A case of unicornuate uterus with pathological rudimentary horn: settling associated adenomyosis with fertility preservation

OBJECTIVE: To report a rare case of a patient with unicornuate uterus, whose rudimentary horn has finally developed into diffuse adenomyosis foci, and to demonstrate a fertility-preserved approach for the treatment of such a condition. DESIGN: Step-by-step video of a rare case report. SUBJECTS: A 29-year-old nulliparous patient presented with severe dysmenorrhea, menorrhagia, and an infertility history for nearly 10 years. Her dysmenorrhea has been worsening in recent 2 years, and she has a strong desire of giving birth. The study was approved by the local ethics committee of Shanghai Jiao Tong University School of Medicine Affiliated International Peace Maternal and Child Health Hospital. The patient included in this video gave consent for publication of the video and posting of the video online including social media, the journal website, scientific literature websites (e.g., PubMed, ScienceDirect, and Scopus) and other applicable sites. EXPOSURE: Transvaginal ultrasonography, magnetic resonance imaging, and laparoscopic resection of rudimentary horn with adenomyosis, endometrioma, and superficial endometriosis were performed. Chromopertubation was performed to assess tubal competency. Laparoscopic resection was performed to relief pain and preserve fertility. MAIN OUTCOME MEASURES: Symptomatic relief, restoration of normal anatomy, and fertile reservation. RESULTS: During the follow-up, her dysmenorrhea resolved. Her ovarian reserve remained at 2.04 ng/mL after the surgery. She was considering embryo transfer. CONCLUSION: We reported a rare case of unicornuate uterus with adenomyosis in the rudimentary horn, with superficial endometriosis lesions and ovarian endometrioma. Although endometriosis is commonly noted in patients with uterine malformation, the development of adenomyosis is rare. The long-term periodic bleeding inside the obstructive rudimentary horn with functional endometrium may help the development of adenomyosis, indicating an advanced stage of this uterine malformation. Early diagnosis of such a uterine malformation is quite necessary to prevent such severe complications. The existence of adenomyosis should be considered in women with infertility with uterine malformations. The laparoscopic resection of the pathological rudimentary horn is possible after careful adhesiolysis without cutting the uterine artery to relief clinical symptoms, restore normal pelvic anatomy, and preserve future fertility.