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      An indolent case of isolated cerebral mucormycosis: an uncommon presentation

      case-report

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          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.

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          Most cited references5

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          A mechanism of susceptibility to mucormycosis in diabetic ketoacidosis: transferrin and iron availability.

          The defect in host defense that makes the diabetic ketoacidotic (DKA) patient susceptible to mucormycosis has not been identified. Sera from 10 DKA patients and three normal volunteers were tested for their capacity to support the in vitro growth of a common etiologic agent of mucormycosis, Rhizopus oryzae. After equilibration with room air none of the normal or DKA sera, each of which was now extremely alkaline, supported growth of R. oryzae. When the sera were placed in a CO2 atmosphere that permitted simulation of the in vivo clinical pH (normal 7.40 and DKA 7.3-6.6), four of seven DKA sera supported profuse fungal growth. No growth occurred in normal serum. The three DKA sera that did not support fungal growth at pH less than or equal to 7.3 contained less iron (x = 13 micrograms/dl) than the four sera that supported profuse fungal growth (x = 69 micrograms/dl). Increasing the iron content of iron-poor DKA serum that did not support R. oryzae growth allowed profuse growth at acidotic conditions but not at pH greater than or equal to 7.4. Simulated acidotic conditions (pH 7.3-6.6) also decreased the iron-binding capacity of normal serum stepwise from 266 micrograms/dl to 0. Our data indicate that acidosis temporarily disrupts the capacity of transferrin to bind iron and suggest that this alteration abolishes an important host defense mechanism that permits growth of R. oryzae.
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            Retrospective Clinical Study of Eighty-One Cases of Intracranial Mucormycosis

            Background: Fungal infections of the central nervous system, especially cerebral mucormycosis or brain abscess are very rare.Cerebral mucormycosis is a rare disease. It is not an independent disease, but a secondary opportunistic infectious disease. Materials and methods: This study has collected the data of 81 cases of intracranial mucormycosis from 28 Chinese hospitals, within 37 years, as well as reviewed the literatures and retrospectively analyzed and summarized this disease's background, clinical classifications, risk factors, pathology, clinical manifestations, diagnosis, treatment, and prognosis. Results: The 81 IM cases were aged between 15 days (the youngest) and 79 years (oldest), with a mean age of 41.6 years. Among them, 12 cases were 14 years old (the adult group ). 45 cases were male and 36 were female, with a male/female ratio of 1.25:1.0. The shortest duration of the disease was three days, and the longest was 248 days. Conclusions: This study helped to realize an early diagnosis and treatment, improve the cure rate, and reduce mortality.
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              Indolent intracranial mucormycosis: case report.

              We present a patient with isolated intracranial mucormycosis that remained indolent for several months. The mucormycosis formed a mass in the prepontine cistern, probably spreading from the sphenoid sinus. We propose that at least some cases of what is considered isolated CNS mucormycosis develop from such unrecognized indolent infections. Mucormycosis may present as a mass in the basilar cisterns, especially in immunocompromised patients.
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                Author and article information

                Journal
                Access Microbiol
                Access Microbiol
                acmi
                acmi
                Access Microbiology
                Microbiology Society
                2516-8290
                2019
                7 May 2019
                7 May 2019
                : 1
                : 7
                : e000023
                Affiliations
                [ 1] Mercer University School of Medicine , Georgia, USA
                [ 2] Memorial Health University Medical Center, Department of Internal Medicine , Savannah, Georgia, USA
                [ 3] Memorial Health University Medical Center, Department of Pathology , Savannah, Georgia, USA
                Author notes
                *Correspondence: Randi S. Goldstein, Randi.Goldstein@ 123456live.mercer.edu
                Author information
                https://orcid.org/0000-0002-0723-3777
                https://orcid.org/0000-0003-2227-3475
                Article
                000023
                10.1099/acmi.0.000023
                7481736
                32974538
                d612f684-bf1f-4a55-909d-0de5724bfb0d
                © 2019 The Authors

                This is an open-access article distributed under the terms of the Creative Commons Attribution License.

                History
                : 02 January 2019
                : 18 March 2019
                Categories
                Case Report
                Custom metadata
                0

                intracranial mucormycosis,intracerebral mucormycosis,mucormycosis,isolated mucormycosis,atypical mucormycosis presentation

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