Background <p>Fallopian tube leiomyomas are extremely rare benign tumors, particularly when compared to the high incidence of uterine leiomyomas. While often asymptomatic and discovered accidentally, they can present as clinical symptoms such as abdominal pain, ectopic pregnancy, and infertility. The pathogenesis remains unclear, though hormonal factors may play a role.</p> Case presentation <p>We present a case of a 20-year-old female with a history of polycystic ovarian syndrome (PCOS) presenting with secondary infertility, irregular menses, and abdominal pain. Hormonal assays indicated elevated LH levels consistent with PCOS, and ultrasonography revealed a hypoechoic lesion in the right adnexa. Diagnostic laparoscopy identified polycystic ovaries and a distinct lesion located in the ampulla of the right fallopian tube. The mass was excised, and histopathological examination confirmed a benign leiomyoma of the fallopian tube.</p> Conclusion <p>Although tubal occlusion is a common cause of infertility, fallopian tube leiomyoma is a rare etiology that should be considered in the differential diagnosis. This case highlights the importance of investigating tubal pathologies in infertility patients, especially when co-existing conditions like PCOS are present or when patients are unresponsive to standard treatments.</p>