To analyze the diagnosis and treatment of two imported cases with schistosomiasis haematobia, so as to provide insights into improving the diagnosis and treatment and avoiding misdiagnosis and mistreatment of imported schistosomiasis haematobia.The medical records and epidemiological data pertaining to the two cases were collected. The stool and urine samples were collected for identification of Schistosoma eggs using the Kato-Katz technique and direct smear method after centrifugal precipitation, and blood samples were collected for detection of anti-Schistosoma antibody. Following definitive diagnosis, the patients were given praziquantel therapy.The patient 1, a Malagasy, was infected in Madagascar and returned to China for delivery. The case presented intermittent painless terminal hematuria symptoms, and showed no remarkable improvements following multiple-round treatments in several hospitals. In January 2017, she was found to be positive for anti-Schistosoma antibody, negative for feces test, and positive for S. haematobium eggs in urine test, and miracidia were hatched from eggs. Then, the case was diagnosed as schistosomiasis haematobia. Patient 2 worked in Republic of Malawi for many years, and presented intermittent painless terminal hematuria since October 2018; however, no definite diagnosis or effective treatment was received after admission to multiple hospitals. In March 2019, pathological examinations showed a number of eggs in the interstitium of the bladder mass, accompanied by a large number of eosinophils, which was consistent with schistosomiasis cystitis. In April 2019, he was tested positive for serum anti-Schistosoma antibody, negative for the fecal test, and had S. haematobium eggs in urine samples, with miracidia hatched from eggs. Then, the case was diagnosed as schistosomiasis haematobia. Following treatment with praziquantel at a dose of 60 mg/kg, all symptoms disappeared.Overseas imported schistosomiasis haematobia is likely to be misdiagnosed. The training pertaining to schistosomiasis control knowledge requires to be improved among clinical professionals, in order to avoid misdiagnosis and mistreatment.