Introduction: Congenital syphilis (CS) remains a condition of serious clinical and public health importance, particularly in the Aboriginal populations of northern Australia, which have seen a recent resurgence in cases. In 2005, the Northern Territory (NT) Centre for Disease Control (CDC) published guidelines for management of infants at risk of CS. We audited the management and outcomes of infants at risk of CS who were born between 2005 and 2013 in the Darwin and Katherine regions of the NT., Methods: Data, including serology, clinical examination, treatment, follow-up and infant outcomes at 12 months, were extracted from the Syphilis Register, medical and pathology records to assess clinician compliance with the CDC guidelines., Results: Thirty-three infants were identified as being at risk of CS, 26 low risk and 7 high risk. Hospital management at birth conformed well with the guidelines, with 85% of low risk, and 100% of high risk infants receiving treatment and 92% of low risk and 86% of high risk having appropriate serology. Follow-up was poorly compliant, with only 48% of infants completing serological follow-up and less than 15% undergoing clinical examination. No definitive case of CS was identified among the at-risk children., Conclusions: Overall, peri-natal management of infants was performed well, but follow-up was poor. Effective systems to transfer care from hospitals to primary care are required to improve this. The fact that no infant had direct evidence of syphilis infection suggests consideration should be given to modifying the Australian surveillance case definition., (This work is copyright. You may download, display, print and reproduce the whole or part of this work in unaltered form for your own personal use or, if you are part of an organisation, for internal use within your organisation, but only if you or your organisation do not use the reproduction for any commercial purpose and retain this copyright notice and all disclaimer notices as part of that reproduction. Apart from rights to use as permitted by the Copyright Act 1968 or allowed by this copyright notice, all other rights are reserved and you are not allowed to reproduce the whole or any part of this work in any way (electronic or otherwise) without first being given the specific written permission from the Commonwealth to do so. Requests and inquiries concerning reproduction and rights are to be sent to the Online, Services and External Relations Branch, Department of Health, GPO Box 9848, Canberra ACT 2601, or by email to copyright@health.gov.au.)