Background: This report describes the case management of a 32 year old special needs patient with life-threatening odontogenic infection. The combination of schizophrenia, Down and Eisenmenger syndromes presented significant challenges to managing his oral health, particularly within the rural context. In this case, dental treatment was limited to a full dental clearance during a high risk general anaesthesia session. Method: A comprehensive work-up prior to general anaesthesia was an essential aspect of care. This included a full medical history and examination, communication between medical specialists, the dentist and family consultation. The anaesthetic procedure was undertaken using a careful regimen of drugs and monitoring to minimize the impact on his cardiovascular system. Techniques to minimize bleeding from extraction sites were also important. Results: Three weeks postsurgically the patient was reviewed and his family reported that he was interacting positively with them after years of surliness and conflict. This was attributed to a managed psychotropic medication regimen and improved dental condition, which has led to a sustained improvement in quality of life. Conclusions: The management of acute odontogenic infection for special needs patients in the rural setting requires a local interdisciplinary team approach, careful consideration of related pathophysiology and its potential impact on general anaesthesia, and close consultation with family and carers.