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Ebstein 畸形合并右位主动脉弓患者的 CHARGE 综合征。

Ebstein Anomaly and Right Aortic Arch in Patient with Charge Syndrome.

机构信息

Department of Pediatrics, Riga Stradins University, LV-1007 Riga, Latvia.

Clinic for Pediatric Cardiology and Cardiac Surgery, Children's Clinical University Hospital, LV-1004 Riga, Latvia.

出版信息

Medicina (Kaunas). 2021 Nov 13;57(11):1239. doi: 10.3390/medicina57111239.

Abstract

Ebstein anomaly is a rare congenital heart disease characterized by a varying degree of anatomical and functional abnormalities of tricuspid valve and right ventricle. It often coexists with other congenital cardiac malformations. Up to 79-89% of patients with Ebstein anomaly have interatrial communication in the form of patent oval foramen or atrial septal defect and more than one-third has other types of cardiac malformations. Association between Ebstein anomaly and right aortic arch is extremely rare and only few cases have been described in the literature so far. Much rarer than with other cardiac malformations, Ebstein anomaly is associated with non-cardiac malformations or genetic syndromes. Several cases of association between Ebstein anomaly and Charge syndrome have been reported, nevertheless, Ebstein anomaly accounts for less than 1% of cardiac defects seen in patients with Charge syndrome. In this case report, we present a unique case of a patient with Charge syndrome where both Ebstein anomaly and right aortic arch are present. The diagnosis of Ebstein anomaly and right aortic arch was established prenatally. In the first years of life, the patient did not exhibit any remarkable symptoms. However, over time, deterioration of right ventricle function and increased tricuspid regurgitation were observed, requiring consideration of surgical treatment at the age of five. In addition, delay in physical, motor, and mental development was observed and thus, at the age of five, the patient was consulted by a medical geneticist and a gene panel to test for structural heart defects was ordered. The test showed a mutation in chromodomain helicase DNA binding protein 7 (CHD7) gene, which, along with clinical features, allowed to establish a diagnosis of Charge syndrome. To the best of the authors' knowledge, this is the first case report of a patient with Charge syndrome, Ebstein anomaly, and right aortic arch that has been described in the literature.

摘要

埃布斯坦畸形是一种罕见的先天性心脏病,其特征为三尖瓣和右心室的解剖和功能异常程度不一。它常与其他先天性心脏畸形并存。多达 79-89%的埃布斯坦畸形患者存在房间隔沟通,表现为卵圆孔未闭或房间隔缺损,超过三分之一的患者存在其他类型的心脏畸形。埃布斯坦畸形与右位主动脉弓的关联极为罕见,迄今为止文献中仅描述了少数几例。埃布斯坦畸形比其他心脏畸形更为罕见,与非心脏畸形或遗传综合征相关。已有数例埃布斯坦畸形与 CHARGE 综合征相关的报道,然而,埃布斯坦畸形在 CHARGE 综合征患者中占心脏缺陷的比例不到 1%。在本病例报告中,我们介绍了一例 CHARGE 综合征患者,同时存在埃布斯坦畸形和右位主动脉弓。埃布斯坦畸形和右位主动脉弓的诊断均在产前确立。在生命的最初几年,患者未表现出任何明显的症状。然而,随着时间的推移,观察到右心室功能恶化和三尖瓣反流增加,需要在 5 岁时考虑手术治疗。此外,还观察到身体、运动和智力发育迟缓,因此,患者在 5 岁时由医学遗传学家咨询,并进行了基因检测以排除结构性心脏缺陷。检测结果显示染色质解旋酶 DNA 结合蛋白 7(CHD7)基因突变,结合临床特征,可诊断为 CHARGE 综合征。据作者所知,这是文献中首例描述的 CHARGE 综合征、埃布斯坦畸形和右位主动脉弓患者。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a09b/8619708/b67a9793bfac/medicina-57-01239-g001.jpg

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