A 69-year-old Japanese woman had a macular oedema in her right eye, which occurred secondary to BRVO and was unresponsive to intravitreal injection of anti-vascular endothelial growth factor aflibercept. She was subsequently treated by STTA injection at the referral hospital twice. During the treatments, her eye was anaesthetized with 4% topical lidocaine. A sterilized eyelid speculum was used, and the superior-temporal conjunctiva was incised using sterilized scissors to inject 20 mg of triamcinolone acetonide into the posterior subtenon space. After the injection, the conjunctiva was not sutured because this step is usually unnecessary. These procedures were performed in an outpatient treatment room with wearing sterilized gloves. A 1.5% topical levofloxacin ophthalmic solution was administered four times daily for 3 days post-injection. Four months after the second injection, the patient noted a conjunctival mass that became progressively larger with a little pain and redness. She was then referred to Kindai University with a clinical diagnosis of orbital fat herniation.\nDuring her first visit, best-corrected visual acuity (BCVA) was 6/30 in the right eye and 6/6 in the left eye. Intraocular pressure was normal in both eyes. Slit-lamp microscopic examination revealed no inflammation in both the anterior chamber and vitreous. A soft, well-demarcated mass mimicking herniated orbital fat was observed in the super-temporal quadrant, which corresponded to the posterior STTA site (Fig. a). Magnetic resonance imaging (MRI) of the orbits revealed a hypo-intensity lesion on T2-weighted imaging at the super-temporal peribulbar area (Fig. b). Her white cell count and C reactive protein levels were normal.\nTo excise the conjunctival mass, first, we made an incision in the conjunctiva, and found that yellowish pus was discharged. The local administration of antibiotics was performed empirically based on the presumed diagnosis of bacterial infection. Although a strain of dematiaceous fungi was isolated from the pus 1 month later, the antifungal drug susceptibility of the strain was not determined at that time. We did not immediately administer antifungal drugs because there were no significant changes in clinical findings initially. The abscess was debrided again 2 months after the surgery through a superior forniceal incision because the orbital abscess at the same lesion had developed gradually. During the second surgery, the wound was irrigated with 0.1% amphotericin B after three large abscesses were removed. Postoperatively, she was treated with 400 mg/day of oral voriconazole because of Fungiflora Y staining of the pus (Fig. a) and the presence of a large amount of fungi by pathological examination of the abscess by Grocott’s staining (Fig. b).\nWe obtained the pathogenic strain from the abscess during the second surgery and observed it macroscopically and microscopically. The colony on potato dextrose agar medium (PDA; Difco, Becton, Dickinson and Company, Sparks, USA) had a diameter of 10 mm 14 days after inoculation and with incubation at 25 °C. The colony appeared as a moist-form pigmented yeast-like colony (Fig. a). The slide culture using lactophenol cotton blue staining revealed cylindrical conidogenous cells producing ellipsoidal conidia (Fig. b). From the Basic Local Alignment Search Tool search using the sequence of the ribosomal RNA gene internal transcribed spacer domain base, the sequence (the accession number LC566592 in the DDBJ) of this strain showed 100% similarity to those of some strains of E. dermatitidis. The isolate was thus identified as E. dermatitidis on the basis of morphology and phylogeny. The pathogenic strain in the current case was deposited at the Medical Mycology Center, Chiba University in Japan, as IFM 65961.\nMinimum inhibitory concentrations (MICs) of several antifungal drugs and the minimal effective concentration of micafungin against the strain were determined using of the broth dilution method according to M38-A2 of the Clinical and Laboratory Standard Institute (Table ). The inhibitory concentrations of amphotericin B, itraconazole, voriconazole, and miconazole against the pathogenic strain were found to be low. Based on these results, topical and systemic voriconazole were administered for 3 months. The patient’s serum beta-D-glucan levels were normal during the follow-up periods since the first surgery, and no recurrence was observed after the second drainage.
