Chronic orbital and calvarial fungal infection with Apophysomyces variabilis in an immunocompetent patient

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• 作者：Natalie Wolkow ; MD ; PhD^a ; ^b ; Frederick A. Jakobiec ; MD ; DSc^a ; ^b ; ^c ; ^{Fred_Jakobiec@meei.harvard.edu} ; Anna M. Stagner ; MD^a ; ^b ; ^c ; Mary E. Cunnane ; MD^a ; ^d ; Anne L. Piantadosi ; MD ; PhD^e ; Nesli Basgoz ; MD^e ; Daniel Lefebvre ; MD^a ; ^f
• 关键词：Apophysomyces ; Saksenaea ; mucor ; rhino-orbito-cerebral mucormycosis ; fungus ; calvarial lytic lesions ; amphotericin B ; posaconazole ; diploic venous system
• 刊名：Survey of Ophthalmology
• 出版年：2017
• 出版时间：January-February 2017
• 年：2017
• 卷：62
• 期：1
• 页码：70-82
• 全文大小：2537 K
• 卷排序：62

文摘

Apophysomyces is a rare fungal organism causing rhino-orbito-cerebral mycotic infections with high morbidity and mortality, typically in immunocompetent individuals. Several cases of Apophysomyces elegans orbital disease have been reported. Herein, we report a case of Apophysomyces variabilis infection involving the orbit, sinuses, and calvarium in an immunocompetent 74-year-old woman, with a review of the literature. Unlike prior cases of A. elegans classic rhino-orbito-cerebral infection, our case included diffuse calvarial lytic lesions and overlying soft tissue nodules, but without parenchymal intracranial involvement. There was radiographic and clinical evidence of infarction of the orbital contents and cavernous sinus thrombosis. Anastomoses between the superior orbital (ophthalmic) vein and diploic veins of the calvarium are believed to be primarily responsible for the unusual mode of spread on the extradural surface of the brain. Although the patient stabilized without definitive surgical intervention, her disease slowly and intermittently progressed for over a year after presentation, requiring multiple courses of antifungal treatment.