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科默雷尔憩室:一名16岁女孩的右位主动脉弓伴左锁骨下动脉异常起源及右椎动脉重复畸形

Kommerell Diverticulum: Right Aortic Arch with Anomalous Origin of Left Subclavian Artery and Duplicity of Right Vertebral Artery in a 16-Year-Old Girl.

作者信息

Silveira João V, Junqueira Flavia P, Silveira Cylmara G, Consolim-Colombo Fernanda M

机构信息

Departmet of Medicine, Universidade Nove de Julho, São Paulo, SP, Brazil.

Departmet of Radiology, Delboni Auriemo, DASA/SP, São Paulo, SP, Brazil.

出版信息

Am J Case Rep. 2019 Feb 21;20:228-232. doi: 10.12659/AJCR.912053.

DOI:10.12659/AJCR.912053
PMID:30787266
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC6394145/
Abstract

BACKGROUND Kommerell diverticulum with a right-sided aortic arch is a rare congenital anatomical condition most often observed in adults. A possible etiology of the subclavian artery's anomalous origin would be an abnormality in regression of the fourth primitive aortic arch during embryonic development. CASE REPORT We report on the case of a 16-year-old female patient presenting with complaints of occasional tachycardia and mild non-specific dyspnea after anxiety crises. Physical examination revealed lower amplitude of the pulses in the left upper limb compared to the right upper limb, and difference in blood pressure (BP) values of 80×60 mmHg, and 100×60 mmHg, respectively. Different radiological imaging modalities were performed to elucidate a possible vascular abnormality. Multislice detector computed tomography angiography of the thoracic aorta and supra-aortic trunks showed a right-sided aortic arch and an aberrant origin of the left subclavian artery with a retroesophageal course and dilation of its emergence (Kommerell diverticulum), as well as duplicity of the right vertebral artery (RVA). Considering the actual small diameter of the diverticulum and the absence of dysphagia or severe external esophageal compression analyzed by the esophagogram, vascular surgery was not indicated. Since complications have been described in the literature, the patient must be kept under observation in the future. CONCLUSIONS Congenital vascular alterations, including Kommerell diverticulum with right-sided aortic arch and the aberrant origin of the left subclavian artery, should be suspected in otherwise asymptomatic young patients with few clinical manifestations. Investigation with different imaging methods helps to clarify the vascular abnormalities, to support a possible surgical procedure indication, and to monitor the patients in follow-up.

摘要

背景

伴有右侧主动脉弓的Kommerell憩室是一种罕见的先天性解剖异常,多见于成年人。锁骨下动脉异常起源的一个可能病因是胚胎发育过程中第四原始主动脉弓退化异常。

病例报告

我们报告一例16岁女性患者,她在焦虑发作后偶尔出现心动过速和轻度非特异性呼吸困难。体格检查发现,与右上肢相比,左上肢脉搏幅度较低,血压值分别为80×60 mmHg和100×60 mmHg。为明确可能的血管异常,进行了不同的放射学成像检查。胸部主动脉和主动脉弓上干的多层螺旋CT血管造影显示右侧主动脉弓、左锁骨下动脉异常起源,走行于食管后方且起始部扩张(Kommerell憩室),以及右侧椎动脉重复畸形。考虑到憩室实际直径较小,且食管造影分析未发现吞咽困难或严重的食管外部压迫,未建议进行血管手术。鉴于文献中已描述有并发症,该患者未来必须接受观察。

结论

对于临床表现较少的无症状年轻患者,应怀疑存在先天性血管改变,包括伴有右侧主动脉弓的Kommerell憩室和左锁骨下动脉异常起源。采用不同的成像方法进行检查有助于明确血管异常,支持可能的手术指征,并在随访中对患者进行监测。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/1231e72eb817/amjcaserep-20-228-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/70d63cabf269/amjcaserep-20-228-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/92e16ef4ec6a/amjcaserep-20-228-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/bf75c416481a/amjcaserep-20-228-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/4e110bffe87f/amjcaserep-20-228-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/1231e72eb817/amjcaserep-20-228-g005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/70d63cabf269/amjcaserep-20-228-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/92e16ef4ec6a/amjcaserep-20-228-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/bf75c416481a/amjcaserep-20-228-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/4e110bffe87f/amjcaserep-20-228-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8264/6394145/1231e72eb817/amjcaserep-20-228-g005.jpg

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A review of the surgical management of right-sided aortic arch aneurysms.右侧主动脉弓动脉瘤手术治疗综述
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Reconstruction of the left-sided brachiocephalic trunk after vascular ring operation in left-handed child with Kommerell's diverticulum.患有Kommerell憩室的左利手儿童血管环手术后左侧头臂干的重建。
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A rare anomalous origin of left subclavian artery from the circle of Willis in combination with right sided aortic arch: A case report.一例罕见的左侧锁骨下动脉起源于 Willis 环并合并右侧主动脉弓的病例报告。
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