J Korean Ophthalmol Soc.  2015 Jun;56(6):956-960. 10.3341/jkos.2015.56.6.956.

Successful Treatment of Rhino-Orbital Mucormycosis with Posaconazole after Combination of Surgical Treatment and Amphotericin B

• Kim BM
• Kim KH

• KMID: 2339164
• DOI: http://doi.org/10.3341/jkos.2015.56.6.956

Abstract

PURPOSE
To report a case of rhino-orbital mucormycosis successfully treated by posaconazole salvage therapy after a surgical intervention combined with amphotericin B treatment.
CASE SUMMARY
A 57-year-old female with uncontrolled diabetes mellitus presented to our department with left periorbital swelling and pain for 3 days. At initial presentation, complete ptosis, complete external ophthalmoplegia, and exophthalmos of 6 mm were observed in the left eye. Pupillary reflex was absent in the left eye. Orbital magnetic resonance imaging showed left focal maxillary sinusitis and left posterior orbital inflammatory infiltration with left optic nerve involvement. Nasal endoscopic examination showed black eschar in both nasal cavities. Histopathological examination of the involved nasal cavities showed non-septated fungal hyphae with blunt-angle branching, thus rhino-orbital mucormycosis was diagnosed. The patient underwent a lid-sparing subtotal exenteration, wide endonasal debridement of the nasal necrotic tissues, and intravenous injections of amphotericin B for 7 weeks. The patient continued to receive oral posaconazole salvage therapy for 6 months and was successfully treated. At the 44-month follow-up, recurrence was not observed, and the patient is wearing a new type of silicone oculofacial prosthesis.
CONCLUSIONS
Rhino-orbital mucormycosis is an aggressive, opportunistic fungal infection that is life-threatening despite exenteration and amphotericin B treatment. Sequential use of oral posaconazole as salvage therapy may be helpful for a successful treatment.

Keyword

Mucormycosis; Oculofacial prosthesis; Posaconazole; Salvage therapy

MeSH Terms

Amphotericin B*
Debridement
Diabetes Mellitus
Exophthalmos
Female
Follow-Up Studies
Humans
Hyphae
Injections, Intravenous
Magnetic Resonance Imaging
Maxillary Sinus
Maxillary Sinusitis
Middle Aged
Mucormycosis*
Nasal Cavity
Ophthalmoplegia
Optic Nerve
Orbit
Prostheses and Implants
Recurrence
Reflex, Pupillary
Salvage Therapy
Silicones
Amphotericin B
Silicones

Figure

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  Figure 1. Nine-cardinal photographs at the first visit showing total ophthalmoplegia and conjunctival chemosis of the left eye.

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  Figure 2. Postcontrast (A) axial and (B) coronal orbital magnetic resonance imaging (MRI) demonstrated heterogenous enhancement of the left posterior orbit and the left maxillary sinusitis.

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  Figure 3. Nasal endoscopic photograph showing the black eschar.

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  Figure 4. Microscopic examination showing the wide non-septated hyphae with obtuse angle branching with in the neuronal tissue (Periodic acid Schiff [PAS], ×100) (A) and the vessel (Gomori-methanamine silver stain, ×400) (B).

• 

  Figure 5. Postoperative external photograph wearing a new oculofacial prosthesis with good cosmetic result.

Reference

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