Aspergillus infections of lateral skull base: a case series.

PURPOSE: While extensive research with accurate classification has been done in mycoses of the paranasal sinuses and anterior skull base, a similar understanding of lateral skull base fungal pathologies is lacking due to relative rarity and diagnostic difficulties. We introduce a series of eleven cases and two different invasive entities of Aspergillus temporal bone diseases-fungal skull base osteomyelitis (SBO)/malignant otitis externa (MOE) and chronic invasive granulomatous fungal disease (CIGFD). METHODOLOGY: A retrospective observational study was conducted at the neuro-otology unit of a tertiary care referral center between July 2017 and November 2022. Diagnosed cases of lateral skull base osteomyelitis with atypical symptoms and lack of response to culture-directed antibiotics were evaluated for fungal origin. Patient data, including history, laboratory findings, serum galactomannan assay, CT and MRI imaging findings, clinical examination findings, and co-morbidities, were analyzed. The treatment course and response were assessed. RESULTS: A total of 11 cases were included in the study. Of these, 9 were cases of Aspergillus-induced skull base osteomyelitis (SBO) and 2 of Aspergillus-induced chronic invasive granulomatous fungal disease (CIGFD). CIGFD presented with persistent ear discharge and slowly progressive post-aural swelling, while all patients of fungal SBO had lower cranial nerve palsies. CIGFD responded to excision and antifungals, while SBO responded well to conservative anti-fungal treatment. CONCLUSION: In cases of lateral SBO not responding to antibiotic therapy, the possibility of fungal etiology should be considered. Aspergillus spp. seems to be the major fungal pathogen.

Neoplasms of the Ear Canal.

Primary EAC neoplasms include benign and malignant lesions of bony, glandular or cutaneous origin. Small, benign slow growing bony neoplasms are often asymptomatic, diagnosed incidentally and might not require intervention. Both malignant and benign neoplasms of cutaneous and glandular origin can present with symptoms of chronic otitis externa, leading to delays in diagnosis. Prompt biopsy of soft tissue lesions associated with non-resolving otitis externa are warranted. Local and regional imaging is helpful to understand disease extent and origin, but even early-stage malignant neoplasms require aggressive surgical treatment.

A Case of Adult Clival Osteomyelitis.

Clival osteomyelitis is a potentially life-threatening skull base infection. It is rare and generally challenging to diagnose and treat. Clival osteomyelitis is typically seen in the pediatric population and is very rare in the adult population. It occurs as a complication of recurring paranasal infections and malignant otitis externa. The exact pathophysiology of osteomyelitis of the clivus is relatively uncertain. Here, we describe a case of a 36-year-old man with medical history significant for hypertension and poorly controlled type 1 diabetes mellitus who experienced recurrent paranasal sinus infection for 2 years. He received multiple antibiotic treatments and underwent adenoidectomy without substantial improvement of symptoms. Ultimately, a diagnosis of the clival osteomyelitis through the help of a computed tomography (CT) scan of the paranasal sinus and neck was made. This diagnosis allowed for adequate intervention and treatment of our patient with subsequent resolution of his presenting symptoms. This case highlights the importance of high suspicion for clival osteomyelitis in patients with recurring sinus infections.

A Case Series of Malignant Otitis Externa Mimicking Malignancy.

Malignant otitis externa is an inflammation of the external auditory canal with preceding osteomyelitis of the temporal bone and the adjacent structures that could be potentially lethal. Malignant otitis externa may present with cranial nerve involvements and massive spread of disease mimicking nasopharyngeal carcinoma or any other malignancies on imaging. Two elderly patients who presented with severe otalgia and significant facial nerve palsy and lower cranial nerve palsies showing extensive spread of disease are reported in this case series. They both had resolution of disease after a prolonged course of antibiotics and cortical mastoidectomy for disease clearance in one of them.

Aspergillus necrotizing otitis externa with temporomandibulozygomatic involvement.

