COVID-19 associated rhino-orbital-cerebral mucormycosis: An observational study from Eastern India, with special emphasis on neurological spectrum.

AIMS: 1: Describe the epidemiology and determine risk factors for COVID-19 associated mucormycosis. 2: Elaborate the clinical spectrum of Rhino-Orbital-Cerebral Mucormycosis (ROCM), pattern of neuroaxis involvement and it's radiological correlates. METHODS: Observational study. Consecutive, confirmed cases of mucormycosis (N = 55) were included. A case of mucormycosis was defined as one who had clinical and radiological features consistent with mucormycosis along with demonstration of the fungus in tissue via KOH mount/culture/histopathological examination (HPE). Data pertaining to epidemiology, risk factors, clinico-radiological features were analysed using percentage of total cases. RESULTS: Middle aged, diabetic males with recent COVID-19 infection were most affected. New onset upper jaw toothache was a striking observation in several cases. Among neurological manifestations headache, proptosis, vision loss, extraocular movement restriction; cavernous sinus, meningeal and parenchymal involvement were common. Stroke in ROCM followed a definitive pattern with watershed infarction. CONCLUSIONS: New onset upper jaw toothache and loosening of teeth should prompt an immediate search for mucormycosis in backdrop of diabetic patients with recent COVID-19 disease, aiding earlier diagnosis and treatment initiation. Neuroaxis involvement was characterized by a multitude of features pertaining to involvement of optic nerve, extraocular muscles, meninges, brain parenchyma and internal carotid artery.

Imaging of COVID-19-associated craniofacial mucormycosis: a black and white review of the "black fungus".

A subset of diabetic COVID-19 patients treated with steroids, oxygen, and/or prolonged intensive care admission develop rhino-orbito-cerebral mucormycosis. Radiologists must have a high index of suspicion for early diagnosis, which prompts immediate institution of antifungal therapy that limits morbidity and mortality. Assessment of disease extent by imaging is crucial for planning surgical debridement. Complete debridement of necrotic tissue improves survival. Imaging features reflect the angioinvasive behaviour of fungal hyphae from the Mucoraceae family, which cause necrotising vasculitis and thrombosis resulting in extensive tissue infarction. Contrast-enhanced magnetic resonance imaging (MRI) is the imaging technique of choice. The classic "black turbinate" on contrast-enhanced imaging represents localised invasive fungal rhinosinusitis (IFRS). A striking radiological feature of disseminated craniofacial disease is non-enhancing devitalised and necrotic soft tissue at the orbits and central skull base. Sinonasal and extrasinonasal non-enhancing lesions in IFRS are secondary to coagulative necrosis induced by fungal elements. Multicompartmental and extrasinonasal tissue infarction is possible without overt bone involvement and caused by the propensity of fungal elements to disseminate from the nasal cavity via perineural and perivascular routes. Fungal vasculitis can result in internal carotid artery occlusion and cerebral infarction. Remnant non-enhancing lesions after surgical debridement portend a poor prognosis. Assessment for the non-enhancing MRI lesion is crucial, as it is a sole independent prognostic factor for IFRS-specific mortality. In this review, we describe common and uncommon imaging presentations of biopsy-proven rhino-orbito-cerebral mucormycosis in a cohort of nearly 40 COVID-19 patients.

Predictors of surgical intervention and visual outcome in bacterial orbital cellulitis.

