Schistosomiasis is a neglected parasitic disease with high morbidity and mortality.Currently, the treatment of this disease depends almost exclusively on praziquantel;however, the emergence of drug resistance to praziquantel in schistosomes makes the development of novel drugs an urgent task.Aldose reductase (AR), an important component that may be involved in the schistosome antioxidant defense system, is believed to be a potential drug target.In this study, we first resolved the Schistosoma japonicum AR (SjAR) structure through the X-ray diffraction method and then identified 10 potential inhibitors from the Maybridge HitFinder library by virtual screening based on this structural model.Further in vitro experiments showed that one of the compounds, renamed as AR9, exhibited good antischistosomal activity as well as significant inhibition on the enzyme activity of recombinant SjAR (rSjAR) protein.Cytotoxicity analysis revealed that AR9 had relatively low toxicity towards host cells.These conclusions suggest that AR9 meets the criteria to become a lead compound for developing novel antischistosomal drugs.Additionally, the work presented here bridges the gap between virtual screening and experimental validation, providing an effective and economical strategy for the development of new anti-parasitic drugs.