INTRODUCTION: Ovarian cancer with breast metastasis is an exceedingly rare entity. We hereby reported a case of ovarian cancer presenting with synchronous breast metastasis. CASE PRESENTATION: We report a case of a 39-year-old patient who was diagnosed with FIGO stage IV ovarian cancer, with pleural effusion and a suspected lesion in the breast. Biopsies of the breast lesion showed invasive carcinoma, immunohistochemistry stains of the breast biopsy specimen were positive for PAX-8, CA-125 markers, and negative for P53, SMA, Her2/neu, Mammaglobin. Immunohistochemistry results combined with the diagnosis of ovarian cancer helped confirm the ovarian origin of breast tumor. DISCUSSION: Ovarian cancer presenting with breast cancer metastasis concurrently is rare. The differential diagnosis between primary breast cancer and breast metastasis from ovarian cancer is challenging and immunohistochemistry results play a crucial role in establishing a definitive diagnosis in such cases. CONCLUSION: Oncologist and pathologist should be able to identify this rare clinical scenario promptly for early diagnosis and treatment which relies on thorough examination, imaging and immunohistochemical studies. Nevertheless, owing to the extensive spread of the disease, the prognosis for this rare clinical scenario is generally poor.