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Background: Genitourinary tuberculosis (GUTB) is rare but fatal if not diagnosed early. The purpose of this study was to investigate the outcomes of GUTB in Taiwan. Methods: We retrospectively reviewed medical records of 57 patients who were diagnosed as GUTB from January 2002 to December 2016, over a 15-year period. Demographic data and clinical manifestations were recorded for analysis. Results: There were 37 males and 20 females with a median age of 71 years. Kidney (24.6%) was the most involved organ. Fever (56.1%) was the major presentation. Sixteen (28.1%) patients presented unfavorable outcome. Compared with the favorable outcome group, the unfavorable outcome group had more malignancy (p = 0.013), fever (p = 0.020), anemia (p = 0007), thrombocytopenia (p = 0.003), and hypoalbuminemia (p = 0.015). In a multivariate analysis, fever (odds ratio: 42.716, 95% confidence interval: 1.032–1767.569; p = 0.048) was identified as prognostic factors for unfavorable outcome. Conclusion: GUTB is often in advanced stages with a high mortality in Taiwan. Establishing a diagnosis is difficult and requires thorough investigation. Fever is associated with unfavorable outcome. Keywords: Genitourinary tuberculosis, Genitourinary tract surgery, Anemia