headache, confusion, and consciousness deterioration Histological examination showed the presence of granulomatous inflammation, central necrosis, and elements compatible with fungi. The hyphal formations were identified as Curvularia species by means of DNA extraction and genome amplification with the polymerase chain reaction in the histologic specimen. A chest and sinuses CT were normal. Therapy with oral itraconazole and intravenous voriconazole and lisosomial amphotericin b was started, but the patient’s conditions continued to deteriorate, and he died 10 days after the start of treatment. Cerebral phaeohyphomycosis due to Curvularia species is very rare, difficult to diagnose, and often lethal despite appropriate therapy [2]. It almost always affects rural laborers in tropical and subtropical regions through inhalation of the hyphae in the respiratory tract, and subsequent infection of the sinuses [4]. Our patient, however, was not a laborer, and a CT of the sinuses was normal.

Funding

This case report has been undertaken by the authors without any form of sponsorship or support from any individual of institution.

Ethics approval and consent to participate

This case report has obtained approval from the head of the Internal Medicine unit at the Department of Internal Medicine of the Azienda Ospedaliera di Cosenza (Italy). Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

CRediT authorship contribution statement

Bova C: Conception and design of study, drafting the manuscript. Vigna E, Gentile M, Fiaschi E: Review of the manuscript for important intellectual content.

Declaration of Competing Interest

Each author declares that he or she has no commercial associations (e.g., consultancies, stock ownership, equity interest, patent/licensing arrangement etc.) that might pose a conflict of interest in connection with the submitted article.