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Congenital tuberculosis presenting as otorrhoea in a preterm infant. A premature infant of 25 weeks' gestational age presented at 8 weeks after birth with otorrhoea from the left ear. Following a course of topical and systemic antibiotics, the patient deteriorated developing facial nerve paralysis and cervical lymphadenitis. Contrast-enhanced CT and MRI of the head showed a destructive process of the left temporal bone. These findings prompted the clinicians to send swabs from the purulent discharge from the ear for acid-fast bacilli stain. Furthermore, surgical exploration and debridement were undertaken. Cultures from ear discharge and biopsy-taken during surgical procedure-revealed the presence of Mycobacterium tuberculosis complex. The patient developed necrotizing otitis media, left temporal bone osteomyelitis and cervical lymphadenitis. The infant's mother was found to have an endometrial biopsy positive for M. tuberculosis suggesting the diagnosis of congenital tuberculosis.