IntroductionWe report a case of a 35-year-old woman, who was referred to our Gynaecology Clinic and had been suffering from subfertility the previous three years. The aim of this report is to present diagnostic pathway, treatment and outcome of a rare case involving schistosomiasis infection, as a non-common cause of infertility. Case report A thorough investigation followed, including recent and past medical as well as social and family history, systematic review, clinical and gynaecological examination, blood and urine tests, cultures, Mantoux test, dye test, cervical smear, chest X-ray, colposcopy, hysteroscopy and, finally, laparoscopy. Schistosomiasis was proven and treatment with Praziquantel started. In the follow-up period, hysterosalpingogram and then laparoscopic adhesiolysis and salpingostomy were arranged to continue the management of her primary infertility. Also cystoscopic evaluation took place and the patient commenced Clomiphene 100 mg. Conclusion Although we were afraid for this woman having at least right-sided tubal disease, her best option for future fertility would be in vitro fertilisation; at 10 months following surgery, she conceived and gave birth to a healthy 3.1 kg male infant with normal delivery at full term.