Mullerian tumor arising from pleura is a rare occurrence and under-reported in medical literature. We hereby present such a case in a 47 years old female who presented with pleural effusion. Pleural biopsy revealed a papillary tumor with mild-to-moderate nuclear atypia. Immunohistochemistry profile with positivity for WT1, Pax8, ER, and PR together with negative mesothelial markers and ruling out primary in female genitalia led us to a diagnosis of primary pleural serous carcinoma of mullerian origin. The patient received paclitaxel and carboplatin chemotherapy and is disease-free till now after 2 years of follow-up.
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