Malakan Rad Elaheh, Momtazmanesh Sara
Children's Medical Center (Pediatric Center of Excellence), Department of Pediatric Cardiology, Tehran, Iran.
Department of Pediatric Cardiology, Tehran University of Medical Sciences, Tehran, Iran.
Ann Pediatr Cardiol. 2021 Jan-Mar;14(1):125-127. doi: 10.4103/apc.APC_215_19. Epub 2020 Dec 1.
We report a 6-month-old female infant with deletion of chromosome 22q11.2 (DiGeorge/VFS TUPLE 1), normal atrial arrangement with concordant atrioventricular connection, pulmonary atresia, large subaortic ventricular septal defect, diminutive native pulmonary arteries, a characteristic weird-shape right aortic arch with arch-on-arch appearance and figure of 8 configuration. We presented the cardiac computed tomographic angiographic and cardiac angiographic features. Using Autodesk 3ds Max 2018 software, we explained and illustrated the speculative embryologic etiology of this bizarre aortic archanomaly with the extensive abnormal remodeling of the left brachiocephalic artery, based on a "five-embryonic aortic arches" concept. As to the best of the authors' knowledge, this is the first report of a genetically confirmed case of DiGeorge syndrome and an exceedingly rare type of right aortic arch anomaly with embryologic explanation according to the "five-embryonic-aortic-arches" concept. It seems that the constellation of pulmonary atresia, bizarreshaped right aortic arch due to abnormal development of the aortic sac, and abnormal remodeling of the left brachiocephalic artery may be strongly suggestive of DiGeorge syndrome.
我们报告一例6个月大的女婴,其22q11.2染色体缺失(迪乔治综合征/室间隔缺损、面部畸形、胸腺发育不全、腭裂及心血管畸形综合征1型),心房排列正常,房室连接一致,肺动脉闭锁,巨大的主动脉下室间隔缺损,细小的原生肺动脉,具有特征性怪异形状的右主动脉弓,呈弓上弓外观及8字形结构。我们展示了心脏计算机断层血管造影和心脏血管造影特征。使用Autodesk 3ds Max 2018软件,基于“五个胚胎主动脉弓”的概念,我们解释并说明了这种奇异主动脉弓异常的推测性胚胎学病因,其与左头臂动脉的广泛异常重塑有关。据作者所知,这是首例经基因确诊的迪乔治综合征病例报告,也是根据“五个胚胎主动脉弓”概念对一种极其罕见的右主动脉弓异常进行胚胎学解释的报告。肺动脉闭锁、因主动脉囊异常发育导致的奇异形状右主动脉弓以及左头臂动脉的异常重塑这一组症状似乎强烈提示为迪乔治综合征。