Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2022 May 4:13:815902.
doi: 10.3389/fneur.2022.815902. eCollection 2022.

Rhino-Orbital Cerebral Mucormycosis in a Patient With Diabetic Ketoacidosis: A Case Report and Literature Review

Nan Dong  1   2 , Ashly E Jordan  3 , Xiaozhu Shen  1 , Xuan Wu  1 , Xianghong Guo  2 , Hongru Zhao  1 , Yajuan Wang  4 , Dapeng Wang  1 , Qi Fang  1
Affiliations
Case Reports

Rhino-Orbital Cerebral Mucormycosis in a Patient With Diabetic Ketoacidosis: A Case Report and Literature Review

Nan Dong et al. Front Neurol. .

Abstract

Background: Rhino-orbital cerebral mucormycosis (ROCM) is a rare, invasive, and fatal fungal disease. Due to the lack of specific clinical manifestations and adequate auxiliary examinations, patients are easily misdiagnosed in the early stage. Early diagnosis and timely therapy are essential for successful treatment.

Case report: We report a 68-year-old man with diabetic ketoacidosis, presented with orbital apex syndrome (OAS), fever, and pansinusitis, which progressively worsened to death only 4 days after admission. It was finally confirmed as a fungal Rhizopus arrhizus infection by metagenomics cell-free DNA next-generation sequencing (mNGS) testing.

Conclusion: Orbital apex syndrome could be the initial presentation for mucormycosis. Thus, it is necessary to evaluate the presence of mucormycosis in patients with OAS, especially in diabetic or immunosuppressed hosts, and mNGS testing and timely antifungal therapy should be strongly recommended in highly suspected cases.

Keywords: diabetes; ketoacidosis; metagenomics cell-free DNA next-generation sequencing; orbital apex syndrome; rhino-orbital cerebral mucormycosis.

PubMed Disclaimer

Conflict of interest statement

YW was employed by Genoxor Medical Science and Technology Inc. The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Figures

Figure 1
Figure 1
Gadolinium-enhanced brain magnetic resonance imaging (MRI) revealed bilateral pansinusitis and multiple lesions scattered in the right cerebral hemisphere and right pons, showing hypointensity on T1-weighted images, and hyperintensity on T2-weighted and fluid attenuated inversion recovery (FLAIR) images, without contrast enhancement.
Figure 2
Figure 2
Result of metagenomic NGS. (A) A total of 78 DNA reads mapped to Rhizopus arrhizus in cerebrospinal fluid. (B) A total of 131 DNA reads mapped to Rhizopus arrhizus in blood.
See this image and copyright information in PMC

References

    1. Chegini Z, Didehdar M, Khoshbayan A, Rajaeih S, Salehi M, Shariati A. Epidemiology, clinical features, diagnosis and treatment of cerebral mucormycosis in diabetic patients: a systematic review of case reports and case series. Mycoses. (2020) 63:1264–82. 10.1111/myc.13187 - DOI - PubMed
    1. Bays DJ, Thompson GR. Fungal infections of the stem cell transplant recipient and hematologic malignancy patients. Infect Dis Clin North Am. (2019) 33:545–66. 10.1016/j.idc.2019.02.006 - DOI - PubMed
    1. Álvarez F, Fernández-Ruiz M, Aguado JM. [Iron and invasive fungal infection]. Rev Iberoam Micol. (2013) 30:217–25. 10.1016/j.riam.2013.04.002 - DOI - PubMed
    1. Skiada A, Pavleas I, Drogari-Apiranthitou M. Epidemiology and diagnosis of mucormycosis: an update. J Fungi 2020; 6. 10.3390/jof6040265 - DOI - PMC - PubMed
    1. Elinav H, Zimhony O, Cohen MJ, Marcovich AL, Benenson S. Rhinocerebral mucormycosis in patients without predisposing medical conditions: a review of the literature. Clin Microbiol Infect. (2009) 15:693–7. 10.1111/j.1469-0691.2009.02884.x - DOI - PubMed

Publication types