A 58-year-old male was admitted to our hospital for consolidation therapy. He had been in remission from acute myeloid leukemia for 3 months. The patient had suddenly developed persistent fever, headache and facial edema during hospitalization. The hematologist-oncologist suspected fungal infection and consulted with the Department of Otorhinolaryngology. The patient was under diabetes medication, and had received ESS 4 years earlier due to inverted papilloma in the left sinus. There were no specific findings during physical examination, including nasal endoscopy. Paranasal sinus (PNS) CT findings also showed no suspicious lesions that might indicate fungal infection (Fig. 1A-C). Therefore, antibiotics and nasal douching were prescribed for the follow-up period. Five days later, the patient showed chemosis and limitation of the entire visual field in the right eye. Visual acuity measured 0.6 in the right eye and 1.2 in the left eye. On the follow-up PNS CT scan 12 days after the initial scan, sinusitis showed an overall improvement, but the lesion in the right sphenoid sinus remained and the right cavernous sinus showed a density change that indicated obvious obstruction of the right internal carotid artery (ICA) and right cavernous sinus (Fig. 1D and E). Preoperative diagnosis was ‘chronic sinusitis (both),’ ‘orbital apex syndrome (right),’ and ‘suspicion of fungal infection (right).’ Emergency ESS was performed and amphotericin B was initiated with suspicion of mucormycosis. Postoperative brain inner ear MRI with contrast enhancement was performed. This revealed total occlusion in the right cavernous ICA (Fig. 1F). On the 6th day after surgery, invasive aspergillosis was confirmed on biopsy (Fig. 1G and H) and the antifungal agent was changed to voriconazole. Twentyone days after surgery, the patient’s extraocular muscle movement (EOM) was restored to normal, and on the 45th day, endoscopic examination showed no sign of recurrence. A follow-up MRI scan taken 1 month later showed that total occlusion in the right cavernous ICA still existed but mild improvement in thrombophlebitis of the right cavernous sinus was noted. The patient was transferred to another hospital after treatment with the antifungal agent for 3 months and lived for a further 15 months without recurrence.

A 63-year-old male visited the emergency room (ER) complaining of a change in mental wellbeing and right-sided weakness. He had a medical history of hypertension and diabetes mellitus, and was currently taking medication for chronic headaches prescribed by our Department of Neurology. The patient had taken a brain MRI scan 2 months earlier due to chronic headache, and the radiologists interpreted it as a simple chronic sphenoid sinusitis (Fig. 2A and B). A week earlier before his visit to our ER, the patient had been admitted to our Department of Endocrinology due to uncontrolled diabetes and during his hospitalization, he received otolaryngology treatment due to headache suspicious of originating from sinusitis. At the time, the otolaryngologist recommended PNS CT, but the patient had refused to proceed. Upon neurological examination during his present visit in the ER, his mental status was assessed as deep drowsy, with right upper extremity motor: 3/5 grade, right lower extremity motor: 3/5 grade, left upper extremity motor: 4/5 grade, and left lower extremity motor: 4/5 grade. Both feet showed positive Babinski sign with accompanying neck stiffness. A contrast enhanced brain MRI scan was taken, and was reported to show an acute infarct in the left thalamus, left temporal lobe medial portion, internal capsule, left basal ganglia, and left occipital lobe, as well as a density change in the left sphenoid sinus and obstruction in the left ICA (Fig. 2C-E). Spiral CT 3-D common carotid angio + brain perfusion results showed total obstruction in the left ICA, and the left middle cerebral artery was supplied by the anterior communicating artery and posterior communicating artery. During his initial stay in the ER, the neurologist suspected invasive fungal sinusitis and referred him to our department for further evaluation and management.

During endoscopic examination, a whitish discharge was found in both nasal cavities. A PNS CT was taken, and a soft tissue mass was found in the left sphenoid sinus extending to and involving the left cavernous sinus (Fig. 2F and G). The radiologist diagnosed left ICA obstruction with left cavernous sinus involvement, due to mucocele or sinus fungal sinusitis in the left sphenoid sinus. Invasive fungal sinusitis was suspected and an emergency ESS was performed. During the surgery, a fungal ball was found in the left sphenoid, with dehiscence to the ICA wall. In order to confirm invasive fungal sinusitis, a biopsy was taken from the sphenoid wall. Voriconazole was given as antifungal agent, and the biopsy results reported aspergillosis (Fig. 2H and I). The patient has been in a coma for over 5 months after the surgery, and still remains in the neurological intensive care unit even at the time of writing of this paper.

A 61-year-old male visited our outpatient clinic due to rightsided headache with throbbing and tightening pain. He had visited the ER twice over the previous 2 weeks with those symptoms and a brain MRI had been performed which yielded no specific findings. He had merely received medication from the neurology department and had been discharged. The patient had no diplopia or EOM limitation upon physical examination, and had no nasal symptoms. He had a medical history of diabetes and had received chemotherapy for 5 months due to leukemia diagnosed a year earlier. Nasal endoscopic examination showed a white silky discharge in the right sphenoid sinus, and the primary physician suspected right sphenoid sinusitis with fungal ball, prescribing PNS CT with a follow-up appointment.

Four days after visiting our outpatient clinic, the patient visited the neurology clinic due to persistent headache and was admitted to control his symptoms. At admission, the patient was alert and showed no abnormal neurologic signs. During hospital day 2, the patient showed fever and laboratory test results reported white blood cell 1.61×10³/μL, neutrophil 590/μL, high-sensitivity C-reactive protein 117.8 mg/L, and erythrocyte sedimentation rate 43 mm/h. Upon reviewing his past medical records, he was found to have shown continuous neutropenia after receiving chemotherapy.

The neurology department consulted the hemato-oncology department to rule out neutropenic fever, and the patient was transferred to the hemato-oncology department for treatment. Laboratory test results for Aspergillus antigen were positive, and the patient was referred to our department to rule out invasive fungal sinusitis. A PNS CT was performed immediately, and soft density progression was seen in the right sphenoid sinus with bone erosion in the superolateral wall when compared to images taken 8 days earlier at our outpatient clinic (Fig. 3A and B). In addition, mucosal thickening had developed in the left sphenoid sinus which had not been present in previous images.

Emergency ESS was performed and the whole sinus revealed pale cadaveric mucosa. After surgery, Voriconazole was administered as antifungal agent. Histological examination revealed fungal hyphae, morphologically consistent with aspergilloma (Fig. 3C and D). Three days after surgery, diplopia with limitation in the right lateral gaze of the right eye had developed. The patient received antifungal agent for 38 days after surgery and was discharged. He is still alive at the time of writing (28 months after surgery).