We present a case of ovarian sex cord tumor with annular tubules (SCTAT) in a patient that received neoadjuvant chemotherapy with 3.5 years of follow-up. Neo-adjuvant chemotherapy is a common practice in advanced cases of ovarian carcinoma and is rarely applied in nonepithelial ovarian tumors. Chemotherapy-induced morphologic alterations of the subsequently resected tissue samples are well recognized; in this case, chemotherapy-induced cytoarchitectural changes created diagnostic difficulties in the subsequently resected specimen.