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An extremely rare case of large cell variant of ovarian small cell carcinoma is described. A 34-year-old woman (gravida 1, para 1) had a unilateral ovarian mass measuring 17 cm in greatest diameter and a metastatic lesion in the omentum. Microscopically, the tumor showed a diffuse arrangement of large, closely packed epithelial cells with abundant eosinophilic cytoplasm and large nuclei with prominent nucleoli. The tumor cells also were arranged in follicle-like and trabecular structures and cords. Immunohistochemically, many tumor cells were diffusely positive for epithelial membrane antigen and some cells contained cytokeratin CAM5.2, vimentin, neurofilament, neuron-specific enolase, or alpha-1 antitrypsin. However, no specific lineage was detected. The tumor was aneuploid by flow cytometry. The patient received chemotherapy postoperatively. However, the patient showed metastases in the inguinal and retroperitoneal lymph nodes. The serum calcium level, which was not measured preoperatively, was mildly elevated postoperatively. The patient was well with no evidence of disease 17 months after diagnosis. This tumor must be distinguished from other primary or metastatic 'undifferentiated' neoplasms, especially ovarian small cell carcinoma of pulmonary type and granulosa cell tumor.