ABSTRACT: This study aims to identify predictive factors associated with surgical intervention and the visual outcome of orbital cellulitis and to evaluate the treatment outcomes.A retrospective study involving 66 patients (68 eyes; 64 unilateral and 2 bilateral) diagnosed with bacterial orbital cellulitis was conducted between November 2005 and May 2019.The mean (± standard deviation) age was 42.1 (± 25.8) years (range: 15 days-86 years). Sinusitis was the most frequent predisposing factor, occurring in 25 patients (37.9%), followed by skin infection in 10 patients (15.2%), and acute dacryocystitis in 9 patients (13.6%). Subperiosteal abscesses were found in 24 eyes and orbital abscesses in 19 eyes. Surgical drainage was performed in 31 eyes. Regarding the abscess volume for surgical drainage, a cut-off of 1514 mm3 showed 71% sensitivity and 80% specificity. There was significant improvement in visual acuity (VA) and decrease in proptosis after treatment (for both, P ≤ .001). Only pre-treatment VA ≤20/200 was a significant predictor for post-treatment VA of 20/50 or worse (adjusted odds ratio: 12.0, P = .003). The presence of a relative afferent pupillary defect was the main predictor of post-treatment VA of 20/200 or worse (adjusted odds ratio: 19.0, P = .003).The most common predisposing factor for orbital cellulitis in this study was sinusitis. VA and proptosis significantly improved after treatment. We found that the abscess volume was strongly predictive of surgical intervention. Pre-treatment poor VA and the presence of relative afferent pupillary defect can predict the worst visual outcome. Hence, early detection of optic nerve dysfunction and prompt treatment could improve the visual prognosis.

Cerebrospinal fluid leak from lateral orbit during exenteration for mucormycosis.

We describe an elderly diabetic patient presenting with sudden onset right-sided proptosis and vision loss secondary to rhino-orbital mucormycosis and central retinal vascular occlusion. He underwent orbital exenteration that was complicated by intraoperative cerebrospinal fluid (CSF) leak from lateral orbital wall. The leak was surgically repaired and the patient recovered well. We postulate the cause of the CSF leak to be twofold: necrotic periorbital tissue due to mucormycosis rendering the thin bones susceptible to damage and second, intraoperative manipulation and dissection at the orbital apex with monopolar cautery and instruments. We describe measures taken to successfully repair the CSF leak and the possible precautions that can be taken to avoid it.

Cavernous sinus-orbital apex aspergillus infection in a diabetic patient: A case report.

RATIONALE: Cavernous sinus-orbital apex aspergillosis is a rare but serious complication of rhinosinusitis. Pathology results are scarce, and this condition is difficult to diagnose based on clinical and radiological results. PATIENT CONCERNS: A 64-year-old woman presented with cavernous sinus-orbital apex syndrome. Axial and sagittal T1 contrast-enhanced magnetic resonance imaging (MRI) showed a right orbital apex mass abutting the right posterior ethmoid sinus, sphenoid sinus, and cavernous sinus. DIAGNOSIS: Cavernous sinus and orbital apex aspergillosis. INTERVENTIONS: Functional endoscopic sinus surgery was performed, and a biopsy of the lesion tissue was submitted for examination, which showed the presence of aspergillus. OUTCOMES: One year after surgical debridement, antifungal, and anticoagulation treatments, the patient is still asymptomatic without recurrence. LESSONS: Early surgical debridement is crucial to confirm the diagnosis of cavernous sinus-orbital apex aspergillosis and prevent mortality. There is no evidence of negative effects of antibiotic and anticoagulation treatments. Despite controversy, among physicians, most opt to treat patients via anticoagulation therapy.

Practice Patterns of Systemic Corticosteroid Use in Complicated Acute Rhinosinusitis Among Rhinologists and Pediatric Otolaryngologist.

