Schistosomiasis is a serious public health issue in the world infecting more than 200 million people. In Maranhão state, Brazil, the disease has a high prevalence in the Baixada Maranhense microregion, where the socioeconomic conditions, high prevalence of freshwater snails, elevated incidence of human infection, and large rodent populations make the area highly conducive to the life cycle and persistence of schistosomiasis. The objective of this study was to record the perception of residents and health/public education professionals regarding this parasitosis and also understand their knowledge of the relationship between schistosomiasis and the environment. We conducted 53 interviews addressing the socioeconomic, behavioral, and environmental issues surrounding schistosomiasis. It was recorded that the population believed the environment is the main cause of the disease and the fisherman may be stigmatized by their chance of having the disease. Health/public educational professionals in the region indicated that there were no regular educational campaigns, which contributed to the high rates of reinfection and the difficulties in preventing and controlling the spread of the disease. These data clearly demonstrate the need for interdisciplinary work that engages the community in gathering and disseminating knowledge and developing solutions to minimize the occurrence of this major health issue in the region.