Coronavirus disease (COVID-19) complicated by rhino-orbital-cerebral mucormycosis presenting with neurovascular thrombosis: a case report and review of literature.

Among the ever-increasing literature of the coronavirus disease 2019 (COVID-19), there have been reports on several complications in association with severe acute respiratory syndrome-coronavirus-2 (SARS-CoV-2), such as secondary bacterial and fungal infections. We report a 61-year-old woman with a past history of diabetes mellitus who presented to our hospital suffering from COVID-19 infection. During the course of her hospitalization, the patient developed chemosis and proptosis in both eyes, ultimately leading to a diagnosis of invasive rhino-orbital-cerebral mucormycosis and cerebrovascular thrombosis. This study strengthens the possible association between the occurrence of COVID-19 and invasive mucormycosis infection, providing new impetus for further investigations to substantiate this correlation.

CT and MRI Findings of Invasive Mucormycosis in the Setting of COVID-19: Experience From a Single Center in India.

An increasing incidence of rhinoorbitocerebral mucormycosis (ROCM) among patients with COVID-19 has recently been reported in India. We report the imaging findings for 25 patients with COVID-19 and invasive ROCM at a single hospital in India. Findings included sinus wall erosions (n = 20), air within bony sinus structures (n = 11), and focal mucosal nonenhancement (n = 8). Orbital, vascular, and intracranial complications were also observed. Radiologists should recognize the increasing incidence of ROCM among patients with COVID-19 to facilitate early diagnosis.

Imaging findings using a combined MRI/CT protocol to identify the "entire iceberg" in post-COVID-19 mucormycosis presenting clinically as only "the tip".

AIM: To analyse combined computed tomography (CT) and magnetic resonance imaging (MRI) characteristics of invasive rhino-orbital mucormycosis (IROM) in post-COVID-19 infection patients for accurate diagnosis and delineation of the extent of involvement. MATERIALS AND METHODS: A retrospective analysis was undertaken of 50 patients who developed IROM post-COVID-19 infection who underwent combined CT/MRI evaluation. RESULTS: The age range of the 50 affected patients was 23-73 years. Out of these, 41 were diabetic. CT/MRI showed predominant involvement of the maxillary (n=26) and ethmoid (n=19) sinuses. Extension of disease to the orbit (n=35), cavernous sinus (n=18), hard palate (n=15), skull base (n=8), and intracranial involvement (n=3) was seen. Perineural spread of the disease was analysed along all divisions of the trigeminal nerve and its branches. MRI showed T2-hypointense soft-tissue thickening with heterogeneous contrast enhancement with corresponding hyperdensities on CT diagnosing the presence of fungal elements. CONCLUSION: Clinicians should be aware of the possibility of IROM post-COVID-19 infection. Conjunctive use of CT, which depicts bone destruction and other reactive bony changes along with MRI, which reveals characteristic findings of soft-tissue thickening of the involved sinuses with extension of disease to the orbits, cavernous sinus, dura, hard palate, skull base, and intracranial structures. Accurate diagnosis and early recognition of the disease and its extension with appropriate use of these techniques helps to initiate appropriate and timely treatment, which is vital to prevent a fatal outcome.

Fungus ball of the maxillary sinus: Retrospective study of 48 patients and review of the literature.

BACKGROUND: Maxillary fungus ball (FB) is the most frequent paranasal localization. OBJECTIVE: To review clinical presentation, surgery and results of treatment in our series of patients with maxillary FB. To review the literature concerning treatment of maxillary FB. PATIENTS AND METHODS: 48 patients with a diagnosis of maxillary FB were treated with endoscopic sinus surgery (ESS) alone or in association with external approaches. Before surgery all patients received computed tomography (CT), nasal endoscopy and dental examination. All the patients were followed for 1 year after surgery. Studies concerning surgical treatment of maxillary FB from 2006 were reviewed. RESULTS: The mean age of patients was 53.6 ±â€¯11.9 years. 20 patients (41.6%) did not present any symptom, 19 patients (39.7%) had nasal symptoms, 3 patients (6.2%) had facial pain, 6 patients (12.5%) had a combination of both. Endoscopic examination was positive in 31 patients (64.6%), 17 patients (35.4%) showed negative findings. Logit regression model demonstrated that clinical symptoms contribute to the prediction of a positive endoscopic examination. 25 patients (52.1%) presented odontogenic factors. Complete clinical and radiological resolution of FB was observed in 46 patients (95.8%). CONCLUSIONS: Comparing our sample to the studies reviewed we may concluded that odontogenic factors were frequently reported and should be treated at the same time of maxillary FB. ESS alone or in association with external approaches is an effective treatment for patients with maxillary FB.

