Sanlaville Damien, Verloes Alain
INSERM U781, Genetic Department, AP-HP Necker-Enfants Malades Hospital, Paris, France.
Eur J Hum Genet. 2007 Apr;15(4):389-99. doi: 10.1038/sj.ejhg.5201778. Epub 2007 Feb 14.
CHARGE syndrome is a rare, usually sporadic autosomal dominant disorder due in 2/3 of cases to mutations within the CHD7 gene. The clinical definition has evolved with time. The 3C triad (Coloboma-Choanal atresia-abnormal semicircular Canals), arhinencephaly and rhombencephalic dysfunctions are now considered the most important and constant clues to the diagnosis. We will discuss here recent aspects of the phenotypic delineation of CHARGE syndrome and highlight the role of CHD7 in its pathogeny. We review available data on its molecular pathology as well as cytogenetic and molecular evidences for genetic heterogeneity within CHARGE syndrome.
CHARGE综合征是一种罕见的、通常为散发性的常染色体显性疾病,2/3的病例是由CHD7基因突变引起的。其临床定义随时间而演变。目前,3C三联征(眼部缺损-后鼻孔闭锁-异常半规管)、无脑回畸形和菱脑功能障碍被认为是诊断该病最重要且恒定的线索。在此,我们将讨论CHARGE综合征表型描述的最新进展,并强调CHD7在其发病机制中的作用。我们回顾了有关其分子病理学的现有数据,以及CHARGE综合征内基因异质性的细胞遗传学和分子证据。