Objectives Malignant Brenner tumors (MBT) of the ovary are rare disease; representing <1% of all ovarian cancers and 3–5% of Brenner tumors. They carry a poor prognosis. They generally affect women during the perimenopausal and postmenopausal periods. The standard treatment is surgery; however, the indication of adjuvant chemotherapy remains controversial. Our aim is to report our experience in the treatment of MBT of the ovary, to better characterize this disease.

Methods A retrospective case series involving 4 patients diagnosed with MBT of the ovary and treated between 2006 and 2014.

Results The median age of our patients was 59.2 years. Three patients were in the menopause period. All women conducted surgery following by adjuvant chemotherapy. Two patients presented a loco regional recurrence that occurred respectively after 9 and 11 months. The treatment was based on chemotherapy. Three patients presented distant metastasis. The treatment combined chemotherapy and surgery.

Conclusions The treatment approach of MBT of the ovary is not well established since its scarcity and poor prognosis. Thus, more case series and meta-analysis should be conducted.