Ovarian cancer is usually limited to the abdomen and frequently remains confined. The occurrence of extrabdominal mestastases is unusual. In this report we describe a rare case of axillary involvement at initial presentation of ovarian cancer in a 48-year-old woman. The axillary mass was the only clinical abnormality. Cytological and histological findings, performed on axillary lymph nodes, showed the presence of psammoma bodies and specific immunohistochemical tumor markers (OC-125 and WT1), supporting the evidence of a metastatic axillary lymphadenopathy from ovarian cancer. Subsequently, chest and abdominopelvic computed tomography showed a right ovarian complex mass of 30 x 25 mm and biochemical tests showed high levels of CA125. Surgical therapy was performed. Histology confirmed the diagnosis, evidencing a poorly differentiated serous-papillary carcinoma of the right ovary. In conclusion, cytological and histological findings can play a crucial role in suggesting the correct origin of a metastatic adenocarcinoma when the clinical presentation is atypical.