Cystic echinococcosis (CE) is a zoonosis caused by Echinococcus granulosus. Uzbekistan is endemic for CE, but estimates of disease burden are lacking. We present findings from a cross-sectional, ultrasound-based survey evaluating the prevalence of human CE in the Samarkand region, Uzbekistan. The survey was conducted between September and October 2019 in the Payariq district, Samarkand. Study villages were selected based on sheep breeding and reported human CE. Residents aged 5-90 years were invited to receive a free abdominal ultrasound examination. The WHO Informal Working Group on Echinococcosis classification was used for cyst staging. Information regarding CE diagnosis and treatment was collected. Of 2,057 screened subjects, 498 (24.2%) were male. Twelve (0.58%) had detectable abdominal CE cysts. In total, five active/transitional (N = 1 CE1, N = 1 CE2, N = 3 CE3b) and 10 inactive cysts (N = 8 CE4, N = 2 CE5) were identified. Two participants had cystic lesions with no pathognomonic features of CE and were given a 1-month course of albendazole for diagnostic purposes. Twenty-three additional individuals reported previous surgery for CE in the liver (65.2%), lungs (21.6%), spleen (4.4%), liver and lungs (4.4%), and brain (4.4%). Our findings confirm the presence of CE in the Samarkand region, Uzbekistan. Additional studies are needed to assess the burden of human CE in the country. All patients with a history of CE reported surgery, even though most cysts found during the current study were inactive. Therefore, it appears there is a lack of awareness by the local medical community of the currently accepted stage-specific management of CE.