BACKGROUND: The use of systemic corticosteroids remains controversial in the treatment of orbital complications of sinusitis. OBJECTIVE: To compare and contrast the current practice patterns of the use of systemic corticosteroid in the management of orbital complications of acute bacterial rhinosinusitis (ABRS) among fellowship-trained members of American Society of Pediatric Otolaryngology (ASPO) and American Rhinologic Society (ARS). STUDY DESIGN: Cross-sectional survey. METHODS: A web-based survey was electronically distributed to 497 members of ASPO and 845 members of ARS focusing on the practice habits and opinions regarding the use of systemic corticosteroids in the treatment of ABRS with orbital complications in children and adults. RESULTS: Survey completion rate was 19.1% among ASPO membership and 10.3% from ARS; 45% of respondents utilized steroids almost always or frequently in children and 36% of the time in adults. Most commonly reported indication for the use of steroid was vision change. Fellowship-trained pediatric otolaryngologists are 2.88 times more likely to prescribe corticosteroids for children than rhinologists ( P < .02). The perceived level of evidence in the literature was split between expert opinions and cohort studies. CONCLUSION: In orbital complications of ABRS, practice patterns of the use of steroid differ significantly between fellowship-trained ASPO and ARS members with respect to its use in children. Overall, corticosteroids are administered with significant frequency, although there is little actual or perceived evidence to support their safety or efficacy. In alignment of practicing evidence-based medicine, further studies are needed to guide the use of steroids in complicated ABRS with respect to timing, dosing, route, duration, and indication.

Complete surgical resection and short-term survival in acute invasive fungal rhinosinusitis.

BACKGROUND: Acute invasive fungal rhinosinusitis (AIFR) is a fulminant fungal infection seen in patients who are immunocompromised. Due to its rarity, there is little evidence regarding the appropriate antifungal treatment regimen, especially the degree of surgical intervention. OBJECTIVE: To assess factors that impact short-term survival in AIFR as defined by survival to hospital discharge and to develop a staging system to predict survival and complete surgical resection. METHODS: Fifty-four patients with histopathologically diagnosed AIFR who met inclusion criteria were identified between 1984 and 2014. Patient characteristics, disease extent, treatment modality, and short-term survival data were collected. Univariate analysis was performed to assess for factors associated with survival and increased likelihood of surgical resection. RESULTS: Of 52 patients with adequate documentation, 36 (69.2%) survived their hospital stay. Complete surgical resection was the only factor associated with improved survival (survival, 95.5%; p < 0.01). A surgical staging system was proposed to guide probability of complete resection and overall prognosis, with stage I disease limited to the nasal cavity, stage II involving the paranasal sinuses, stage III involving the orbit, and stage IV with skull base or intracranial extension. The χ2 analyses showed a decreased likelihood of complete surgical resection with stage III or IV disease compared with stage I (resection, 90.9%) (stage III resection, 37.5% [p = 0.01]; stage IV resection, 16.7% [p = 0.002]). There was a decreased likelihood of survival associated with increasing disease stage compared with stage I (survival, 100%) (stage II survival, 60% [p = 0.009]; stage III survival, 62.5% [p = 0.02]; stage IV survival, 54.6%, [p = 0.006]). CONCLUSION: Although further studies are needed to define specific treatment protocols, analysis of these data indicated that endoscopic sinus surgery with the goal of complete surgical resection may provide the best survival outcomes in select patients when complete surgical resection can be performed. Our staging system represents the first attempt to predict surgical success and prognosis in patients with AIFR.

Successful Treatment of Rhino-Orbital-Cerebral Mucormycosis in a Child With Leukemia.

Rhino-orbital-cerebral mucormycosis (ROCM) is a rare fulminant opportunistic fungal infection that despite relevant treatment has high mortality. We present a case of a 3-year-old girl with acute lymphoblastic leukemia and ROCM, who was treated successfully with excessive surgery, systemic antifungal treatment with amphotericin B (AmB), posaconazole, and terbinafine as well as hyperbaric oxygen. Surgery included, beside extracranial and intracranial removal of infected areas, endoscopic sinus and skull base surgery with local AmB installation and in addition placement of an Ommaya reservoir for 114 intrathecal administrations of AmB. In addition, we review the literature of ROCM in pediatric patients with hematological diseases.

Fungal infections of the orbit.

