Objective: Hospital and laboratory data indicate that human T-lymphotropic virus type 1 (HTLV-1) is endemic to central Australia, but no community-based studies of its prevalence or disease burden have been reported. We determined the prevalence rates of HTLV-1 infection and of HTLV-1-associated diseases in a remote Indigenous community. Setting: A remote Northern Territory community. Design: All residents were asked to complete a health survey and offered a limited clinical examination, together with serological tests for HTLV-1 and Strongyloides, and HTLV-1 proviral load (PVL) assessment. Main outcome measures: HTLV-1 seropositivity rates; HTLV-1 PVL (copies/105 peripheral blood leucocytes [PBL]); presentation with HTLV-1-related clinical disease. Results: HTLV-1 serostatus was determined for 97 of 138 residents (70%). The prevalence of HTLV-1 infection was significantly higher among adults (30 of 74 people tested) than children (1 of 23; P = 0.001). Nine of 30 HTLV-1-positive adults had a clinical syndrome that was potentially attributable to HTLV-1 infection (chronic lung disease, seven; symptomatic strongyloidiasis, two). The median HTLV-1 PVL was significantly higher for adults with chronic lung disease than for those who were asymptomatic (chronic lung disease, 649 copies/105 PBL [IQR, 162e2220]; asymptomatic adults, 40 copies/105 PBL [IQR, 0.9e229]; P = 0.017). Ten of 72 adults tested were seropositive for Strongyloides (six of 28 HTLV-1-positive participants and four of 44 HTLV-1-negative participants; P = 0.17), as were three of 15 children tested; the three children were HTLV-1-negative. Conclusion: The prevalence of HTLV-1 infection and the rate of disease potentially attributable to HTLV-1 were high among adults in this remote community.