To study the expression profiles of circulating microRNAs (miRNAs) in newly-developed advanced schistosomiasis patients. Ten healthy controls (Group 1), ten patients with schistosomiasis history (without advanced schistosomiasis, Group 2) and ten patients with newly-developed advanced schistosomiasis (Group 3) were randomly selected from Jiangsu Province. The expression profiles of 570 human related circulatingmiRNAs were measured and analyzed with the Agilent Human microRNAs microarray Rel 12.0. Compared to the healthy controls, four miRNAs were up-regulated, while 16 miRNAs were down-regulated in the patients with newly-developed advanced schistosomiasis. Moreover, six miRNAs were up-regulated, while 21 miRNAs were down-regulated in the patients in Group 2. Among of them, the expression level of miR-383 was significantly decreased by 4.23 and 11.82 folds in Group 2 and Group 3, respectively. There are significant differences among the healthy controls, patients with schistosomiasis history (without advanced schistosomiasis) and the patients with newly-developed advanced schistosomiasisin the expression profiles of circulatingmiRNAs. Moreover, circulating miR-383 might be involved in the development of newly-developed advanced schistosomiasis.