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A 46-year-old woman with medical history of poorly controlled diabetes mellitus, including recent hospitalization for diabetic ketoacidosis, presented with left-predominant frontal headache that was not responsive to 1 month of oral antibiotics. Exploration in the operating room demonstrated black necrotic tissue in multiple areas concerning for fungal invasion (Supplemental Figures 1 and 2, available online at http://www.mayoclinicproceedings.org). She was admitted to her local hospital for high suspicion of mucormycosis, and intravenous (IV) liposomal amphotericin was initiated. The next morning, she was found to have new anisocoria with significant ophthalmoplegia of the left eye, consistent with orbital apex syndrome. She was admitted to the Mayo Clinic Hospital St. Marys Campus in Rochester for further management, where emergent magnetic resonance imaging revealed frank invasion at the left rhino-orbital areas despite initial debridement (Figure 1).
Figure 1Gadolinium-enhanced magnetic resonance image, sagittal view, demonstrating extrasinus invasion into the left retromaxillary region, pterygopalatine fossa, and left masticator space, with accompanying axial view demonstrating left orbital apex extension and inflammatory change, affecting the left intraconal orbit.
The patient underwent a total of 5 surgical debridements. Multiple intraoperative specimens demonstrated invasive ribbon-like hyphae and fungal cultures grew Rhizopus spp (Figure 2; Supplemental Figures 3 and 4, available online at http://www.mayoclinicproceedings.org). Antifungal therapies included oral delayed-release posaconazole, IV caspofungin, IV liposomal amphotericin B, amphotericin nasal irrigations, and amphotericin retro-orbital injections. Symptoms, including her vision, improved, and she was discharged to home with oral posaconazole monotherapy.
Figure 2A Grocott's methenamine silver (GMS) stain showing 90-degree branching of ribbon-like hyphae, consistent with mucormycosis.
Mucormycosis, formerly known as zygomycosis, is a category of syndromes characterized by severe invasive fungal infections, generally seen in diabetic or immunocompromised hosts.
Global guidelines for the diagnosis and management of mucormycosis: an initiative of the European Confederation of Medical Mycology in cooperation with the Mycoses Study Group Education and Research Consortium.
The exact incidence and prevalence of infections by these environmental organisms is unknown, but mortality may range from 40% to 80% depending on the syndrome.
Global guidelines for the diagnosis and management of mucormycosis: an initiative of the European Confederation of Medical Mycology in cooperation with the Mycoses Study Group Education and Research Consortium.
Global guidelines for the diagnosis and management of mucormycosis: an initiative of the European Confederation of Medical Mycology in cooperation with the Mycoses Study Group Education and Research Consortium.
Liposomal amphotericin B, delayed-release posaconazole, and isavuconazole are current mainstays of medical therapy, and adjunctive echinocandin therapy has been suggested to be of potential benefit.
Global guidelines for the diagnosis and management of mucormycosis: an initiative of the European Confederation of Medical Mycology in cooperation with the Mycoses Study Group Education and Research Consortium.
MIC distributions and evaluation of fungicidal activity for amphotericin B, itraconazole, voriconazole, posaconazole and caspofungin and 20 species of pathogenic filamentous fungi determined using the CLSI broth microdilution method.
Caspofungin inhibits Rhizopus oryzae 1,3-beta-D-glucan synthase, lower burden in brain measured by quantitative PCR, and improves survival at a low but not a high dose during murine disseminated zygomycosis.
Global guidelines for the diagnosis and management of mucormycosis: an initiative of the European Confederation of Medical Mycology in cooperation with the Mycoses Study Group Education and Research Consortium.
Otorhinolaryngology and infectious disease departments should therefore be consulted emergently if mucormycosis is suspected.
Acknowledgments
We wish to express our sincerest thanks to our fellow colleagues who participated in the care of this patient. We would also like to acknowledge Dr Julie B. Guerin as the reading physician for the MR-Brain and Orbits, conducted as a critical part of the evaluation in our case.
Drs Grach, Yetmar, and DeSimone made substantial contributions to the concept and design of this manuscript and the critical revision of the manuscript for important intellectual content. Dr Rowan made substantial contributions to the acquisition of data and was involved in the final drafting of the manuscript.
Supplemental Online Material
Supplementary Figure 1Picture of the left posterior nasal cavity showing darkening of the mucosa concerning for poor vascularity.
Global guidelines for the diagnosis and management of mucormycosis: an initiative of the European Confederation of Medical Mycology in cooperation with the Mycoses Study Group Education and Research Consortium.
MIC distributions and evaluation of fungicidal activity for amphotericin B, itraconazole, voriconazole, posaconazole and caspofungin and 20 species of pathogenic filamentous fungi determined using the CLSI broth microdilution method.
Caspofungin inhibits Rhizopus oryzae 1,3-beta-D-glucan synthase, lower burden in brain measured by quantitative PCR, and improves survival at a low but not a high dose during murine disseminated zygomycosis.
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