There is increasing evidence that various in vitro and in vivo models of neuroschistosomiasis may be useful to study its pathophysiology, the clinical manifestations of the infection, and its effects on cerebral and spinal structures as well as the immune system. The use of animal models to study neuroschistosomiasis began in 1996 with the CD1 mice, followed by studies in Swiss mice, BALB/C mice, C57 mice, New Zealand rabbits, and Wistar rats. These animal models make it possible to study factors that contribute to the spread of the disease, its infectivity, and possible treatment effects. However, better models are needed to fully understand the characteristics of the infection, develop immunotherapies and vaccinations, and figure out how to effectively manage and eradicate it in the long term.