With dengue fever emerging as a global health problem and more Australians travelling to endemic areas, imported dengue infection is on the rise and clinicians need to remain vigilant. Primary cardiac and neurologic involvement in dengue infection has been rarely described in the medical literature and the pathophysiology is poorly understood. A rare and fatal case of primary dengue infection in a 34-year- old woman who returned from Papua New Guinea is reported; the unusual features of this case include severe primary dengue infection, myocarditis, and acute cerebral oedema resulting in death. This case demonstrates that severe atypical manifestations and fatality can occur with primary dengue infection.