• Jianqi Li.
• Department of Obstetrics and Gynecology; Guangdong Provincial Key Laboratory of Major Obstetric Diseases; Guangdong Provincial Clinical Research Center for Obstetrics and Gynecology; Guangdong-Hong Kong-Macao Greater Bay Area Higher Education Joint Laboratory of Maternal-Fetal Medicine, The Third Affiliated Hospital of Guangzhou Medical University, Guangzhou, China.
• Medicine (Baltimore). 2023 Nov 3; 102 (44): e35907e35907.

RationaleOvarian squamous cell carcinoma (OSCC) is an exceedingly rare subtype, and high-risk human papillomavirus (HPV)-related OSCC is even rarer.Patient ConcernsAn 8-cm diameter ovarian cyst was detected during a routine B-ultrasound examination, and the patient underwent laparoscopic surgery. Postoperative pathological examination revealed HPV-16-related OSCC involving adjacent uterine tissue. The patient received postoperative radiotherapy and chemotherapy.DiagnosesHigh-risk HPV-related OSCC.InterventionsNo.OutcomesThe patient was finally diagnosed with high-risk HPV-related OSCC and underwent surgical treatment.Lessons SubsectionsIn patients with high-grade cervical intraepithelial neoplasia who have undergone cervical conization and maintained annual HPV negativity, the possibility of high-risk HPV-related OSCC should be considered, despite its extreme rarity. Vigilance is essential in the presence of ovarian cysts even after HPV clearance.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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