Clinical features, diagnosis, and outcomes of rhino-orbito-cerebral mucormycosis- A retrospective analysis

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Abstract

Background and Purpose:

Rhino-orbito-cerebral mucormycosis (ROCM) is a rare disease with acute and fulminant manifestation. This infection is associated with high morbidity and mortality rates. Herein, we reviewed the manifestations, underlying conditions, medical treatments, and surgical interventions in ROCM patients admitted to a tertiary referral center in northern Iran over a seven-year period.

Materials and Methods:

In a retrospective analysis, 15 cases of ROCM were identified from 2007 to 2013 in Bu Ali Sina Hospital, Sari, Iran. All the ROCM cases were clinically diagnosed and confirmed by histopathological and/or mycological examination. The relevant demographic data, clinical, ophthalmic, and neurologic manifestations, underlying conditions, medical treatments, and surgical interventions were recorded and analyzed.

Results:

The mean age of the patients was 54±11 years (age range: 28–70 years); 26.7% of the patients were male and 73.3% female (male: female ratio of 1: 2.7). Uncontrolled diabetes was noted in at least 86.7% (13/15) of the cases. The maxillary sinuses were the most frequently involved sites (66.7% of the cases) followed by the ethmoid sinus. Amphotericin B in combination with surgical debridement was used in the treatment of 80% of the cases. Furthermore, 73.3% of the patients who were diagnosed early and underwent medical and extensive surgical debridement of the infected tissues survived.

Conclusion:

Uncontrolled diabetes mellitus is considered to be the main predisposing factor for ROCM. To prevent and reduce mortality rate of this acute disease, early diagnosis based on clinical findings and biopsy is recommended.

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Most cited references 68

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Novel perspectives on mucormycosis: pathophysiology, presentation, and management.

Brad Spellberg, John Edwards, Ashraf Ibrahim (2005)

Mucormycosis is a life-threatening fungal infection that occurs in immunocompromised patients. These infections are becoming increasingly common, yet survival remains very poor. A greater understanding of the pathogenesis of the disease may lead to future therapies. For example, it is now clear that iron metabolism plays a central role in regulating mucormycosis infections and that deferoxamine predisposes patients to mucormycosis by inappropriately supplying the fungus with iron. These findings raise the possibility that iron chelator therapy may be useful to treat the infection as long as the chelator does not inappropriately supply the fungus with iron. Recent data support the concept that high-dose liposomal amphotericin is the preferred monotherapy for mucormycosis. However, several novel therapeutic strategies are available. These options include combination therapy using lipid-based amphotericin with an echinocandin or with an azole (largely itraconazole or posaconazole) or with all three. The underlying principles of therapy for this disease remain rapid diagnosis, reversal of underlying predisposition, and urgent surgical debridement.

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Zygomycetes in human disease.

J A Ribes, C L Vanover-Sams, D Baker (2000)

The Zygomycetes represent relatively uncommon isolates in the clinical laboratory, reflecting either environmental contaminants or, less commonly, a clinical disease called zygomycosis. There are two orders of Zygomycetes containing organisms that cause human disease, the Mucorales and the Entomophthorales. The majority of human illness is caused by the Mucorales. While disease is most commonly linked to Rhizopus spp., other organisms are also associated with human infection, including Mucor, Rhizomucor, Absidia, Apophysomyces, Saksenaea, Cunninghamella, Cokeromyces, and Syncephalastrum spp. Although Mortierella spp. do cause disease in animals, there is no longer sufficient evidence to suggest that they are true human pathogens. The spores from these molds are transmitted by inhalation, via a variety of percutaneous routes, or by ingestion of spores. Human zygomycosis caused by the Mucorales generally occurs in immunocompromised hosts as opportunistic infections. Host risk factors include diabetes mellitus, neutropenia, sustained immunosuppressive therapy, chronic prednisone use, iron chelation therapy, broad-spectrum antibiotic use, severe malnutrition, and primary breakdown in the integrity of the cutaneous barrier such as trauma, surgical wounds, needle sticks, or burns. Zygomycosis occurs only rarely in immunocompetent hosts. The disease manifestations reflect the mode of transmission, with rhinocerebral and pulmonary diseases being the most common manifestations. Cutaneous, gastrointestinal, and allergic diseases are also seen. The Mucorales are associated with angioinvasive disease, often leading to thrombosis, infarction of involved tissues, and tissue destruction mediated by a number of fungal proteases, lipases, and mycotoxins. If the diagnosis is not made early, dissemination often occurs. Therapy, if it is to be effective, must be started early and requires combinations of antifungal drugs, surgical intervention, and reversal of the underlying risk factors. The Entomophthorales are closely related to the Mucorales on the basis of sexual growth by production of zygospores and by the production of coenocytic hyphae. Despite these similarities, the Entomophthorales and Mucorales have dramatically different gross morphologies, asexual reproductive characteristics, and disease manifestations. In comparison to the floccose aerial mycelium of the Mucorales, the Entomophthorales produce a compact, glabrous mycelium. The asexually produced spores of the Entomophthorales may be passively released or actively expelled into the environment. Human disease with these organisms occurs predominantly in tropical regions, with transmission occurring by implantation of spores via minor trauma such as insect bites or by inhalation of spores into the sinuses. Conidiobolus typically infects mucocutaneous sites to produce sinusitis disease, while Basidiobolus infections occur as subcutaneous mycosis of the trunk and extremities. The Entomophthorales are true pathogens, infecting primarily immunocompetent hosts. They generally do not invade blood vessels and rarely disseminate. Occasional cases of disseminated and angioinvasive disease have recently been described, primarily in immunocompromised patients, suggesting a possible emerging role for this organism as an opportunist.

