Department of Clinical Genetics, Academic Medical Center, Amsterdam, The Netherlands.
Am J Med Genet C Semin Med Genet. 2013 Aug;163C(3):178-84. doi: 10.1002/ajmg.c.31365. Epub 2013 Jun 21.
Left ventricular noncompaction (LVNC) is a relatively common genetic cardiomyopathy, characterized by prominent trabeculations with deep intertrabecular recesses in mainly the left ventricle. Although LVNC often occurs in an isolated entity, it may also be present in various types of congenital heart disease (CHD). The most prevalent CHD in LVNC is Ebstein anomaly, which is a rare form of CHD characterized by apical displacement and partial fusion of the septal and posterior leaflet of the tricuspid valve with the ventricular septum. Several reports of sporadic as well as familial cases of Ebstein anomaly associated with LVNC have been reported. Recent studies identified mutations in the MYH7 gene, encoding the sarcomeric β-myosin heavy chain protein, in patients harboring this specific phenotype. Here, we will review the association between Ebstein anomaly, LVNC and mutations in MYH7, which seems to represent a subtype of Ebstein anomaly with autosomal dominant inheritance and variable penetrance.
左心室心肌致密化不全(LVNC)是一种较为常见的遗传性心肌病,其特征为左心室主要部位存在明显的心肌小梁,小梁间深陷隐窝。虽然 LVNC 常以孤立性病变存在,但也可能存在于各种先天性心脏病(CHD)中。LVNC 中最常见的 CHD 是Ebstein 畸形,这是一种罕见的 CHD,其特征为三尖瓣隔瓣和后瓣的瓣尖部分或全部脱入右心室,与室间隔融合。已有散发性和家族性 Ebstein 畸形伴 LVNC 的病例报道。最近的研究发现,携带这种特定表型的患者中存在 MYH7 基因突变,该基因编码肌球蛋白重链蛋白的肌小节β亚单位。在此,我们将回顾 Ebstein 畸形、LVNC 和 MYH7 突变之间的关系,这似乎代表了一种具有常染色体显性遗传和可变外显率的 Ebstein 畸形亚型。