Sino-Orbital Fungal Infection by Tilletiopsis minor, a Rare Human Pathogen, Diagnosed by Internal Transcribed Spacer Sequencing.

Orbital infection can be caused by numerous pathogens, and accurate diagnosis informs appropriate therapy. The authors report a case of a 78-year-old man with well-controlled diabetes mellitus and recurrent sino-orbital infection following multiple surgical procedures with negative microbiologic results. This case presented a diagnostic and treatment challenge and was aided by the use of internal transcribed spacer sequencing for pathogen identification. The fungal pathogen, Tilletiopsis minor, has not previously been described as a human pathogen in the sinus and orbit. This report describes a novel orbital pathogen and highlights the importance of diagnostic diligence and utilizing internal transcribed spacer sequencing in the workup of atypical orbital infection.

The significance of Computed Tomography in invasive paranasal mucormycosis.

INTRODUCTION: Early diagnosis of acute invasive fungal rhinosinusitis (AIFR) is crucial for patients prognosis and may reduce the extent of surgical debridement. Initial evaluation usually includes paranasal Computed Tomography (CT), with an emphasis on bony erosion which is considered a specific but insensitive radiologic sign. Most studies made no distinction between Aspergillus and Mucor species while addressing CT findings. In this study, we seek to evaluate whether bony erosion on paranasal CT is a significant and reliable finding in the initial evaluation of invasive paranasal mucormycosis. METHODS: A retrospective review of pre-operative non-contrast craniofacial CT scans of patients diagnosed with acute invasive fungal rhinosinusitis (AIFR) caused by Mucor species for the presence of bony erosion. RESULTS: A total of 13 patients (9 males, 4 females) were included. Twelve patients were immunosuppressed due to various hematological malignancies. Six patients underwent debridement due to gross intraoperative findings of bony fungal invasion, but only one patient had evidence of bony erosion on the pre operative paranasal CT. CONCLUSION: Bony erosion on paranasal CT is an exceptionally insensitive radiologic sign for establishing or rejecting the diagnosis of Mucor induced AIFR. The mainstay of confirming or rejecting the diagnosis of AIFR is by physical examination, endoscopy and oriented biopsy of suspicious mucosal lesions.

The accessibility of topical treatment in the paranasal sinuses on operated cystic fibrosis patients assessed by scintigraphy.

BACKGROUND: Nasal irrigations with antibiotics are used to eradicate Pseudomonas aeruginosa from the upper airways in patients with cystic fibrosis (CF) and thereby avoid lung colonisations; nevertheless, the efficacy is uncertain. METHODOLOGY: The aim of this study was to investigate the accessibility and durability of solutions in the sinuses before and after sinus surgery. The participants irrigated their noses with radioactively marked saline and were evaluated using a dynamic SPECT/CT scan. The preoperative and postoperative (after 30 days) examinations were compared. RESULTS: Twelve CF patients were included. In 10 out of the 24 scanned maxillary sinuses an improvement was seen postoperatively compared with the preoperative fluid volume. Notably, in 7 out of the 24 sinuses the mucosa was so swollen postoperatively that no fluid was detected. Ten patients had developed their frontal sinuses. We observed no fluid in the frontal or sphenoid sinuses, neither before nor after surgery. At best, a mean of 23% of the maxillary sinuses were filled with fluid; thus, all sinuses had postoperatively areas of the mucosa that did not have contact with the fluid. A mean of 76% of the initial volume was present after 30 min in the maxillary sinuses. CONCLUSION: Fluid-depositing using nasal irrigation will not sufficiently or not at all get in contact with all the sinus mucosa despite of sinus surgery. Thus, the efficacy of topical deposition of antibiotics is presumably reduced.

