Invasive mucormycosis of the maxillary sinus: extensive destruction with an indolent presentation.

BACKGROUND

Mucormycosis (zygomycosis) is a rare, aggressive, invasive fungal infection that usually afflicts immunosuppressed patients. Indolent presentations are rare, especially in the setting of immune suppression.

METHODS

Case report and review of the pertinent English-language literature.

CASE REPORT: A 64-year-old male patient with diabetes mellitus and rheumatoid arthritis, treated chronically with infliximab, presented with toothache, headache, and right facial numbness. Therapy with intravenous glucocorticoids and antibiotics resulted in transitory improvement before his referral to a tertiary-care center, where imaging studies and biopsy revealed rhinocerebral mucormycosis. Four weeks after initial presentation, a radical right maxillectomy, followed by long-term therapy with amphotericin B lipid complex, resulted in clinical improvement. Five reconstructive procedures were required to obliterate the facial defect and restore contour. Although biopsies during the reconstructive procedures revealed persistent fungal colonization, there was no clinical recurrence during nearly five years of followup.

CONCLUSIONS

Indolent rhinocerebral mucormycosis is rare and is seldom survived by immunosuppressed patients. Multimodal therapy with surgical debridement and antifungal chemotherapy is required for an optimal outcome. Discontinuance of immunosuppressive therapy, if possible, is a cornerstone of management.