Moerman P, Dumoulin M, Lauweryns J, Van der Hauwaert L G
Department of Pathology, Gasthuisberg University Hospital, Leuven, Belgium.
Br Heart J. 1987 Sep;58(3):274-8. doi: 10.1136/hrt.58.3.274.
The clinical and necropsy findings in four cases of interrupted right aortic arch and right descending aorta associated with DiGeorge syndrome (congenital absence or hypoplasia of the thymus and parathyroids) are described. All patients had a mirror image of type B interruption, namely a right aortic arch with reversed branching pattern and an interruption between the right common carotid and right subclavian artery. In two patients there was a doubly committed subarterial ventricular septal defect and in the two other patients there was a perimembranous septal defect. Three patients had a bicuspid aortic valve. In a consecutive series of 185 necropsies in infants and children with congenital heart disease there were no cases of interrupted right aortic arch that were not associated with DiGeorge syndrome. These observations and previous reports indicate that the concurrence of these two rare conditions is more than fortuitous. In patients with an interrupted aortic arch the clinician should be aware of the common association with DiGeorge syndrome. If the interruption is associated with a right-sided descending aorta it is highly probable that the patient has DiGeorge syndrome.
本文描述了4例右位主动脉弓中断并右位降主动脉与迪格奥尔格综合征(胸腺和甲状旁腺先天性缺失或发育不全)相关的临床及尸检结果。所有患者均为B型中断的镜像表现,即右位主动脉弓伴分支模式反转,且右颈总动脉与右锁骨下动脉之间中断。2例患者存在双动脉下室间隔缺损,另外2例患者存在膜周部室间隔缺损。3例患者有二叶式主动脉瓣。在一系列连续的185例先天性心脏病婴幼儿尸检中,未发现不伴有迪格奥尔格综合征的右位主动脉弓中断病例。这些观察结果及既往报道表明,这两种罕见病症的并发并非偶然。对于主动脉弓中断的患者,临床医生应意识到其常与迪格奥尔格综合征相关。如果中断与右侧降主动脉相关,则患者极有可能患有迪格奥尔格综合征。