In the Australian Aboriginal population, type 2 diabetes occurs at a much higher prevalence, with a much younger age of onset of the disease and its complications. Despite the clear association with malignant otitis externa, no previous studies have examined malignant otitis externa in this population. This study explores the pattern of malignant otitis externa amongst Australian Aboriginal patients in the Northern Territory. Retrospective case series. Otolaryngology unit in a tertiary referral hospital in Northern Territory, Australia. Patients admitted with malignant otitis externa between January 2007 and October 2016 were identified by reviewing case notes. Patients diagnosed with malignant otitis externa based on results from clinical, microbiological and radiological criteria were included. Complications rates, duration of hospital stay and parenteral antibiotics, age of onset and causative organisms. Nine patients were included. Six were Australian Aboriginal - all from regional centres. The most common causative organism was Pseudomonas aeruginosa. There was a higher-than-expected occurrence of fungal malignant otitis externa (33% of Australian Aboriginal patients), who tended to be younger at diagnosis, had longer hospital stays and had a higher disease-specific mortality. Over half of the patients did not receive follow-up gallium bone scans to monitor disease resolution, reflecting the limitations of rural health care. Malignant otitis externa in the Australian Aboriginal population is a challenging disease with high complication and mortality rates. Their rural and remote distribution is a significant barrier to specialist investigation and care. Providing effective care for this disease requires improved access to high-quality primary health care and tertiary specialist services.