INTRODUCTION: Aspergillus necrotizing otitis externa (NOE) is a rare disease, often associated with delayed diagnosis, the management of which is poorly defined. SUMMARY: The authors report a case of Aspergillus flavus necrotizing otitis externa with temporomandibular arthritis and temporozygomatic osteomyelitis with Staphylococcal coinfection in a diabetic patient. The diagnosis and discontinuation of treatment were guided by PET-CT scan. A favourable course without sequelae was observed after repeated surgical curettage and 3 months of antifungal therapy. DISCUSSION: Aspergillus flavus is the agent most commonly incriminated in NOE. Indirect diagnostic tests (serology) may be negative. The diagnosis is based on imaging-guided surgical biopsy with histological examination and standard and fungal microbiological culture. Treatment requires a combination of surgery and antifungal therapy. The duration of antifungal therapy is poorly defined and discontinuation of therapy can be guided by PET-CT scan.

Thirty-Day Readmission and Prolonged Length of Stay in Malignant Otitis Externa.

OBJECTIVES: To determine independent risk factors for 30-day readmission, prolonged length of stay (PLOS), and discharge to a rehabilitation facility for those with malignant otitis externa. METHODS: Retrospective cohort study of patients hospitalized with malignant otitis externa (International Classification of Diseases, 9th edition, code 380.14) in the Nationwide Readmissions Database (2013-2014). Overall and disease-specific complication and mortality data were analyzed using chi-squared and multivariate analysis. RESULTS: There were 1267 cases of malignant otitis externa extracted. A PLOS of ≥8 days (90th percentile) was found in 14.2% (n = 180) of patients, and 13.7% (n = 174) were discharged to a facility. Patients were readmitted within 30 days at a rate of 12.5% (n = 159). The overall rates of uncomplicated and complicated diabetes were found to be 42.1% and 17.8%, respectively. Factors independently associated with PLOS included undergoing a surgical procedure (odds ratio [OR] 2.08, P < .001), and having central nervous system complications (OR 3.21, P < .001). Independent risk factors for disposition to a facility included nutritional deficiency (OR 1.91, P = .029), PLOS (OR 4.61, P < .001), and age 65-79 years (OR 6.57, P = .001). Readmission was independently linked to PLOS (OR 3.14, P < .001). Diabetes was not an independent risk factor for any outcome. CONCLUSIONS: Thirty-day readmission, PLOS, and ultimate discharge to a rehabilitation facility were common and closely intertwined. Despite the classic association between diabetes and malignant otitis externa, diabetes was not an independent risk factor for any of our outcomes. LEVEL OF EVIDENCE: 4 Laryngoscope, 130:2220-2228, 2020.

Malignant (necrotizing) externa otitis: the experience of a single hyperbaric centre.

INTRODUCTION: Malignant otitis externa (MOE) is a potentially life-threatening infection of the soft tissues of the external ear, quickly spreading to involve the periosteum and bone of the skull base. Treatment includes antibiotics and eventually surgery. Hyperbaric oxygen treatment (HBOT) has been proposed as an adjunctive therapy. However, in the tenth consensus conference, this disease was considered as a non-indication for HBOT. The aim of this study was to evaluate the effectiveness of HBOT in MOE treatment. METHODS: Retrospective and observational study was conducted of patients with MOE treated in our centre. Staging of the disease was made according to the clinicopathological classification system. RESULTS: From March 1998 to November 2016, 16 patients were referred. 6% patients were on stage 1 of the disease at the time they were referred, 20% in stage 2, 7% in stage 3a, 13% in stage 3b and 53% in stage 4. Seven (43.75%) patients had VII nerve palsy and three (18.75%) patients had multiple nerve palsy. Average length of symptoms of disease was 5 months (maximum 11 months). Average number of sessions was 33 and the length of hospitalization prior to HBOT (median 90 days) was significantly longer than the time between beginning HBOT and cure (p = 0.028, Wilcoxon signed rank test). There were no fatalities due to MOE and all patients were considered free of disease after HBOT. CONCLUSION: HBOT was well tolerated and revealed to be a helpful adjuvant treatment in MOE. According to our data, HBOT should be considered for patients who failed conventional treatments and in severe cases.

Effective Inexpensive Management of Necrotizing Otitis Externa Is Possible in Resource-Poor Settings.