A 48-year-old man with diabetes mellitus who had failed to adhere to his treatment presented to the emergency department with complaints of a right-sided headache and toothache for two weeks, with nausea and vomiting for two days prior to presentation. The patient was managed for septic shock secondary to dental abscesses. Non-contrast brain computed tomography (CT) was performed. The CT scan showed no significant intracranial abnormality other than pansinusitis, without obvious retro-orbital fat streakiness (Figure ). Four days later, tooth number 17 and 18 in the left mandibular region, both of which had abscesses, were extracted. The patient noted progressive painless blurring of the vision in his right eye post-extraction of the abscessed teeth and was referred to the ophthalmology team two days later. At this time, the patient was receiving day five of treatment with intravenous (IV) amoxicillin-clavulanate, (1.2 g three times a day), with the addition of IV metronidazole (400 mg twice a day).\nThe visual acuity (VA) in his right eye was 4/60, with no improvement in the pinhole test. A relative afferent pupillary defect was present, with an associated decrease in the optic nerve function, in addition to ophthalmoplegia and restrictions in the superior and lateral gaze. No proptosis, ptosis or lid swelling was present, and all other anterior segment findings were normal. A fundus examination of the right eye showed only proliferative diabetic retinopathy. The VA in the patient’s left eye was 6/18 and 6/9 in the pinhole test. An examination of the anterior segment was unremarkable. Moderate non-proliferative diabetic retinopathy was detected in the posterior segment. Dental caries were present in the upper molars, with mucopurulent discharge from the right sphenoid sinus region. The clinical diagnosis was OAS, and emergency functional endoscopic sinus surgery and septoplasty were performed by the ear, nose and throat (ENT) team. Intra-operatively, pus (6cc) was drained from the right sphenoid sinus. The pus breached the anterior sphenoid wall, tracked down to the septum polypoidal mucosa over the right maxillary sinus and right anterior and posterior ethmoid sinuses.\nEmpirical treatment with IV ceftriaxone (1 g twice a day) and IV fluconazole (400 mg once a day) was started. Despite the commencement of IV antibiotic and antifungal treatment, the patient’s condition deteriorated, and the VA in this right eye was classified as no perception of light (NPL) and almost total ophthalmoplegia four days post-presentation (Figure ). Treatment with IV methylprednisolone was not started in view of the infective nature of the disease and the possibility of a fungal infection, which could worsen his condition.\nMethicillin-resistant Staphylococcus aureus was isolated from the pus and from a nasal swab. IV vancomycin (1 g twice a day) and IV metronidazole (400 mg twice a day) were added. An urgent non-contrast CT brain scan was repeated and revealed features suggestive of fungal pansinusitis, with the involvement of the right orbital apex and right orbital contents (Figure ). No suspicious intracranial lesion was seen. The patient underwent emergency nasal debridement for the second time and septotomy. A bony dehiscence was noted over the right posterior ethmoid supralaterally, exposing the orbital apex.\nOn day 13 after the first surgical drainage, tissue culture from the septal wall yielded S. apiospermum. IV amphotericin B (35 mg once a day) was commenced. However, the patient’s renal profile deteriorated following the amphotericin B treatment. Thus, the antifungal regime was changed to oral voriconazole (200 mg twice a day), but the patient failed to respond to the treatment. Forty days after the initial presentation, he developed invasive pansinusitis, with disease progression involving the base of the skull (both orbits) and intracranial extension.\nAt this stage, the VA in the patient’s right eye was classified as NPL in all four quadrants, with almost total ophthalmoplegia. The VA in his left eye was 6/12, with full extraocular movement. The neurosurgical team decided against any surgical intervention. Functional endoscopic sinus surgery was performed for the third time, with nasal toileting. Intra-operatively, minimal crusting was observed over the right nasal cavity. The right sphenoid, pterygoid, maxillary sinuses and the frontal sinuses were clear, with no pus. The mucosa was healthy, with minimal bleeding. The left maxillary sinus was also clear.\nIV amphotericin B was restarted, but the patient developed gastrointestinal intolerance. The antifungal treatment was again changed to oral voriconazole (200 mg twice a day). Fifty-seven days after the initial presentation, the VA in the patient’s left eye decreased suddenly to 6/30 (reduced Snellen chart), and his colour vision decreased. A plain and contrast-enhanced CT scan of the brain, orbit and the paranasal sinuses showed invasive pansinusitis, with bilateral orbital apex involvement (Figure ). It also showed an increasing intracranial extension involving the right cavernous sinus anteriorly, the roof of the sphenoid sinus, the floor of the anterior cranial fossa and right medial cranial fossa anteriorly, as well as osteomyelitis changes at the base of the skull. In view of the acute progression of the infection, surgical intervention was considered. However, the area of the lesion was too extensive and was beyond ophthalmology coverage. Following a reassessment by the neurosurgical team, a decision was made to continue medical treatment rather than perform a surgical intervention.\nSIxty-seven days after the initial presentation, the patient’s VA decreased to NPL in both eyes. Although his Glasgow coma scale was full, his general condition worsened, with lethargy, irritability and dizziness, accompanied by nausea and vomiting. The patient decided to discharge himself from the hospital (at own risk discharge) and subsequently failed to attend an appointment in the ophthalmology clinic.