• Yue Wang, Shourong Hu, and Huifang Wang.
• Department of Ultrasound, Peking University Shenzhen Hospital, Shenzhen, China.
• Medicine (Baltimore). 2021 Dec 30; 100 (52): e28477e28477.

RationalePersistent urogenital sinus (PUG) with uterus didelphys and double vagina is a rare urogenital anomaly. The diagnosis is based on magnetic resonance examination and cystoscopy. To the best of our knowledge, there is no literature report of PUG diagnosed by ultrasound alone.Patient ConcernA 23-year-old woman presented with atypical menstruation and recurrent hematuria for 13 years and recurrent lower abdominal pain for 12 years.DiagnosisPUG was diagnosed through multiple ultrasound modalities, including transabdominal 2-dimensional ultrasound, transrectal bi-plane high-frequency ultrasound, and contrast-enhanced ultrasound. We diagnosed this malformation preoperatively by accurately measuring the length of urethra and common channel through multimodal ultrasound imaging.InterventionsUrethra separation and reconstruction, vaginal pull-through and artificial vaginoplasty, and bilateral hysterosalpingectomy were performed.OutcomesThe postoperative course was uneventful. She was urinating normally after half a year and used continuous vaginal dilatation to avoid stenosis.LessonsPUG associated with uterus didelphys and double vagina is an extremely rare malformation of the reproductive system. Multimodal ultrasound imaging can be used to diagnose this malformation preoperatively clearly and to accurately measure the length of urethra and common channel, providing an imaging basis for preparing an operative plan.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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