© 2016 Wiley Periodicals, Inc. Introduction: Duchenne muscular dystrophy (DMD) is an incurable neuromuscular disorder of childhood. Healthcare, caregiving, and other resource needs of affected individuals are thought to be substantial; however, the economic burden associated with DMD has not yet been assessed specifically in Australia. Methods: Australian households with a child with DMD were asked to complete a cross-sectional survey. Data were collected on annual resource utilization including hospital and medical services, equipment, home modifications, informal care, and working days lost. Results: Mean healthcare costs were found to be $10,046 Australian dollars per affected individual and were markedly higher than average Australian health expenditures at each age group. The mean total cost was $46,700 (median $32,300), with healthcare costs contributing 22% of total costs. Conclusions: The annual economic cost of DMD was found to be high, reflecting a significant socioeconomic burden, especially in boys who reach adulthood, where household resource use and caregiving burden is highest.