An indolent case of isolated cerebral mucormycosis: an uncommon presentation

Abstract

Introduction 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. Case presentation 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. Conclusion 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.

Immunocompetent isolated cerebral mucormycosis presenting with obstructive hydrocephalus: illustrative case

“…To our knowledge, four additional cases of isolated intracerebral mucormycosis that would have met their inclusion criteria have been published since. 9 , 18–20 We believe that our case marks the 60th case and, notably, is the only one in the modern era of MRI with obstructive hydrocephalus but without specific infectious radiographic findings.…”

Bilateral Cerebral Mucormycosis in an Immunocompetent Female

“…20 The presentation was uncommon because despite his known social risk factor of IV substance abuse, the patient had no evidence of expected cutaneous manifestations or rhinocerebral involvement. 20 Multiple cases of cerebral mucormycosis have been reported after tooth extractions. 21 Dental surgical trauma may compromise local vascularity and provide an entry port for the microorganisms.…”

“…19 Even in the presence of risk factors such as IV drug use, the route of infection is not always clear. 20 In Montgomery et al, an immunocompetent 36-year-old male with a history of IV substance abuse presented with isolated intracranial mucormycosis involving the right basal ganglia and associated cerebral edema. 20 The presentation was uncommon because despite his known social risk factor of IV substance abuse, the patient had no evidence of expected cutaneous manifestations or rhinocerebral involvement.…”

“…12 Uncertainties and lack of early diagnostic tools cause delays in initiating appropriate antifungal treatment which likely contribute to poor outcomes in mucormycosis. 20 Molecular diagnostic methods are likely more effective than tissue biopsy and histopathology in the identification of Mucorales and may be useful in the diagnosis of microscopic pathogens. 27 Zhang et al emphasize the application of next-generation sequencing technology in early stages of intracranial mucormycosis to provide earlier identification and therefore improve treatment and prognosis for patients, 28 showing that other investigative methods may prove difficult (the positive culture rate is only 50% for all types of mucormycosis with a confirmed pathologic diagnosis and only 38% for cerebral mucormycosis).…”

COVID-19-Associated mucormycosis: Case series from a tertiary care hospital in South India

“…Mucormycosis is an opportunistic fungal infection caused by genus Rhizopus, Mucor , Rhizomucor, Cunningamella, Licthemia, Syncephalastrum, Sakseneae, and Cokeromyces of order Mucorales and class Zygomycetes. These are saprophytic fungi found in soil and the environment [ 5 ]. The clinical types of mucormycosis include pulmonary mucormycosis, gastrointestinal mucormycosis, cutaneous mucormycosis, rhino-orbito-cerebral mucormycosis and disseminated mucormycosis [ 6 ].…”