McMahon Colin J, Breathnach Colm, Betts David R, Sharkey Freddie H, Greally Marie T
Department of Pediatric Cardiology, Our Lady's Children's Hospital, Crumlin, Dublin, Ireland.
Am J Med Genet A. 2015 May;167A(5):1134-41. doi: 10.1002/ajmg.a.36978. Epub 2015 Mar 21.
We describe a 6-year-old male, diagnosed at birth with double outlet right ventricle (DORV), anterior aorta, multiple ventricular septal defects, pulmonary stenosis, microcephaly and mildly dysmorphic craniofacial findings. Chromosomal analysis showed a normal male karyotype but on subsequent array comparative genomic hybridization (array CGH) analysis a de novo 2.5 Mb loss in chromosome 13q at 13q33.3q34, together with an inherited gain at 4p12, were detected. The propositus underwent placement of a Blalock Taussig shunt and subsequently a Glenn and Fontan operation was performed. In this report we propose that COL4A1 and COL4A2 may be candidate genes for congenital heart disease (CHD) in individuals with a deletion in 13q within the 6Mb critical region for cardiac development proposed by Huang et al., [2012].
我们描述了一名6岁男性,出生时被诊断为右心室双出口(DORV)、主动脉在前、多个室间隔缺损、肺动脉狭窄、小头畸形以及轻度颅面畸形。染色体分析显示为正常男性核型,但随后的阵列比较基因组杂交(array CGH)分析检测到13号染色体q33.3q34区域有一个2.5 Mb的新生缺失,同时在4p12区域有一个遗传获得。先证者接受了布莱洛克-陶西格分流术,随后进行了格林手术和方坦手术。在本报告中,我们提出,对于Huang等人[2012年]提出的心脏发育关键区域6Mb内13q缺失的个体,COL4A1和COL4A2可能是先天性心脏病(CHD)的候选基因。