PMC7509339_01
Computed tomography (CT) at the level of the maxillary sinus (A and C and E) and the ethmoid sinus (B and D and F). (A and B) The nasal septum fracture and soft tissue density in sinus cavity were observed at the onset, those were not remarkable changed just before endoscopic sinus surgery (C and D). (E and F) Follow-up CT scan after endoscopic surgery (before additional surgery) showed the removal of fractured nasal septum and the improvement of mucosal thickening.
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At the onset, a computed tomography (CT) scan showed the partial fracture of septum and soft tissue density in the paranasal sinuses (Figure 1A and B). However, the gangrene still continued to expand towards the soft palate despite treatment with L-AMB for two weeks, and, indeed, CT examination showed no major improvement of sinus abnormal lesion (Figure 1C and D). Follow-up CT scan (Figure 1E and F) and MRI (Figure 5C and D) showed no intracranial extension and the gradual improvement of the mucosal thickening, however, he unfortunately experienced a skin perforation of the nasal root after antifungal treatment for seven months, and an additional operation was performed to remove residual necrotic tissue on the palate and paranasal sinuses eight months after his first admission to our hospital.
Endoscopic findings of progressive gangrene. (A) The destroyed nasal cavity was heavily crusted on admission. (B) Necrotic tissue on the oral palate at admission. (C) Sphenoid sinuses and (D) palate defect four months after additional necrotomy. Almost no necrotic tissue remained at either site.
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Eastern Cooperative Oncology Group performance status was grade 3. Endoscopic examination revealed the perforation of nasal septum, large amounts of crusts in the nasal cavity (Figure 2A), and necrotic tissue on the hard to soft palate (Figure 2B). Presently, there is almost no necrotic tissue in his nasal cavity and paranasal sinus (Figure 2C and D) and MRI shows only slight residual mucosal thickening (Figure 5E and F).
Magnetic resonance imaging (MRI) on admission. Horizontal views of (A and D) T1-weighted, (B and E) T2-weighted, and (C and F) FLAIR images at the level of (A-C) the maxillary sinus and (D-F) the eyeballs. The nasal septum was disrupted (A-C) and structural deformation with mucosal thickening and fluid retention were observed in the maxillary, ethmoid and sphenoid sinuses (A-F).
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Magnetic resonance imaging (MRI) showed destruction of the nasal structure with loss of the septum, and abnormal mucosal thickening and mucus retention in the sinuses, but no tumor was detected in the sinonasal region (Figure 3).
Histopathological findings. (A) Infiltrations of Candida species and Actinomycetes in the background of necrotic tissue of the soft palate (hematoxylin and eosin (HE) staining, x 600). (B) Infiltration of Mucor in the background of necrotic tissue of the soft palate (HE, x400). (C-E) Low- (x100) (C) and high- (x400) (D) power magnification views of resected gangrene. Brown spores of Aspergillus niger and surrounding calcium oxalate crystals were identified in the background of necrotic tissue (E).
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Repeated conventional biopsies of small tissue samples from the surface of sinus gangrene using biopsy forceps showed the presence of necrotic tissue with numerous fungi, including Candida species and Mucor, and Actinomycetes, which extended deep below the mucosa (Figure 4A and B). The resected specimen showed extensive infiltration of Aspergillus niger in the background of necrotic tissue, but again without evidence of tumor tissue (Figure 4C-E), based on which we made a final conclusive diagnosis of CIFRS, despite the absence of finding for fungal vessel invasion.
MRI findings along with clinical course. Horizontal views of (A and C and E) T1-weighted, and (B and D and F) T2-weighted images at the level of the maxillary sinus after endoscopic sinus surgery (A and B), before the additional operation (C and D), and five months after the additional operation (E and F).
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MRI after endoscopic surgery showed the removal of septum, but the residual soft tissue (Figure 5A and B). Follow-up CT scan (Figure 1E and F) and MRI (Figure 5C and D) showed no intracranial extension and the gradual improvement of the mucosal thickening, however, he unfortunately experienced a skin perforation of the nasal root after antifungal treatment for seven months, and an additional operation was performed to remove residual necrotic tissue on the palate and paranasal sinuses eight months after his first admission to our hospital. Presently, there is almost no necrotic tissue in his nasal cavity and paranasal sinus (Figure 2C and D) and MRI shows only slight residual mucosal thickening (Figure 5E and F).