INTRODUCTION: Necrotizing otitis externa resolves best with antimicrobial treatment. How to care for these patients and monitor their resolution remains a problem. Our protocol in Bangalore can manage these patients inexpensively and well. MATERIALS AND METHODS: Patients who were referred to our patients became the subjects for this paper. They were managed through our protocol, which consists of IV ciprofloxacin and meropenem, weekly labs, weekly examinations, and photodocumention. RESULTS: Fifty-one people presented with necrotizing otitis externa (NOE) between October 2015 and November 2017 and completed our entire protocol. Forty-six had complete resolution of their disease, while 5 had to undergo surgical removal of necrotic bone. Six of 8 patients with facial weakness had improvement in their House-Brackmann scores. Reduction of self-reported nocturnal pain, dissolution of ear canal granulations, and normalization of the erythrocyte sedimentation rate (ESR) proved to be the most accurate indicators of disease regression. CONCLUSION: Patients are monitored closely with review of their otalgia, examination of their canal, repeated ESRs, effective control of their diabetes, and radiological imaging. All this can be done in a resource-poor country, which in turn serves as a model for the wealthier nations.

Fungal Malignant Otitis Externa Involves a Cascade of Complications Culminating in Pseudoaneurysm of Internal Maxillary Artery: A Case Report.

BACKGROUND Pseudomonal infection is the most common cause of malignant otitis externa (MOE), which typically affects elderly diabetic patients. Fungi are a rare cause of MOE. MOE can be life-threatening if not recognized and treated promptly. It can result in a wide spectrum of complications, including skull-base osteomyelitis, cranial nerve palsy, cerebral venous thrombosis, and brain abscess. Pseudoaneurysm formation of the intracranial vessels is a life-threatening complication of MOE that is seldom reported in the literature. CASE REPORT We report the case of a 66-year-old diabetic man with MOE who was initially treated with antipseudomonal antibiotics after negative initial culture results. His MOE resulted in a cascade of complications, including facial nerve palsy, skull base osteomyelitis, and sigmoid sinus thrombosis, and culminated in left maxillary artery pseudoaneurysm formation resulting in massive epistaxis and hemodynamic instability. Endovascular embolization resulted in a successful obliteration of the pseudoaneurysm. A subsequent functional endoscopic sinus surgical (FESS) tissue biopsy confirmed Candida glabrata as the etiological agent. The patient was successfully treated with antibiotics and antifungal and anticoagulation therapy, and was discharged home in good condition. CONCLUSIONS A high index of suspicion for the diagnosis of fungal MOE, particularly in intractable cases of MOE with negative initial cultures, should be maintained. Pseudoaneurysm formation is a life-threatening complication of MOE that is seldom reported in the literature and should be suspected in any patient with MOE who presents with epistaxis or intracranial bleeding.

Necrotizing Otitis Externa-Analysis of 83 Cases: Clinical Findings and Course of Disease.

OBJECTIVES: Evaluate the relationship between clinical findings and course of disease among patients with necrotizing otitis externa (NOE). STUDY DESIGN: Retrospective case series review. SETTING: Tertiary referral center. PATIENTS: Adult patients with no previous history of chronic ear disease, hospitalized due to NOE between the years 1990 to 2015. MAIN OUTCOME MEASURES: 1) Duration of hospitalization and 2) necessity for surgery. RESULTS: Eighty-one patients were included in the study, corresponding to 83 effected ears. Thirty-two patients (38.5%) were hospitalized longer than 20 days and 20 patients (24.0%) underwent surgery. Otalgia was the most common complaint (n = 71, 85.5%). Pseudomonas Aeruginosa (PA) was the most common isolated bacteria (n = 40, 48.1%). Shifting incidence of culture results was noted, as rates of PA NOE decreased and rates of sterile culture and fungal NOE increased. Duration of complaints and presence of aural discharge at admission were associated with prolonged hospitalization (p = 0.010, p = 0.011, respectively). Advanced age, duration of hospitalization, and rates of readmission were associated with surgery (p = 0.037, p < 0.001, p < 0.001 respectively). CONCLUSIONS: Duration of complaints and presence of aural discharge may indicate advanced NOE and require longer in-hospital treatment. Elderly patients are at increased risk for conservative treatment failure and are more likely to require surgery. With shifting incidence of pathogens, a wider empirical treatment covering nontraditional pathogens should be considered.

FDG-PET/CT for diagnosis and follow-up of necrotizing (malignant) external otitis.

