BACKGROUND:Schistosomiasis is a parasitic disease which affects almost 300 million people worldwide each year. It is highly endemic in Mozambique. Prevention and control of schistosomiasis relies mainly on mass drug administration (MDA), as well as adoption of basic sanitation practices. Individual and community perceptions of schistosomiasis are likely to have a significant effect on prevention and control efforts. In order to establish a baseline to evaluate a community engagement intervention with a focus on schistosomiasis, a survey to determine knowledge, attitudes and practices relating to the disease was conducted. METHODOLOGY/PRINCIPAL FINDINGS:A representative cross-sectional household survey was carried out in four districts of Nampula province, Mozambique. Interviews were conducted in a total of 791 households, using a structured questionnaire. While awareness of schistosomiasis was high (91%), correct knowledge of how it is acquired (18%), transmitted (26%) and prevented (13%) was low among those who had heard of the disease. Misconceptions, such as the belief that schistosomiasis is transmitted through sexual contact (27%), were common. Only about a third of those who were aware of the disease stated that they practiced a protective behaviour and only a minority of those (39%) reported an effective behaviour. Despite several rounds of MDA for schistosomiasis in the recent past, only a small minority of households with children reported that at least one of them had received a drug to treat the disease (9%). CONCLUSION/SIGNIFICANCE:Poor knowledge of the causes of schistosomiasis and how to prevent it, coupled with persisting misconceptions, continue to pose barriers to effective disease prevention and control. To achieve high levels of uptake of MDA and adoption of protective behaviours, it will be essential to engage individuals and communities, improving their understanding of the causes and symptoms of schistosomiasis, recommended prevention mechanisms and the rationale behind MDA.