Septic thrombosis of cavernous sinus extended to the ipsilateral internal jugular vein and transversal sinus with favorable outcome: Clinical and radiological features of a Lemierre syndrome.

An 86-year-old patient was hospitalized for fever and left acute orbital syndrome (red eye with moderate visual impairment, chemosis, ophthalmoplegia without proptosis, or any fundus involvement). CT scan showed superior ophthalmic vein and cavernous sinus enlargement complicating ipsilateral sphenoidal sinusitis. Magnetic resonance imaging (MRI) demonstrated the left cavernous sinus thrombosis extended to the ipsilateral jugular vein and transversal sinus (Lemierre syndrome). Intravenous broad-spectrum antibiotics and curative anticoagulation were prescribed. Blood cultures allowed the identification of Streptococcus intermedius and Staphylococcus warneri species. Evolution was favorable and the patient was discharged 3 weeks after. Antibiotics and anticoagulation were carried out for a total duration of 4 and 12 weeks, respectively. Lemierre syndrome is a potentially life-threatening emergency rarely encountered; thus, ophthalmologists should be cognizant of clinical and radiological features. Broad-spectrum antibiotics are the mainstay of treatment. Curative anticoagulation may be added if no blood coagulation disorder nor bleeding on neuroimaging has been identified.

Non-Exenteration Management of Sino-Orbital Fungal Disease.

PURPOSE: To describe the non-exenteration management of sino-orbital fungal infection, a life-threatening condition for which orbital exenteration is generally considered a first-line treatment. METHODS: A retrospective case series is presented of 7 orbits in 6 consecutive patients admitted and treated at 2 major metropolitan tertiary teaching hospitals in Sydney, New South Wales, Australia. RESULTS: Seven orbits in 6 consecutive patients with sino-orbital fungal infection were treated conservatively with surgical debridement and intravenous antifungal agents. Four patients were immunosuppressed and the other 2 patients were otherwise healthy. All presented with pain, proptosis, or loss of vision. Causative organisms found were Mucormycoses, Aspergillus, and Scedosporium prolificans. Exenteration was avoided in all patients as part of their planned management and 5 patients, including 1 with bilateral disease, survived their disease without exenteration. Medical treatment included intravenous liposomal amphotericin B or voriconazole. A single immunosuppressed patient deteriorated and as a last resort, exenteration was performed, but this made no difference to his clinical course and in retrospect could have been avoided as he died of multiple cerebral metastases diagnosed shortly after his deterioration. CONCLUSION: The authors recommend that patients with sino-orbital fungal disease preferably be treated conservatively, without orbital exenteration.

Aspergillus Mycetoma Causing Epiphora and Ipsilateral Facial Pain.

Tearing is a frequently encountered chief complaint in an ophthalmologist's office. Certain associated atypical symptoms may warrant further workup. The authors present a case of a patient presenting with painful tearing which elicited further evaluation with CT imaging. This revealed a maxillary sinus fungus ball as the cause for the patient's tearing.

A retrospective analysis of eleven cases of invasive rhino-orbito-cerebral mucormycosis presented with orbital apex syndrome initially.

BACKGROUND: Rhino-orbito-cerebral mucormycosis(ROCM) is an invasive fungal infection that usually occurs in immunocompromised patients and sometimes presents as orbital apex syndrome(OAS) initially. It is rapidly fatal without an early diagnosis and treatment. We report the cases of invasive ROCM presenting with OAS initially in order to raise the attention of clinicians. METHODS: We retrospectively investigated eleven cases of invasive ROCM presenting initially with OAS admitted between January 2006 and December 2013. We analyzed clinical features, results of laboratory and radiological examinations, nasal endoscopy, aggressive surgical excision and debridement, and medical management outcomes of each case. RESULTS: A total of eleven cases of invasive ROCM with OAS as an initial sign were presented. Mucormycosis was accompanied by type II diabetes mellitus in nine cases, renal transplant in one case, and injury caused by traffic accident in one case. Anterior rhinoscopy revealed palatine or nasal necrotic lesions in all patients, and transethmoidal optic nerve decompression was carried out in three patients at the same time. CT scan revealed rhino-orbital-cerebral involvement in every patient. All patients were given intravenous amphotericin B. Nine patients underwent surgical debridement of necrotic tissue. Three patients survived. CONCLUSIONS: ROCM is a severe, emergent and fatal infection requiring multidisciplinary management. It may often present with OAS initially. For ophthalmologist, mucormycosis must be considered in immunocompromised patients presenting with OAS initially, and anterior rhinoscopy is imperative before hormonotherapy, even in the cases absent of ketoacidosis induced by diabetes mellitus.

