Co-occurrence of Unicornuate Uterus with Noncommunicating Functioning Rudimentary Horn and VACTERL Association-A Case Report.

IF 1.7 4区医学 Q3 OBSTETRICS & GYNECOLOGY Journal of pediatric and adolescent gynecology Pub Date : 2024-11-12 DOI:10.1016/j.jpag.2024.11.001

Jibladze Ana, Kristesashvili Jenaro, Asanidze Elene, Kutateladze Mariam, Chapidze Ia

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Abstract

Introduction: The co-occurrence of a unicornuate uterus with a noncommunicating, functioning rudimentary horn and VACTERL association represents an extremely rare condition, with only 3 similar cases reported in the literature.

Case: The patient, aged 12 years and 9 months, presented with pelvic pain and severe dysmenorrhea, which started shortly after her menarche at 12 years and 4 months. At birth, she exhibited 3 characteristic components of VACTERL association: bronchoesophageal fistula/esophageal atresia, anal atresia, and polydactyly. Magnetic resonance imaging revealed a unicornuate uterus with a noncommunicating, functioning rudimentary horn, and subsequently, VACTERL association was diagnosed. The rudimentary horn was laparoscopically excised 8 months post-menarche, resulting in symptom resolution.

Conclusion: Health care providers should consider the possibility of genital anomalies when diagnosing VACTERL association, especially in cases presenting with severe early dysmenorrhea.

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单角子宫与无交流功能原始角和 VACTERL 相关性并存--病例报告。

导言：病例：患者 12 岁零 9 个月，在 12 岁零 4 个月初潮后不久出现盆腔疼痛和严重痛经。出生时，她表现出 VACTERL 相关的三个特征：支气管食道瘘/食道闭锁、肛门闭锁和多趾。核磁共振成像显示 UUNCFRH，随后确诊为 VACTERL 联体。初潮后8个月，通过腹腔镜切除了发育不全的肛角，症状得到缓解：结论：医疗服务提供者在诊断VACTERL合并症时应考虑生殖器异常的可能性，尤其是出现严重早期痛经的病例。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Journal of pediatric and adolescent gynecology 医学-妇产科学

CiteScore

3.90

自引率

11.10%

发文量

251

审稿时长

57 days

期刊介绍： Journal of Pediatric and Adolescent Gynecology includes all aspects of clinical and basic science research in pediatric and adolescent gynecology. The Journal draws on expertise from a variety of disciplines including pediatrics, obstetrics and gynecology, reproduction and gynecology, reproductive and pediatric endocrinology, genetics, and molecular biology. The Journal of Pediatric and Adolescent Gynecology features original studies, review articles, book and literature reviews, letters to the editor, and communications in brief. It is an essential resource for the libraries of OB/GYN specialists, as well as pediatricians and primary care physicians.