Case Presentation: A 44 year old African American man presented to the hospital for evaluation and treatment following generalized tonic-clonic seizures. He denied recent and remote travel, fever, chills, sick contacts, constitutional symptoms, and felt relatively well at time of interview. He was on maintenance immunosuppression with prednisone 20 mg daily for sarcoidosis. MRI demonstrated extensive, bilateral leptomeningeal nodular enhancement with adjacent vasogenic edema. The patient underwent lumbar puncture, which was significant for 10 WBCs/cu mm (atypical lymphocytes and monocytes), elevated protein (169 mg/dl), normal glucose with negative smears and cultures. Neurosurgery was consulted and performed right frontal craniotomy and biopsy, which revealed multiple granulomas, foci of central hyalinization, and negative acid-fast and fungal stains. Findings were consistent with sarcoidosis, but infection could not be excluded. (1,3)-Beta-D-Glucan was positive at 494 pg/ml, and serum galactomannan index was positive at 2.72. Because of these results and the potential severity of illness, the patient was treated with empirical liposomal amphotericin B, developed acute kidney injury, and was transitioned to voriconazole. Ten days following surgery, cultures of brain tissue grew Penicillium species. After six days of treatment, repeat galactomannan assay was negative (0.07). The patient was discharged from the hospital and instructed to continue voriconazole 200mg orally twice daily for 3 months. After two weeks of therapy, the patient’s (1,3)-Beta-D-Glucan was negative as was the final (1,3)- Beta-D-Glucan after 3 months of treatment.

Discussion: Penicilliosis has clinical manifestations and histopathologic findings similar to histoplasmosis. Typically by the time diagnosis is established, the disease has disseminated and can be identified in bone marrow, liver, spleen, skin or bone with definitive diagnosis made by culture. Growth typically takes four to seven days, but may take up to a few weeks. When infection is suspected, initiation of treatment should be immediate as mortality rates can be as high as 97%. Appropriate agents for treatment include liposomal amphotericin B, itraconazole, or voriconazole resulting in resolution of infection in up to 95% of patients. Rarely reported cases in the literature will be presented.

Conclusions: Penicillium is a diverse genus with over 300 species, of major importance in food spoilage, and frequently thought of as an environmental contaminant when grown in the laboratory. In patients with immunocompromise, this group of organisms must be considered pathogenic until proven otherwise.