Malignant external otitis (MEO) is a rare inflammatory and infectious condition, typically caused by Pseudomonas aeruginosa , that mainly affects diabetic or immunocompromised elderly patients and is associated with severe morbidity and mortality. It begins in the external auditory canal and rapidly progresses through the skull base, leading to osteomyelitis and may result in cranial neuropathy, especially of the facial nerve. Here we describe a rare neurological presentation of MEO in a 65-year old diabetic man, who presented with an 8-month progressing left otitis externa and evolved with ipsilateral proptosis, ophthalmoplegia, blindness, facial palsy, hearing loss and contralateral evolvement of the temporal bone with hearing impairment. He was initially treated with oral ciprofloxacin and after one week was transferred to our tertiary hospital, where antibiotic therapy was switched to meropenem and vancomycin due to the severity of the case and to the hospital’s microbiological profile. The patient underwent left canal wall-up mastoidectomy with insertion of ear ventilation tube bilaterally, with good recovery of right ear hearing capacity, but with no improvements of neurological deficits nor left hearing function. All microbiological tests performed were negative, and this was interpreted as a possible consequence of the early use of antibiotics. Unfortunately, the patient was infected by Sars-CoV-2 during hospitalization and passed away after ten days of COVID-19 intensive care unit internment.

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