Department of Genetics, University Medical Centre Groningen, Groningen, The Netherlands.
Heart. 2011 Aug;97(15):1228-32. doi: 10.1136/hrt.2010.211433. Epub 2011 Feb 22.
To determine whether offering cardiac screening to relatives of patients with left ventricular outflow tract obstructions (LVOTOs) would be justified.
LVOTOs have been recognised as a group of congenital heart diseases with 'high heritability'. One of the LVOTOs, the bicuspid aortic valve, is often asymptomatic, but has become known to be associated with sudden, unexpected cardiac death. However, the need for cardiac screening of first-degree relatives of patients with LVOTO has not been determined owing to the lack of studies in well-defined cohorts of consecutive patients.
The families of a cohort of 249 consecutive paediatric patients with LVOTO were offered genetic counselling. Of 182 consenting index patients, 40 patients (22%) appeared to have associated non-cardiac congenital anomalies (LVOTO-NCA). In the other 142 patients with LVOTO, cardiac screening of 449 first-degree relatives was performed.
Cardiac screening disclosed a cardiac anomaly in 34 first-degree relatives (8%). In 23 (68%) of these the cardiac anomaly was a bicuspid aortic valve. Twenty-four of these anomalies were newly detected by our screening programme (71%). These 34 cardiac anomalies were found in the families of 28 index cases (20%).
This study shows that of the patients with LVOTO without NCA, 20% had (an) affected first-degree relative(s), frequently with undetected bicuspid aortic valves. These data suggest that cardiac screening of relatives of patients with LVOTO without NCA is justified. This may help prevent sudden, unexpected, cardiac death or life-threatening complications in relatives with undetected bicuspid aortic valves.
确定是否有理由为左心室流出道梗阻(LVOTO)患者的亲属提供心脏筛查。
LVOTO 已被认为是一组具有“高遗传性”的先天性心脏病。其中一种 LVOTO,即二叶式主动脉瓣,通常无症状,但已被认为与突发、意外的心脏性猝死有关。然而,由于缺乏对 LVOTO 连续患者队列的研究,尚未确定 LVOTO 患者一级亲属进行心脏筛查的必要性。
对一组 249 例连续 LVOTO 患儿的家族进行了遗传咨询。在 182 名同意的索引患者中,40 名(22%)患者似乎存在相关的非心脏先天性异常(LVOTO-NCA)。在另外 142 例 LVOTO 患者中,对 449 名一级亲属进行了心脏筛查。
心脏筛查发现 34 名一级亲属存在心脏异常(8%)。其中 23 例(68%)的心脏异常为二叶式主动脉瓣。这些异常中有 24 例是通过我们的筛查计划新发现的(71%)。这些 34 个心脏异常存在于 28 个索引病例(20%)的家族中。
本研究表明,在无 NCA 的 LVOTO 患者中,20%的一级亲属存在(一个或多个)受影响的亲属,这些亲属常伴有未被发现的二叶式主动脉瓣。这些数据表明,对无 NCA 的 LVOTO 患者亲属进行心脏筛查是合理的。这可能有助于预防未被发现的二叶式主动脉瓣患者亲属发生突发、意外的心脏性猝死或危及生命的并发症。