研究动态
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缪勒管异常的一个误导性案例:肌瘤变性和生长,涉及非功能性、非沟通性的初级角。

A misleading case of Mullerian anomaly: fibroid degeneration and growth involving non-functional, non-communicating rudimentary horn.

发表日期:2024 Jul 27
作者: Luca Giannella, Leonardo Natalini, Andrea Ciavattini
来源: FERTILITY AND STERILITY

摘要:

报告一种罕见的、误导性的肌瘤变性,涉及一位单角子宫女性的非功能性、非沟通性角。虽然功能性的初级角的存在可能会导致建议将其去除的体征和症状,但非功能性的病例很少被报道,并且由于它们明显的功能不活动,因此从未报道过需要去除它们。之前的报告没有显示非功能性的初级角退化过程可能导致患者不适。这是一个分步解说视频,展示了肌瘤变性和非功能性、非沟通性的初级角生长的独特案例。单角子宫角(ASRM 分类 2021)及其手术治疗。大学学术医院“A.O.U.”马尔凯 - 马尔凯理工大学”安科纳,意大利。一名 48 岁白人未产绝经前妇女因腹痛和附件肿块增大而被转诊到我们机构。她的个人病史显示原发性不孕症,之前诊断为单角子宫考虑到这些病例中存在异位输尿管的可能性,进行了完整的计算机断层扫描,未发现泌尿生殖系统改变,术前成像(超声评估、计算机断层扫描和磁共振)提供了可疑卵巢纤维瘤的临时诊断。考虑到患者的年龄、缺乏怀孕意愿以及附件肿块体积增大,计划进行腹腔镜手术以进行肿块切除和预防性双侧输卵管切除术,因此,患者被告知潜在恶变的风险较低。双方一致决定在手术结束时通过小型剖腹手术切除完整的肿块。进入腹腔后,发现右侧单角子宫与同侧阔韧带、输卵管和卵巢具有解剖连续性。这些结构完全附着在右骨盆壁上。在另一侧,与右半子宫相比,在颅骨上发现一个圆形肌瘤样肿块,与左侧阔韧带、输卵管和卵巢直接相连。鉴于术中发现的变化,进行了左侧残角截肢和预防性双侧输卵管切除术。显示腹腔镜治疗的单角子宫(ASRM 分类 2021)中无功能、非交通残角的肌瘤变性和生长.腹腔镜子宫角切除术成功,术中及术后无并发症发生。在六个月的随访中,患者健康状况良好。组织病理学检查证实了子宫肌瘤变性和子宫内膜缺失。文献报道中缺乏有症状的无功能角的病例,导致妇科医生认为它们是一种沉默的苗勒氏管异常。这个独特的案例表明,即使是无功能的初级角也可能经历需要手术的症状转变过程。该信息可能有助于对女性进行更全面的咨询,并有助于在误导性苗勒管异常的诊断检查中考虑发生这种情况的可能性。考虑到这一点,与附件病理学的预期相比,随着技术方面的变化,也可以更好地规划手术治疗。版权所有 © 2024。由 Elsevier Inc. 出版。
To report a rare, misleading fibroid degeneration involving a non-functional, non-communicating horn in a woman with a unicornuate uterus. While the presence of a functional rudimentary horn may lead to signs and symptoms that recommend its removal, non-functional cases are rarely reported, and due to their apparent functional inactivity, the need for their removal has never been reported. No previous report showed the possibility of a degenerative process in a non-functional rudimentary horn causing patient discomfort.This is a step-by-step narrated video showing a unique case of fibroid degeneration and growth of a non-functional, non-communicating rudimentary horn in a unicornuate uterus (ASRM classification 2021) and its surgical management.University Academic Hospital ''A.O.U. delle Marche - Università Politecnica delle Marche" Ancona, Italy.A 48-year-old Caucasian nulliparous premenopausal woman was referred to our institution because of abdominal pain and an enlarging adnexal mass. Her personal history showed primary infertility with a previous diagnosis of unicornuate uterus. Given the possibility of ectopic ureters in these occurrences, a complete computed tomography scan was performed, and no genitourinary alterations were found. Pre-operative imaging (ultrasound evaluation, computed tomography, and magnetic resonance) provided a provisional diagnosis of a suspicious ovarian fibroma.Considering the patient's age, lack of desire for pregnancy, and volumetric increase of the adnexal mass, a laparoscopic intervention to perform mass removal and prophylactic bilateral salpingectomy was planned. The patient was counseled about the low risk of an underlying malignant transformation. Therefore, the decision to remove the intact mass via a mini-laparotomy at the end of the surgery was shared. Once the abdominal cavity was entered, the right unicornuate uterus was found in anatomical continuity with the ipsilateral broad ligament, fallopian tube, and ovary. These structures were wholly attached to the right pelvic wall. On the other side, cranially compared to the right hemi-uterus, a roundish myoma-like mass was detected in direct connection with the left broad ligament, fallopian tube, and ovary. In light of a changed intraoperative finding, the amputation of the left rudimentary horn and prophylactic bilateral salpingectomy were performed.Showing the fibroid degeneration and growth of a non-functional, non-communicating rudimentary horn in a unicornuate uterus (ASRM classification 2021) treated laparoscopically.Laparoscopic removal of the uterine horn was successful, and no intra- and postoperative complications occurred. The patient was in good health at the six-month follow-up visit. The histopathological examination confirmed the fibroid degeneration and the absence of endometrium.The lack of symptomatic cases of rudimentary non-functional horns reported in the literature led gynecologists to consider them a silent Mullerian anomaly. This unique case demonstrates that even non-functional rudimentary horns can undergo symptomatic transformation processes requiring surgery. This information may be helpful for more comprehensive counseling of women and for considering the possibility of this occurrence in the diagnostic work-up of misleading Müllerian anomalies. With this in mind, surgical treatment can also be better planned as the technical aspects change compared to what is expected for an adnexal pathology.Copyright © 2024. Published by Elsevier Inc.