OBJECTIVE: Imaging is important for the diagnosis and follow-up of necrotizing external otitis (NEO). The best imaging modality is controversy. To suggest 2-deoxy-2-[fluorine-18] fluoro-D-glucose-positron emission tomography/computed tomography (18F-FDG-PET/CT) as an alternative to technetium-99m and gallium-67 scans for diagnosis and assessment of response to treatment for patients with NEO. STUDY DESIGN: Case series. METHODS: Tertiary referral center. From 2013 through 2017, 12 patients were diagnosed with NEO based on clinical features and positive FDG uptake within the temporal bone on PET/CT. Mean age was 74 ± 11.5; 83% of the patients were male; and 83% had diabetes. RESULTS: PET/CT scans were reviewed independently by two nuclear medicine specialists. Imaging located osteomyelitis in external ear canal, mastoid, temporomandibular joint, and nasopharyngeal region (100%, 50%, 16%, 8%, respectively). Mean follow-up was 16 months. Eight patients (67%) underwent a second PET/CT scan after active otitis resolved and after at least 6 weeks of antibiotic treatment. The scan demonstrated no or substantially reduced FDG uptake and treatment was stopped. The patients had no NEO symptoms at the end of follow-up. One patient had significant uptake, and antibiotic treatment was continued until a third scan demonstrated no FDG uptake. Two patients died before the second PET/CT, and two were lost to follow-up. CONCLUSION: 18F-FDG-PET/CT is a reliable imaging modality for diagnosis, disease localization, and decision making regarding treatment cessation. 18F-FDG-PET/CT should be considered as the imaging modality of choice for initial diagnosis and follow-up in NEO patients. Larger, controlled studies are warranted. LEVEL OF EVIDENCE: 4 Laryngoscope, 129:961-966, 2019.

Malignant external otitis following radiotherapy for oral cancer: A case report.

RATIONALE: Although an ototoxicity is well-known as adverse event of the radiotherapy, it is not widely known that immunosuppressed patients who underwent radiotherapy in head and neck region have risk of malignant external otitis. PATIENT CONCERNS: A 68-year-old man with diabetes, who had been diagnosed as intraosseous squamous cell carcinoma of the right mandible, underwent surgical resection. He received a total of 60 Gy/30Fr postoperative radiation. Four months after the course of radiation, he suffered from right aural fullness, otalgia and otorrhea. DIAGNOSES: Clinical examination revealed granulation and existence of Pseudomonas aeruginosa in the external auditory canal. Computed tomography showed expansive inflammation and erosion in the temporal bone. The patient is elderly and diabetes. These findings led to the diagnosis of malignant external otitis. INTERVENTIONS: The ear irrigation and administration of quinolones were started. Afterwards, fistula was formed in the oral cavity, and connected to the right external auditory canal. Therefore, irrigations were performed not only from ear but also from the oral fistula. OUTCOMES: Eight weeks after starting treatment, the malignant external otitis was completely healed. LESSONS: Physicians should raise awareness of malignant external otitis in immunosuppressed patients with oral cancer after radiotherapy.

Malignant external otitis: The shifting treatment paradigm.

IMPORTANCE: Malignant external otitis (MEO) is an aggressive infection occurring in immunocompromised hosts. Increasing antimicrobial resistance is making the disease more difficult to treat. OBJECTIVE: Determine if there has been a shift in the microbiology and outcomes of MEO. DESIGN: A retrospective case series at a tertiary care institution. SETTING: Inpatient and outpatient tertiary care hospital. PARTICIPANTS: 12 cases of recent MEO were reviewed. MAIN OUTCOMES AND MEASURES: The primary outcome was progression of disease. Secondary outcomes were drug resistance and complications of MEO. RESULTS: Only 4 patients were cured of MEO. Four patients expired during the study period and at least one of these deaths was a direct result of the MEO. 7 patients developed Cranial nerve palsies, and 3 patients developed abscesses. CONCLUSIONS: Select cases of MEO now require multi-drug and long-term parenteral antibiotic therapy with extended hospital stays.

Lyric hearing aid: a rare cause of benign necrotising otitis externa/external ear canal cholesteatoma.

