This case is a presentation of isolated central nervous system (CNS) Mucormycosis in an immunocompetent patient. This case is unique in its demonstration of isolated CNS involvement while lacking clear evidence elucidating an entry point.

The patient is a 36-year-old man without a pertinent past medical history, who initially presented with altered mental status and a 5-day history of progressively slurred speech. His social history is significant for intravenous drug use and outdoor pest control work. The patient’s head computed tomography (CT) scan without contrast demonstrated the presence of possible bilateral infarcts or masses involving the basal ganglia and periventricular white matter. The patient then progressed to facial diplegia with new onset hemiplegia. High-dose steroids were initiated due to concern for neurosarcoidosis. A lumbar puncture was ordered due to minimal improvement and suggested an inflammatory process. A stereotactic brain biopsy was then performed, demonstrating non-caseating granulomatous inflammation with giant cells. Liposomal amphotericin B was added to cover possible fungal etiology. The pathology report was consistent with an isolated cerebral mucormycosis infection. The etiology remained elusive with clear paranasal sinuses and no cutaneous manifestations. Due to extensive gray matter involvement, the patient was not a candidate for surgery.

This is a report of mucormycosis in a seemingly immunocompetent patient with either isolated CNS involvement or disseminated mucormycosis without an identifiable source. Although this patient did have two risk factors including intravenous drug use and outdoor working history, his lack of peripheral involvement demonstrates an uncommon presentation.

  • This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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