Abstract
Introduction Necrotising otitis externa (NOE) is a rare life-threatening complication of simple otitis externa which can be difficult to diagnose and manage. It is very rarely centred on the temporomandibular joint (TMJ). Fungi cause NOE in approximately 5–20% of patients, and a high index of suspicion is required for diagnosis, particularly when there is no improvement with prolonged topical and intravenous antibiotic therapy. Objective To report a novel case of fungal NOE centred on the left TMJ in an immunocompromised adult male with a focus on investigations and optimal management. Case Report. A 67-year-old male with comorbid chronic renal impairment presented to our otolaryngology department with prolonged left otalgia and otorrhoea. Subsequent cross-sectional imaging demonstrated left NOE centred on the TMJ. Poor resolution with prolonged courses of systemic and topical anti-pseudomonal antibiotics prompted maxillofacial surgical input for left TMJ exploration, washout, and biopsy from the joint capsule. The causative organism was identified as Aspergillus flavus on PCR analysis. The patient was successfully treated with oral posaconazole and repeated topical insertions of amphotericin B-soaked ribbon gauze to the left ear. Discussion. A combination of various imaging modalities including CT, MRI, Tc-99, and gallium-67 are utilised in clinical practice both to diagnose NOE and subsequently monitor disease progression or resolution. Immunocompromised patients with confirmed fungal NOE may require a combination of treatments including surgical debridement and prolonged antifungal therapy for a number of months, if not lifelong, treatment. Initiating empirical antifungal therapy may be justified in some patients. However, this should be judged on a case-by-case basis and guided by discussion with the local microbiology and infectious diseases departments. However, there is no national guideline or consensus regarding treatment of these patients, especially in cases of fungal NOE.
Highlights
Necrotising otitis externa (NOE) is a rare life-threatening complication of simple otitis externa which can be difficult to diagnose and manage
Mardinger et al have shown an involvement of the temporomandibular joint (TMJ) in only 14% of cases with a high mortality rate of 50% [1]. ere has been an exponential increase seen in cases of NOE; recent analysis of Hospital Episodes Statistics data for England showed a sixfold increase in the number of cases from 1999 (n 67) to 2013 (n 421), likely due to an ageing population in the UK [2]
Other causative agents include Aspergillus, Staphylococcus aureus, Proteus mirabilis, Klebsiella oxytoca, Burkholderia cepacia, and Candida parapsilosis [15]. It is often precipitated by iatrogenic trauma to the external auditory canal such as aural irrigation which could have been a factor in this case study, as the patient originally underwent microsuction to remove wax from both ear canals [16]. is may have caused local trauma, microangiopathy, and local hypoperfusion leading to an invasive fungal infection in an already immunocompromised host [17]
Summary
Necrotising otitis externa (NOE) is a rare life-threatening complication of otitis externa, affecting the skull base, mastoid, and temporal bones. e disease is seldom centred on the temporomandibular joint. In 1987, Cohen and Friedman attempted to list diagnostic criteria for NOE using obligatory and occasional categories outlined as follows (Table 1) [7]. These criteria overemphasise the role of diabetes mellitus and side-line other forms of immunosuppression which are important to consider. Various case studies have reported a higher number of complications associated with fungal NOE such as cranial nerve palsies, pseudoaneurysms in the internal maxillary artery, and temporomandibular joint destruction [1, 11, 12]. We report an unusual case of fungal NOE centred on the left TMJ in a 67-year-old male with comorbid chronic renal impairment. Since no universal guideline or consensus exists regarding optimal investigations and treatment of this condition, we have presented our practice and reviewed the literature to shed further light and guide clinicians managing this important condition
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