Fungal infections of the orbit can lead to grave complications. Although the primary site of inoculation of the infective organism is frequently the sinuses, the patients can initially present to the ophthalmologist with ocular signs and symptoms. Due to its varied and nonspecific clinical features, especially in the early stages, patients are frequently misdiagnosed and even treated with steroids which worsen the situation leading to dire consequences. Ophthalmologists should be familiar with the clinical spectrum of disease and the variable presentation of this infection, as early diagnosis and rapid institution of appropriate therapy are crucial elements in the management of this invasive sino-orbital infection. In this review, relevant clinical, microbiological, and imaging findings are discussed along with the current consensus on local and systemic management. We review the recent literature and provide a comprehensive analysis. In the immunocompromised, as well as in healthy patients, a high index of suspicion must be maintained as delay in diagnosis of fungal pathology may lead to disfiguring morbidity or even mortality. Obtaining adequate diagnostic material for pathological and microbiological examination is critical. Newer methods of therapy, particularly oral voriconazole and topical amphotericin B, may be beneficial in selected patients.

Apical Orbital Aspergillosis Complicating Giant Cell Arteritis.

A 75-year-old woman with new onset headaches and left vision loss, temporal scalp tenderness, and jaw claudication was found to have biopsy-proven giant cell arteritis (GCA). Despite treatment and improvement with prednisone, she later developed left orbital apex syndrome, and an orbital biopsy revealed aspergillosis. After antifungal treatment, extraocular motility improved although vision in the left eye remained no light perception. Clinicians should be aware that fungal orbital apex disease may mimic or complicate steroid-treated GCA.

A Case of Orbital Histoplasmosis.

Histoplasma capsulatum var capsulatum is a dimorphic fungus endemic to the Ohio and Mississippi River Valleys of the United States. In this case report, a 33-year-old woman who presented with a right orbital mass causing progressive vision loss, diplopia, and facial swelling is described. Lateral orbitotomy with lateral orbital wall bone flap was performed for excisional biopsy of the lesion. The 1.5 × 1.8 × 2.3 cm cicatricial mass demonstrated a granulomatous lesion with necrosis and positive staining consistent with Histoplasma capsulatum var capsulatum infection. To the authors' knowledge, this is the first case of orbital histoplasmosis to be reported in the United States and the first case worldwide of orbital histoplasmosis due to Histoplasma capsulatum var capsulatum.

Granulicatella elegans Causing Periorbital Infection During Orthodontic Treatment.

Granulicatella elegans is a normal component of the oral flora and is an unusual causative agent of infective endocarditis. A case of G. elegans periorbital infection of the eyelid after dental treatment is reported. A healthy 35-year-old man presented with painful swelling of the left upper eyelid. He was empirically treated with oral amoxicillin for 1 week. He presented 3 months later with the same clinical features. G. elegans and Staphylococcus epidermidis were identified in bacterial cultures from wound aspirates. Probable relapse of periorbital infection was successfully treated with a 6-week course of oral amoxicillin. This is the first reported case of a non-bloodstream infection caused by G. elegans. Clinicians should be aware of G. elegans as an unusual causative agent of periorbital infection. Within the limitations of this case report, prolonged antibiotic therapy is recommended for a G. elegans periorbital infection to minimize the risk of relapse.

Orbital cellulitis with periorbital abscess secondary to methicillin-resistant Staphylococcus aureus (MRSA) sepsis in an immunocompetent neonate.

This article advocates the need for early incision and drainage of periorbital abscesses. We report a case of a 1.5-month-old neonate with orbital cellulitis and periorbital abscess, which had rapidly developed over a period of 3 days. Treatment history revealed methicillin-resistant Staphylococcus aureus sepsis treated with intravenous vancomycin, and incision and drainage of abscesses at multiple sites (left parotid region, upper and lower limbs). A small swelling noted on the left temporal region on discharge from the hospital was treated with oral cotrimoxazole. However, it spread rapidly to involve the periorbital tissue and the bones of the orbital walls to form a periorbital abscess and orbital cellulitis.

Alternaria-Associated Fungus Ball of Orbit Nose and Paranasal Sinuses: Case Report of a Rare Clinical Entity.

