John Anitha S, McDonald-McGinn Donna M, Zackai Elaine H, Goldmuntz Elizabeth
Division of Cardiology, The Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine, Philadelphia, Pennsylvania 19104, USA.
Am J Med Genet A. 2009 May;149A(5):939-42. doi: 10.1002/ajmg.a.32770.
The 22q11.2 deletion syndrome is characterized by a highly variable phenotype including a range of cardiac malformations. The most common cardiovascular features include a subset of conotruncal defects, perimembranous ventricular septal defects and aortic arch anomalies. This report describes a series of patients with 22q11.2 deletion syndrome with the novel cardiac finding of mild aortic root dilation. A chart review was performed on 93 patients with documented 22q11.2 deletion without significant congenital heart disease to determine the number of patients with aortic root dilation. Patients ranged in age from 1 to 13 years of age. Of these 93 patients, 10 patients were found to have aortic root dilation on a screening echocardiogram. Seven of these patients did not have any additional risk factors while three patients had a bicuspid aortic valve (BAV). Four of 10 patients had additional minor cardiac anomalies including repaired ventricular septal defect (1), patent ductus arteriosus(1), arch anomalies (1), and left pulmonary artery stenosis (1). Three patients had isolated cases of aortic root dilation. Interestingly, several of these patients did not have aortic root dilation on their initial echocardiograms. The purpose of this study is to draw attention to a novel cardiac finding in patients with 22q11.2 deletion that may be of clinical importance. Further long-term study is warranted to assess the need for echocardiographic screening in the 22q11.2 deleted population for aortic root dilation into adolescence and adulthood.
22q11.2缺失综合征的特征是具有高度可变的表型,包括一系列心脏畸形。最常见的心血管特征包括一组圆锥干畸形、膜周部室间隔缺损和主动脉弓异常。本报告描述了一系列22q11.2缺失综合征患者,他们有轻度主动脉根部扩张这一新颖的心脏表现。对93例记录有22q11.2缺失且无明显先天性心脏病的患者进行了病历回顾,以确定主动脉根部扩张的患者数量。患者年龄在1至13岁之间。在这93例患者中,10例在筛查超声心动图时发现有主动脉根部扩张。其中7例患者没有任何其他风险因素,而3例患者有二叶式主动脉瓣(BAV)。10例患者中有4例还有其他轻微心脏异常,包括室间隔缺损修补术后(1例)、动脉导管未闭(1例)、弓部异常(1例)和左肺动脉狭窄(1例)。3例患者为孤立性主动脉根部扩张病例。有趣的是,这些患者中有几例在初次超声心动图检查时没有主动脉根部扩张。本研究的目的是提醒人们注意22q11.2缺失患者中一种可能具有临床重要性的新颖心脏表现。有必要进行进一步的长期研究,以评估在22q11.2缺失人群中对主动脉根部扩张进行超声心动图筛查直至青春期和成年期的必要性。