Background: Schistosomiasis remains one of the most common parasitic diseases worldwide causing considerable deaths especially among people in the Sub-Saharan region. This study determined the association between urinary Schistosomiasis and lipid profile among school children in Fante Akura, Yeji. Materials and Methods: This simple randomized case-control study was conducted among 50 primary school students with Schistosoma haematobium infection and 50 healthy control students in Fante Akura, Yeji, from January, 2014 to March, 2014. Urine and blood samples were obtained and examined for the presence of S. haematobium and assessed their lipid profile respectively. A structured questionnaire was employed to obtain information from the study participants on their socio-demographic characteristics as well as on the risk factors that can predispose study participants to S. haematobium infection. Results: The mean serum level of low-density lipoprotein cholesterol (LDL-C) was reduced significantly in schistosome-infected participants in comparison to controls (P<0.001). The mean serum levels of triglyceride (TG) (p=0.028), LDL-C (p=0.011) were significantly higher in participants with light S. haematobium infection intensity compared to participants with heavy S. haematobium infection intensity (P=0.028). The mean serum level of total cholesterol (TC) and high-density lipoprotein cholesterol (HDL-C) were higher (P>0.05) in participants with light SH infection intensity compared to participants with heavy SH infection intensity. Conclusion: S. haematobium infection presented significant changes in serum levels of total cholesterol, triglycerides and low-density lipoproteins in participants infected with S. haematobium with a corresponding raised urine parasite count.