A recent fatal case of confirmed nosocomial tuberculosis (TB) transmission to a neonate in a kangaroo mother care (KMC) unit highlighted the infection risk to hospitalised neonates in South Africa, a high-burden TB setting. The index case was a 9-week-old infant who presented to another hospital's intensive care unit with severe respiratory distress shortly after discharge from the KMC unit. Contact tracing identified that the infant had been exposed to a postpartum woman with undiagnosed pulmonary TB while in the KMC unit. Molecular testing confirmed nosocomial transmission between the index case and the presumed source case in the KMC unit. We describe the subsequent process of tracing other TB-exposed infants and mothers, the difficulty in confirming TB infection/disease in pregnancy, and the provision of isoniazid preventive therapy in this cohort. We discuss the practical implementation of TB screening approaches in maternity and neonatal wards in high-burden TB settings.