To present our case series and management of Scedosporium apiospermum infections of the middle ear and mastoid, and review the current literature on this rare yet potentially life-threatening condition.

Medical records of patients treated at the Royal Victorian Eye and Ear Hospital for S apiospermum middle ear and mastoid infections between 2009 and 2019 were reviewed. A literature search was conducted using PubMed, Medline and Cochrane Library databases.

Two patients were identified in our institution: a 62-year-old diabetic woman with otogenic skull base osteomyelitis, and a 12-year-old boy with unilateral chronic suppurative otitis media which developed after tympanostomy tube insertion. The persistence of otalgia and otorrhoea despite prolonged antibiotic treatment characterised these cases. Both patients received voriconazole, and achieved disease resolution without complications. Ten relevant cases were identified after review of the literature. Despite treatment, there were three patient deaths, and four patients with otological or neurological complications.

The presence of a middle ear or mastoid infection refractory to appropriate topical and systemic antibiotics should prompt clinicians to consider a fungal infection. The role of surgical debridement in the treatment of S apiospermum infection of the middle ear and mastoid is equivocal.