Knowledge about community awareness and practices related to a disease, among other things, helps to plan relevant control strategies. This study assessed the knowledge, attitude, and practices (KAPs) of the community in Abobo district of Gambella Regional State in Southwestern Ethiopia about urogenital schistosomiasis (UGS), which is endemic to the region. A community-based cross-sectional study was conducted in 2022. A pretested structured questionnaire was administered. Multivariable logistic regression was used to examine factors associated with KAPs variables. Most study participants (90.6%) responded they have previously heard of UGS. Over 95% of the participants knew at least 1 symptom of UGS and 30% knew the transmission cycle. About 15.9% and 26.8% knew keeping environmental hygiene and refraining from using cercariae infested water, as preventive methods, respectively. Over half of the participants (50.1%) disagreed or did not know that urinating close to the river/dam water contributes to transmission, and almost all participants had contact with the dam/river water in one way or another. Education status and history of having the infection were the key significant correlates of most KAPs variables. Most of the community members had poor knowledge and attitudes about the mode of transmission and preventive measures of UGS. Thus, besides school-based mass drug administration (MDA), UGS control efforts in the region should incorporate health education in conjunction with safe water supplies, and provision of sanitary facilities to effectively reduce the transmission of the disease.