An 80-year-old Caucasian man presented with an incidental and asymptomatic lesion in his right ear thought to be secondary to his use of hearing aids for presbycusis. He used Lyric hearing aids, designed for 24 hours-a-day use for 4 months at a time and had no other previous otological problems. He underwent a bony meatoplasty and vascular flap reconstruction via a retroauricular approach to remove the lesion for histological analysis and regrafting of the area. The lesion was confirmed on histopathology as an ear canal cholesteatoma.

The treatment of pseudoaneurysms with flow diverters after malignant otitis externa.

Background We report a case of bilateral malignant otitis externa complicated with bilateral petrous internal carotid artery pseudoaneurysms and their successful treatment with a flow diverter. Case report A 68-year-old woman with serious complications of type II diabetes mellitus had malignant otitis externa on the right side. She was treated with combined antibiotic therapy and underwent mastoidectomy for mastoiditis. She presented at our hospital with acute hemorrhage from the right external auditory canal. The emergency computed tomography (CT) angiography revealed a multiobulated pseudoaneurysm at the petrous segment of the right internal carotid artery. The pseudoaneurysm was treated with a 5 × 40-mm Surpass flow diverter. Three months later, she developed a malignant external otitis on the left side. As the infection progressed, a left-sided mastoiditis, a brain abscess, and a pseudoaneurysm at the petrous segment of the left internal carotid artery developed. The pseudoaneurysm caused bleeding from the left ear, and was treated with a 5 × 50-mm Surpass flow diverter. No recurrent bleeding was observed. Four months later, a follow-up angiography showed complete occlusion of the pseudoaneurysm on the left side, but a residual aneurysm could be detected on the right side. One year after the first intervention, the follow-up CT and magnetic resonance angiography revealed the complete occlusion of the aneurysms bilaterally. Conclusion The use of a flow diverter appears to be an efficient and safe method to occlude carotid pseudoaneurysms even in an inflammatory milieu.

Skull base osteomyelitis secondary to malignant otitis externa mimicking advanced nasopharyngeal cancer: MR imaging features at initial presentation.

PURPOSE: Skull base osteomyelitis (SBOM) is an inflammatory process which often arises from malignant otitis externa (MOE); the diffuse skull base and adjacent soft tissue involvement may be mistaken at initial imaging for advanced nasopharyngeal carcinoma (NPC), especially if there is no prior knowledge of MOE, direct spread from the sphenoid sinus or in atypical presentations of MOE. This study aims to evaluate imaging features on MR that may differentiate SBOM from NPC. MATERIALS AND METHODS: The MR examinations of 26 patients diagnosed with SBOM between January 1996 and January 2013 were retrospectively reviewed. Comparison was also made with the MR images of 22 consecutive patients with newly diagnosed advanced T3 and T4 NPC between July 2011 and August 2012. Imaging features in both conditions were compared, including the presence of a nasopharyngeal bulge, nasopharyngeal mucosal irregularity, lateral extension, architectural distortion (or lack thereof), increased T2 signal and enhancement patterns. RESULTS: The most prevalent findings in SBOM were lateral extension, increased T2 signal in adjacent soft tissues, lack of architectural distortion and enhancement greater than or equal to mucosa. The combination of these 4 findings was found to best differentiate SBOM from advanced NPC, and found to be statistically significant (p<0.001). CONCLUSION: We suggest that the combination of lateral extension, increased T2 signal, lack of architectural distortion and enhancement greater than or equal to mucosa is helpful in differentiating SBOM from advanced NPC.

[Malignant externa otitis: the modern principles of diagnostics and treatment]. / Zlokachestvennyi naruzhnyi otit: sovremennye printsipy diagnostiki i lecheniia.

The objective of the present study was to elucidate the specific features of the clinical course of malignant externa otitis in the context of the present-day concepts. A total of 5 patients presenting with the confirmed diagnosis of malignant external otitis were available for the examination. The analysis of the clinical observations provided the basis for the characteristic of pathogenesis of this condition, diagnostic principles, and treatment modalities for the management of the pathology in question. It is concluded that the patients suffering from malignant externa otitis must remain under medical care and observation during a long period and the strategy for the treatment of each concrete patient should be chosen on an individual basis taking into consideration the presence of concomitant pathologies and their adequate therapy.