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维厄桑斯动脉环形成伴有巨大动脉瘤样扩张的冠状动脉-肺动脉瘘:一例报告

Vieussens' arterial ring forming a coronary-pulmonary artery fistula with large aneurysmal dilatation: a case report.

作者信息

Moriyama Shuji, Nakata Kosuke, Doi Hideki, Matsumura Toshiyuki, Fukui Toshihiro

机构信息

Department of Cardiovascular Surgery, Kumamoto Rosai Hospital, Yatsushiro, Kumamoto, Japan.

Department of Cardiovascular Medicine, Kumamoto Rosai Hospital, Yatsushiro, Kumamoto, Japan.

出版信息

Eur Heart J Case Rep. 2024 Sep 14;8(10):ytae507. doi: 10.1093/ehjcr/ytae507. eCollection 2024 Oct.

Abstract

BACKGROUND

Vieussens' arterial ring (VAR) is an embryonic conotruncal ring remnant that represents a rare vascular anomaly involving a connection between the right coronary artery (RCA) and the left anterior descending artery (LAD). We describe a unique case of VAR associated with large aneurysmal dilatation, which presents as the formation of a fistula between the coronary artery and pulmonary artery.

CASE SUMMARY

An 80-year-old Japanese woman presented with an asymptomatic mediastinal mass that was incidentally detected on computed tomography. Subsequent imaging over 2 years revealed the progression of the two masses connecting the RCA to the LAD measuring 8 × 7 mm and 28 × 21 mm in diameter. Transthoracic echocardiography identified a cystic lesion anterior to the right ventricular outflow tract, and colour Doppler imaging confirmed flow into the pulmonary artery. Furthermore, coronary angiography revealed a large aneurysm arising from the LAD, with an efferent vessel communicating with the pulmonary artery. Surgical intervention involved resection of the aneurysms and closure of the coronary-pulmonary artery fistula, which yielded favourable postoperative outcomes.

DISCUSSION

Vieussens' arterial ring is a rare but clinically significant anomaly with varied presentations; the incidence of VAR remains uncertain. In this case, surgical resection and closure of the fistula were performed to mitigate the risk of rupture and to address the potential haemodynamic consequences. Understanding and documenting such cases will contribute to refining treatment approaches and improving patient care in cardiovascular medicine.

摘要

背景

维厄桑斯动脉环(VAR)是胚胎期圆锥动脉干环的残余部分,是一种罕见的血管异常,表现为右冠状动脉(RCA)与左前降支动脉(LAD)之间存在连接。我们描述了一例与巨大动脉瘤样扩张相关的独特VAR病例,其表现为冠状动脉与肺动脉之间形成瘘管。

病例摘要

一名80岁日本女性因计算机断层扫描偶然发现无症状纵隔肿块。随后2年的影像学检查显示,连接RCA与LAD的两个肿块逐渐增大,直径分别为8×7 mm和28×21 mm。经胸超声心动图在右心室流出道前方发现一个囊性病变,彩色多普勒成像证实有血流进入肺动脉。此外,冠状动脉造影显示LAD处有一个大动脉瘤,有一条传出血管与肺动脉相通。手术干预包括切除动脉瘤和闭合冠状动脉-肺动脉瘘,术后效果良好。

讨论

维厄桑斯动脉环是一种罕见但具有临床意义的异常,表现多样;VAR的发病率尚不确定。在本病例中,进行了手术切除和瘘管闭合,以降低破裂风险并解决潜在的血流动力学后果。了解并记录此类病例将有助于完善心血管医学的治疗方法并改善患者护理。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/15c4/11450478/0a1af888665f/ytae507il2.jpg

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