Schistosomiasis has been associated to urinary bladder, liver, colorectal and cervical cancer. However, its role in ovarian malignancy has not been described. With the premise that long-standing inflammation secondary to chronic infection predisposes to cancer by promoting an environment that cultivates genomic lesions and tumor initiation, does chronic infection with Schistosomiasis also predispose to ovarian malignancy? We presented a case of a 54 year-old with chronic Schistosoma infection, who was diagnosed with high-grade serous carcinoma of the right ovary. In this case, the infection reached the pelvic organs infesting the right fallopian tube and left ovary. With chronic inflammation, there was subsequent damage to deoxyribonucleic acid (DNA) caused by mutation and activation of oncogenes, oxidative stress from fluke-derived products and physical damage of host tissues as the parasites developed. Ultimately, these mechanisms led to tumor initiation, promotion and progression. However, only the right fallopian tube developed the cancer. Consistent with the hypothesis that premalignant lesions at the fimbriated end of the fallopian tube is the origin of high grade ovarian serous carcinoma, we supposed that the primary site of malignancy is the right fallopian tube. Based on proximity, the malignant cells could have seeded to the right ovary, leading to the high grade serous carcinoma of the patient.