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自发性单侧双胎活胎异位妊娠:对报告病例、诊断挑战及管理方法的系统评价

Spontaneous Unilateral Twin Live Ectopic Pregnancy: A Systematic Review of Reported Cases, Diagnostic Challenges, and Management Approaches.

作者信息

Sidhar Meenakshi, Malik Neeru, Dhankar Vijay, Lochan Dakshika, Rastogi Vyomesh, Sehgal Nidhi Prabha, Talwar Nikhil, Madan Nikita, Zomorrodian Albert A, Kochar Rishabh

机构信息

Department of Pathology, Dr. Baba Saheb Ambedkar Medical College and Hospital, New Delhi, IND.

Department of Obstetrics and Gynecology, Dr. Baba Saheb Ambedkar Medical College and Hospital, New Delhi, IND.

出版信息

Cureus. 2025 Aug 18;17(8):e90378. doi: 10.7759/cureus.90378. eCollection 2025 Aug.

Abstract

Unilateral twin live ectopic pregnancy is a rare and potentially life-threatening obstetric condition, marked by the implantation of two viable embryos within the same fallopian tube. These cases pose significant diagnostic and therapeutic challenges, particularly when fetal cardiac activity is present. This systematic review consolidates existing literature to explore the clinical presentation, underlying pathophysiology, diagnostic modalities, and management strategies associated with this uncommon condition, with a focused discussion on maternal outcomes. A comprehensive literature search was conducted across PubMed, Embase, Scopus, and the Cochrane Database of Systematic Reviews for articles published from January 1945 to May 2025. Both MeSH terms and non-MeSH keywords, including "Unilateral," "Twin," "Live," and "ectopic pregnancy," were used, yielding 503 records. After applying inclusion and exclusion criteria and manual screening, 17 case reports published between 1994 and 2023 were included for final analysis. Eligible studies were limited to English-language reports describing spontaneous unilateral twin ectopic pregnancies with confirmed fetal cardiac activity and adequate clinical details. Studies lacking these elements were excluded. The Joanna Briggs Institute (JBI) Critical Appraisal Checklist for Case Reports was used to assess the risk of bias (ROB) in this study. Due to the descriptive nature of the included studies, formal grading of evidence was not applied. All 17 included cases described women aged 24 to 44 years, typically presenting between six and 12 weeks of gestation with abdominal pain, vaginal bleeding, and amenorrhea. Transvaginal ultrasonography (TVUS) was the most frequently used diagnostic modality, revealing two gestational sacs with cardiac activity within a single fallopian tube. Serum serial β-human chorionic gonadotropin (β-hCG) levels were consistently elevated above the discriminatory threshold. In one particular case, advanced imaging such as magnetic resonance imaging (MRI) was used to clarify anatomy or assess rupture risk. All cases except one were managed surgically, most commonly through laparoscopic salpingectomy or salpingostomy. Methotrexate therapy was attempted in one case and is generally deemed inappropriate due to the presence of viable embryos. No concurrent intrauterine pregnancies were observed. Favorable maternal outcomes were reported in all cases when diagnosis and treatment were timely. Findings from this review are limited by the rarity of the condition, reliance on isolated case reports, and the lack of standardized diagnostic or treatment protocols. There is also potential for publication bias and underreporting of negative outcomes. Nonetheless, this review emphasizes the importance of early and accurate diagnosis using TVUS, supplemented by MRI when indicated, followed by prompt surgical management to optimize maternal safety. Increased awareness, reporting, and documentation of such rare presentations are crucial to guiding future clinical decision-making and preventing catastrophic complications. No external funding was received for this review. This review was not registered in any database due to its retrospective, descriptive nature.

摘要

单侧双胎存活异位妊娠是一种罕见且可能危及生命的产科疾病,其特征是两个存活胚胎在同一输卵管内着床。这些病例带来了重大的诊断和治疗挑战,尤其是在存在胎儿心脏活动的情况下。本系统评价整合现有文献,以探讨与这种罕见疾病相关的临床表现、潜在病理生理学、诊断方法和管理策略,并重点讨论孕产妇结局。我们在PubMed、Embase、Scopus和Cochrane系统评价数据库中进行了全面的文献检索,以查找1945年1月至2025年5月发表的文章。使用了医学主题词(MeSH)和非MeSH关键词,包括“单侧”“双胎”“存活”和“异位妊娠”,共检索到503条记录。在应用纳入和排除标准并进行人工筛选后,纳入了1994年至2023年发表的17例病例报告进行最终分析。符合条件的研究仅限于描述有确诊胎儿心脏活动且临床细节充分的自发性单侧双胎异位妊娠的英文报告。缺乏这些要素的研究被排除。本研究使用乔安娜·布里格斯研究所(JBI)病例报告批判性评价清单来评估偏倚风险(ROB)。由于纳入研究的描述性性质,未应用正式的证据分级。所有17例纳入病例均描述为年龄在24至44岁之间的女性,通常在妊娠6至12周时出现腹痛、阴道出血和闭经。经阴道超声检查(TVUS)是最常用的诊断方法,显示单个输卵管内有两个有心脏活动的妊娠囊。血清连续β-人绒毛膜促性腺激素(β-hCG)水平持续高于鉴别阈值。在一个特定病例中,使用了诸如磁共振成像(MRI)等先进成像技术来明确解剖结构或评估破裂风险。除1例病例外,所有病例均采用手术治疗,最常见的是通过腹腔镜输卵管切除术或输卵管造口术。有1例尝试了甲氨蝶呤治疗,但由于存在存活胚胎,一般认为不合适。未观察到并发宫内妊娠。所有病例在诊断和治疗及时的情况下均报告了良好的孕产妇结局。本综述的结果受到该疾病罕见性、依赖孤立病例报告以及缺乏标准化诊断或治疗方案的限制。也存在发表偏倚和负面结果报告不足的可能性。尽管如此,本综述强调了使用TVUS进行早期准确诊断的重要性,并在必要时辅以MRI,随后迅速进行手术管理以优化孕产妇安全。提高对这种罕见表现的认识、报告和记录对于指导未来临床决策和预防灾难性并发症至关重要。本综述未获得外部资金。由于其回顾性、描述性性质,本综述未在任何数据库中注册。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c61a/12442142/3029e7043758/cureus-0017-00000090378-i01.jpg

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