Pseudomonas Aeruginosa: A Masquerader in Sino-Orbital Infections.

PURPOSE: To report 2 immunocompromised patients with sino-orbital necrotizing pseudomonas infections and review the literature. METHODS: This is a noncomparative, retrospective case series, and review. The clinical data of 2 patients with histopathologic and microbiologic diagnoses of pseudomonas sinus infections causing orbital cellulitis were obtained from medical records. A retrospective literature review was performed on all reported cases of periorbital pseudomonas infections. RESULTS: One patient with acquired immune deficiency syndrome was noted to have orbital cellulitis with clear visualization of eschar in the middle turbinate on nasal endoscopy. A second patient also had orbital cellulitis with ophthalmoplegia and presence of eschar in the sinus. Both patients had some degree of erosion through the lamina papyracea found on orbital imaging and both had intact vision without optic neuropathy. Pseudomonas infection was confirmed in both cases with permanent histopathology and cultures from conservative sinus debridement. CONCLUSIONS: Pseudomonas sino-orbital infections must be considered in the differential diagnosis in cases of eschar and orbital wall erosion especially when vision is preserved in immunocompromised individuals. This finding obviates the need for radical debridement including orbital exenteration, which can be indicated in cases of invasive fungal disease.

Chronic invasive sinus and intracerebral aspergillosis controlled by combination therapy with micafungin and a daily dose of 400 mg itraconazole oral solution.

Chronic invasive aspergillosis of the sinus is frequently fatal in the absence of early surgical and chemotherapeutic intervention because of its invasion of vascular tissue. We attempted to control a case of inoperable invasive aspergillosis of the sinus with micafungin and itraconazole oral solution. We prescribed a daily oral dose of 400 mg of itraconazole, which is twice the usual dose, and monitored the serum concentration of the drug. Finally, we were able to control the spread of the lesion. This case indicates that combination therapy with micafungin and a daily dose of 400 mg itraconazole oral solution is an alternative treatment strategy for inoperable invasive aspergillosis of the sinus.

The Role of Adjunctive Therapies in the Management of Invasive Sino-Orbital Infection.

OBJECTIVE: Invasive sino-orbital fungal infections are life-threatening complications of immunonosupression that are difficult to treat. Currently there are no standard treatment guidelines. The most widely accepted therapy includes parenteral anti-fungal therapy and surgical debridement of sinuses with orbital exenteration, a procedure that is not only disfiguring, but may increase morbidity. Injection of retrobulbar Liposomal Amphotericin B (L-AMB) is an alternative approach that provides local administration to infected tissues. The adjunct use of anti-fungal retrobulbar injections not been extensively reviewed in treating sino-orbital infection. We are reporting the multimodal approach of using L-AMB retrobulbar injections in combination with sinus debridement, intravenous (IV) anti-fungal therapy, and hyperbaric oxygen (HBO) for the management of sino-orbital infection. METHOD & RESULTS: Review of literature of 12 cases and retrospective evaluation of one patient with sino-orbital Aspergillus flavus infection on chemotherapy for T-cell acute lymphocytic leukemia treated with retrobulbar Amphotericin B, IV anti-fungal agents, and hyperbaric oxygen therapy. Clinical characteristics, radiographic features, management techniques, and clinical outcomes are described. CONCLUSION: Retrobulbar Amphotericin B injection may be an effective adjunct to hyperbaric oxygen and parenteral anti-fungals in the control of sino-orbital fungal infections.

