Chronic invasive fungal sinusitis with orbital and olfactory cleft involvement secondary to indolent mucormycosis

Abstract

Purpose: Chronic invasive fungal sinusitis secondary to indolent mucormycosis is a rare clinical entity, and the ideal management is controversial. A case of indolent mucormycosis successfully managed with conservative debridement and retrobulbar amphotericin B is herein reported.

Observations: A 42-year-old man with diabetes mellitus and kidney transplant presented with chronic invasive fungal sinusitis with left orbital involvement from indolent mucormycosis. The patient was treated with aggressive systemic antifungal therapy, left retrobulbar injection of liposomal amphotericin B, reduction in immunosuppression, and conservative surgical debridement. Although the left olfactory cleft was involved, the cribriform plate was not resected due to risk of seeding the intracranial space. Given mild orbital involvement, no orbital debridement was performed and the patient had resolution of his orbital findings with systemic and retrobulbar amphotericin B. The patient had clinical and radiographic stability at 6-month follow-up.

Conclusions: Conservative resection with subsequent long-term antifungal treatment can be a successful regimen in indolent mucormycosis. Retrobulbar amphotericin B may be a prudent orbit-sparing adjuvant therapy in indolent mucormycosis.

Keywords: Chronic invasive fungal sinusitis; Conservative debridement; Fungal orbital cellulitis; IFS, Invasive Fungal Sinusitis; Indolent mucormycosis; TRAMB, transcutaneous retrobulbar injection of amphotericin B; Transcutaneous retrobulbar injection of amphotericin B.

Fig. 1

CT imaging prior to diagnosis (A,C) and at time of diagnosis (B,D) showing unilateral soft tissue thickening within the left nasal cavity with new bone erosion at the anterior skull base on the follow-up imaging (white arrows).

Fig. 2

A. Periorbital edema including left upper eyelid secondary to left orbital involvement by invasive fungal rhinosinusitis, prior to initiation of therapy. B. Two days after left transcutaneous retrobulbar injection of amphotericin B, the patient had marked improvement in his upper eyelid edema (as well as associated orbital signs), but developed a fluctuant, watery lower eyelid edema – qualitatively different from earlier edema, and likely representing local inflammatory tissue reaction to retrobulbar amphotericin B. C. Five days after retrobulbar amphotericin B, the watery lower eyelid edema had resolved.

Fig. 3

MRI with contrast at time of diagnosis demonstrating T2 hypointense soft tissue thickening within the nasal cavity extending to the superomedial orbit (A,B,C F). The soft tissue thickening demonstrates “lack of enhancement” (asterisk in B,C &F) and diffusion restriction (D&E). There is accompanying dural enhancement (arrows in B&C).

Fig. 4

MRI from follow-up at one month (A,B) and five months (C,D). Images show progressive resolution of T2 hypointense soft tissue thickening (A, C). Post-contrast images (B, D) show resolution of dural enhancement (arrows).