Alternaria-associated fungus ball of maxillar, ethmoidal paranasal sinuses, nasal cavity and orbit with bone erosion is extremely rare. Till recently, only two cases of this infection in immune competitive patients have been reported. We are herein describing the case of immune-competent woman who suffered of nasal congestion for 10 years. Patient was treated for tumor-like lesion in right maxillar sinus, where propagation in right nose cavity, right ethmoidal cells and right orbita was present. The organism that was seen in surgical removal of fungal debris by histological study, in using mycological testing, was proven as Alternaria alternata. Combination of surgical intervention and treatment with itraconazole eradicated fungal infection, and the disease was not relapsed in follow-up period of 2 years.

Post-operative endophthalmitis caused by Acremonium falciforme with orbital and extra-orbital involvement following combined cataract and glaucoma surgery: a case report.

INTRODUCTION: In this report, we describe an unusual case of post-operative Acremonium falciforme endophthalmitis with orbital and extra-orbital involvement following combined cataract and glaucoma surgery. CASE PRESENTATION: A 68-year-old Caucasian man with glaucoma presented with endophthalmitis characterized by pain, redness and impaired vision in the left eye fifteen days after combined cataract and filtering surgery. He subsequently underwent a pars plana vitrectomy, with vitreous sampling, silicone oil placement and intra-vitreal injection of antibiotics, but only after a second vitrectomy we identified Acremonium falciforme as the causative agent for the endophthalmitis. An antifungal systemic and topical therapy was started, but meanwhile the infection extended to orbital and peri-orbital tissues. Following these procedures, even if the eye went slowly in phthisis, we were able to limit the further extension and circumscribe the orbital and extra-orbital involvement. CONCLUSION: To our knowledge, this report is the first describe Acremonium falciforme endophthalmitis with orbital and extra-orbital involvement, following anterior segment combined surgery. Ophthalmologists and physicians should be aware of the extension risk of a fungal panophthalmitis, but also to potentially serious side effects related to systemic therapy.

[Two cases of allergic fungal rhinosinusitis with bone destruction].

We report herein two cases of allergic fungal rhinosinusitis accompanied by bone destruction of the adjacent nasal sinuses. The first case involved a 21-year-old man who presented with left exophthalmos. Computed tomography (CT) showed soft tissue lesions in the left paranasal sinuses and destruction of the left lamina papyraceae, as well as infiltration of the lesion into the orbit. In the second case, a 39-year-old man, CT showed bone destruction of the skull base and medial wall of the left orbit. In both cases, total serum immunoglobulin (Ig)E level was >1000 IU/mL and fungus-specific IgEs were increased. Fungal hyphae were identified within the mucus on histopathological examination in both cases; however, no fungal invasion of the mucosa was apparent. Final diagnosis was allergic fungal rhinosinusitis (AFRS) in both cases. AFRS is a relatively new disease concept that was proposed in the early 1980s, with disease characteristics very similar to eosinophilic rhinosinusitis. Occasionally, AFRS must be differentiated from malignant disease or invasive fungal rhinosinusitis, so an understanding of the clinical features is important.

Subcutaneous phaeohyphomycosis of the face presenting as rhinoentomophthoramycosis.

OBJECTIVES: Subcutaneous phaeohyphomycosis is the most common variety of phaeohyphomycosis and presents as asymptomatic or mildly painful, localized cysts, abscesses, or sometimes chromoblastomycosis-like lesions over the feet, legs, or hands in about 60-85% of cases. It usually afflicts adults with some degree of immunosuppression. METHODS: We report a 30-year-old man, who presented with features of recurrent sinusitis (episodes of fever, nasal stuffiness, paranasal sinus pain, and tenderness), mucosal hypertrophy of the maxillary sinuses, and mid-face swelling that was freely mobile and suggestive of rhinoentomophthoramycosis. He had no other associated illness. RESULTS: Histology suggested tumid lupus erythematosus. The diagnosis was based on Fonsecaea pedrosoi cultured from a biopsy specimen. CONCLUSIONS: Combination therapy with itraconazole and saturated solution of potassium iodide (SSKI) was more effective than itraconazole used alone.