Objective To assess associations between anterior and/or fundal uterine leiomyoma and overactive bladder syndrome. Methods The present cohort study recruited women diagnosed with fundal/anterior uterine leiomyoma by standardized transvaginal ultrasonography at the Medical University of Vienna, Austria, between January 1, 2010, and December 31, 2013, in addition to an age-matched control group of women without uterine leiomyoma. The International Consultation on Incontinence Questionnaire Overactive Bladder Module (ICIQ-OAB) was mailed to all eligible participants. The main outcome was the ICIQ-OAB sum score. Results Among 304 questionnaires sent out, 129 were returned. After the exclusion of incomplete datasets, 80 women were included in the analysis (uterine leiomyoma group, 43; control group, 37). The mean ± SD ICIQ-OAB sum score was 9.7 ± 10.2 for women with uterine leiomyoma and 4.2 ± 5.3 for women in the control group; thus, the ICIQ-OAB sum score was on average 5.5 points higher in the uterine leiomyoma group (P=0.003). The Spearman correlation coefficient between the total volume of leiomyoma per woman and the ICIQ-OAB sum score was 0.072 (P=0.645). Conclusion The study found a significant association between anterior and/or fundal leiomyoma and overactive bladder syndrome. The presence of uterine leiomyoma should be ruled out during the evaluation of overactive bladder.