Squamous cell carcinoma of the endometrium is extremely rare, accounting for approximately 1% of uterine body malignancies. The present report describes a case of squamous cell carcinoma of the uterine body arising from ichthyosis due to persistent pyometra. A 68‑year‑old Japanese woman, gravida 1 para 1, was referred to the Gynecological Department of Tokyo Women's Medical University, due to watery brown vaginal discharge which had persisted for 10 days. Pelvic magnetic resonance (MRI) imaging revealed a tumorous lesion occupying the cervical uteri, measuring 3 cm in diameter, and pyometra. The endometrial biopsy specimen obtained after drainage of the purulent fluid in the uterus was insufficient for obtaining a definitive pathological diagnosis. Under a clinical diagnosis of cervical tumor and pyometra, total transabdominal hysterectomy with bilateral salpingo‑oophorectomy was performed. Histopathologically, two primary cancers were diagnosed, i.e., cervical endometrioid carcinoma and squamous cell carcinoma of the endometrium. The patient was treated with additional chemotherapy with carboplatin (area under the curve, 6) and paclitaxel (175 mg/m2). The patient has remained alive, without recurrence, for 1 year to date. In this case, preexisting cervical endometrioid carcinoma caused stenosis of the uterine orifice and also resulted in pyometra. The uterine body squamous cell carcinoma appeared to have been caused by persistent inflammatory stimuli to the endometrium. One must be cautious regarding cancer in elderly postmenopausal females presenting with pyometra. The findings in this case report suggest that chronic inflammation of the endometrium may be associated with primary squamous cell carcinoma of the endometrium.

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