Granulomatous invasive aspergillosis of paranasal sinuses masquerading as actinomycosis and review of published literature.

Cutaneous aspergillosis is a common systemic mycosis affecting immunosuppressed patients. Here, we describe a novel morphological type of cutaneous aspergillosis in a young immunocompetent woman who presented with a chronic history of multiple nodules and discharging sinuses over left side of the face, mimicking cervicofacial actinomycosis. Skin biopsy showed granulomatous inflammation, and of septate fungal hyphae with acute-angled branching, morphologically resembling Aspergillus. This was confirmed on fungal culture as Aspergillus flavus.

Fungal ball within Onodi cell mucocele causing visual loss.

The Onodi cell is a pneumatized posterior ethmoid cell located laterally and superiorly to the sphenoid sinus and closely related to the optic nerve. A mucocele is a benign, expansile, cystlike lesion of the paranasal sinuses that is filled with mucoid secretion. Therefore, optic neuropathy caused by an infected mucocele in an Onodi cell is uncommon. Furthermore, fungal infection superimposed on an Onodi cell mucocele is extremely rare and has not been reported previously. Here, we describe the first case of fungal ball within Onodi cell mucocele causing visual loss, which was completely removed via transnasal endoscopic approach.

Rhinocerebral Mucor circinelloides infection in immunocompromised patient following yogurt ingestion.

The purpose of this case report is to illustrate the cause of this patient's headache and sinus pain in the setting of a unique environmental exposure: the patient ingested yogurt only days before presentation. This particular brand of yogurt caused controversy in early September 2013 when the manufacturer voluntarily recalled all flavors. The yogurt was found to be contaminated with Mucor circinelloides. The recall was triggered by the FDA, after receiving many complaints from consumers affected by temporary gastrointestinal symptoms such as abdominal cramping, diarrhea, and nausea. This patient was diagnosed with Rhinocerebral mucormycosis through fungal culture of the affected area. He was specifically colonized with Mucor circinelloides, a variant that rarely causes disease in humans. According to a literature review, only eight cases of mucormycosis in adults caused by this strain were documented before 2009.

Spectrum of orbital fat necrosis in rhino-orbital-cerebral mucormycosis in post-COVID-19 patients.

PURPOSE: Rhino-orbital-cerebral mucormycosis (ROCM) is the most common presentation of mucormycosis. Sinonasal-orbital debridement with exenteration is a life-saving procedure in ROCM patients in view of severe involvement of sinuses and the eyeball. Following the second wave of coronavirus disease 2019 (COVID-19), there was a massive surge in mucormycosis cases in India in post-COVID-19 patients. Of over 300 cases of mucormycosis admitted in our hospital, many underwent exenteration and these specimens were evaluated histopathologically, where fat necrosis was found to be a prominent finding. The spectrum of fat necrosis in ROCM in orbital fat has not been described in literature. Hence, we sought to evaluate the significance and spectrum of orbital fat necrosis in ROCM. METHODS: This 3-month retrospective study included 15 cases of ROCM which underwent exenteration. Clinical data, radiologic details, and histopathologic findings were tabulated. Sections were also subjected to Periodic acid Schiff (PAS) and Gomori's methenamine silver (GMS) stains for confirming the fungus. RESULTS: All 15 cases showed fat stranding on computed tomography (CT) scan. On histopathologic examination, various tissue reaction patterns observed included acute/chronic inflammatory infiltrate, suppurative granulomas with giant cells, coagulative and fat necrosis, broad aseptate fungal hyphae with or without angioinvasion, and neural invasion. Fungal hyphae were confirmed with PAS and GMS stains. The spectrum of fat necrosis observed in all the cases included 1) acute necrotizing fat necrosis, 2) ghost adipocytes with or without saponification, and 3) crystalline/gouty fat necrosis. CONCLUSION: Fat necrosis is a significant finding in ROCM, both on CT scan and histopathology. All three patterns of fat necrosis may be observed simultaneously in a case of ROCM.