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Background: Tuberculous otitis media is very rare in the developed world and usually appears as a consequence of lung tuberculosis. Currently, Mycobacterium tuberculosis is the etiological agent in less than 0.1% of the cases of otitis media chronica. Case presentation: We report a rare and fatal case of a child with tuberculous otitis media and petrous apicitis, extended to brain abscess and meningoencephalitis. The patient presented with meningeal signs, facial nerve palsy, bloody-purulent ear discharge, hearing loss, and abducens nerve palsy – a clinical picture of Gradenigo's syndrome. The cerebrospinal fluid cultured positive for M.tuberculosis. QuantiFERON-TB GOLD assay proved positive. Conclusion: In the antibiotic era, tuberculosis of the middle ear with CNS involvement is a diagnostic challenge and clinicians should be aware of similar association not only in the developing countries, but especially in the industrialized world, since the disease is very rare and may result in delayed treatment with fatal consequences.