von Elten Kelley, Sawyer Taylor, Lentz-Kapua Sarah, Kanis Adam, Studer Matthew
Department of Pediatrics, MCHK-PE, Tripler Army Medical Center, 1 Jarrett White Road, Honolulu, HI 96859, USA.
Pediatr Cardiol. 2013 Jun;34(5):1244-6. doi: 10.1007/s00246-012-0367-8. Epub 2012 May 26.
Wolf-Hirschhorn Syndrome (WHS) is a genetic syndrome that includes a typical facial appearance, mental retardation, growth delay, seizures, and congenital cardiac defects. A deletion of the terminal band of the short arm of chromosome 4, with a breakpoint at the 4p15 to 4p16 region, is the most common genetic mutation causing WHS. Congenital heart disease associated with WHS typically includes atrial and ventricular septal defects, though there are a few case reports of associated complex congenital heart disease. Here we report a case of an infant with a large 4p deletion, with a breakpoint at the 4p12 region, and hypoplasic left heart syndrome. We discuss a possible link between the size of the chromosomal deletion in WHS and the severity of the cardiac defect.
沃尔夫-赫希霍恩综合征(WHS)是一种遗传性综合征,其特征包括典型的面部外观、智力发育迟缓、生长发育延迟、癫痫发作和先天性心脏缺陷。4号染色体短臂末端带的缺失,断点位于4p15至4p16区域,是导致WHS最常见的基因突变。与WHS相关的先天性心脏病通常包括房间隔和室间隔缺损,不过也有一些关于相关复杂性先天性心脏病的病例报告。在此,我们报告一例患有大片段4p缺失(断点位于4p12区域)并伴有左心发育不全综合征的婴儿病例。我们讨论了WHS中染色体缺失大小与心脏缺陷严重程度之间可能存在的联系。