• Liya Ding, Wangwang Liu, Hui Li, Dingpin Huang, Yang Chen, and Huimin An.
• Department of Pathology, Sir Run Run Shaw Hospital, Zhejiang University School of Medicine, Hangzhou 310000, China.
• Medicine (Baltimore). 2024 Jul 12; 103 (28): e38792e38792.

RationaleIchthyosis uteri is a rare pathological condition characterized by the replacement of the endometrial lining by stratified squamous epithelium. Yet its occurrence with endometrial adenocarcinoma is very rare.Patient ConcernsA 68-year-old woman has been experiencing sporadic, minor vaginal hemorrhages for a few months. The gynecological evaluation revealed a uterine enlargement and imaging demonstrated an irregular mass within the uterus.DiagnosisEndometrial adenocarcinoma with transitional cell differentiation; ichthyosis uteri with dysplasia.InterventionsRadical hysterectomy with pelvic lymphadenectomy was performed followed by postoperative radiotherapy.OutcomesPostoperative follow-up at 8 months showed a favorable outcome without signs of recurrence and metastasis.LessonsAdequate pathological sampling is crucial to identifying the accompanying lesions of ichthyosis uteri. Finding molecular alterations in various pathological morphologies is important to understand the evolution of disease.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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