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Pathogenesis of mucormycosis.

Ashraf S Ibrahim, Brad Spellberg, Thomas Walsh … (2012)

Mucormycosis is a life-threatening infection that occurs in patients who are immunocompromised because of diabetic ketoacidosis, neutropenia, organ transplantation, and/or increased serum levels of available iron. Because of the increasing prevalence of diabetes mellitus, cancer, and organ transplantation, the number of patients at risk for this deadly infection is increasing. Despite aggressive therapy, which includes disfiguring surgical debridement and frequently adjunctive toxic antifungal therapy, the overall mortality rate is high. New strategies to prevent and treat mucormycosis are urgently needed. Understanding the pathogenesis of mucormycosis and the host response to invading hyphae ultimately will provide targets for novel therapeutic interventions. In this supplement, we review the current knowledge about the virulence traits used by the most common etiologic agent of mucormycosis, Rhizopus oryzae. Because patients with elevated serum levels of available iron are uniquely susceptible to mucormycosis and these infections are highly angioinvasive, emphasis is placed on the ability of the organism to acquire iron from the host and on its interactions with endothelial cells lining blood vessels. Several promising therapeutic strategies in preclinical stages are identified.

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Author and article information

Journal

Journal ID (nlm-ta): Curr Med Mycol

Journal ID (iso-abbrev): Curr Med Mycol

Journal ID (publisher-id): CMM

Title: Current Medical Mycology

Publisher: Iranian Society of Medical Mycology (Sari, Iran )

ISSN (Print): 2423-3439

ISSN (Electronic): 2423-3420

Publication date (Print): December 2016

Volume: 2

Issue: 4

Pages: 15-23

Affiliations

[1 ]Student Research Committee, Faculty of Medicine, Mazandaran University of Medical Sciences, Sari, Iran

[2 ]Department of Medical Mycology and Parasitology, School of Medicine, Mazandaran University of Medical Sciences, Sari, Iran

[3 ]Invasive Fungi Research Center (IFRC), Mazandaran University of Medical Sciences, Sari, Iran

[4 ]Department of Medical Laboratory Sciences, School of Paramedicine/Infectious and Tropical Diseases Research Center, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran

[5 ]Department of Otolaryngology, School of Medicine, Mazandaran University of Medical Sciences, Sari, Iran

[6 ]Department of Pathology, School of Medicine, Mazandaran University of Medical Sciences, Sari, Iran

[7 ]Department of Medical Mycology and Parasitology, School of Medicine, Arak University of Medical Sciences, Arak, Iran

[8 ]Division of Mycology, Department of Microbiology, School of Medicine, University of Çukurova, Adana, Turkey

Author notes

[* ]Corresponding author: Tahereh Shokohi, Department of Medical Mycology and Parasitology, Invasive Fungi Research Center (IFRC), School of Medicine, Mazandaran University of Medical Sciences, Sari, Iran. Email: shokohi.tahereh@gmail.com

Article

DOI: 10.18869/acadpub.cmm.2.4.15

PMC ID: 5611692

PubMed ID: 28959791

SO-VID: 818129e3-7f32-4f8e-9c91-b1737719ad7d

License:

This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial 4.0 International License ( http://creativecommons.org/licenses/bync/4.0/) which permits copy and redistribute the material just in noncommercial usages, provided the original work is properly cited.

History

Date received : 2 May 2017

Date revision received : 20 June 2017

Date accepted : 25 June 2017

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Clinical features, diagnosis, and outcomes of rhino-orbito-cerebral mucormycosis- A retrospective analysis

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Abstract

Background and Purpose:

Materials and Methods:

Results:

Conclusion:

Related collections

GLUT4 biology

Most cited references 68

Novel perspectives on mucormycosis: pathophysiology, presentation, and management.

Zygomycetes in human disease.

Pathogenesis of mucormycosis.

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Cited by 21

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