Oude Luttikhuis M E, Williams D K, Trembath R C
Department of Genetics, University of Leicester, UK.
J Med Genet. 1996 Oct;33(10):873-6. doi: 10.1136/jmg.33.10.873.
Type E brachydactyly is a digital malformation which characteristically causes an asymmetrical shortening of one or more metacarpals or metatarsals or both. Although commonly seen as part of a syndrome, it can be inherited as an autosomal dominant characteristic, the gene acting with variable expressivity, but complete penetrance. As an Albright hereditary osteodystrophy (AHO)-like syndrome including brachydactyly type E and mental retardation may be caused by (micro) deletions at chromosome 2q37, this region together with the AHO locus at chromosome 20q13 were considered as candidate loci for brachydactyly type E. In this paper we described a family with isolated autosomal dominant type E brachydactyly in whom molecular analysis excludes linkage to these regions, providing support for further genetic heterogeneity of this trait.
E型短指畸形是一种手指畸形,其特征是导致一根或多根掌骨或跖骨或两者不对称缩短。虽然它通常被视为综合征的一部分,但它可以作为常染色体显性特征遗传,该基因具有可变的表达,但完全显性。由于包括E型短指畸形和智力迟钝在内的一种类似奥尔布赖特遗传性骨营养不良(AHO)的综合征可能由2q37染色体上的(微)缺失引起,该区域以及20q13染色体上的AHO位点被认为是E型短指畸形的候选位点。在本文中,我们描述了一个孤立的常染色体显性E型短指畸形家族,其中分子分析排除了与这些区域的连锁关系,为该性状的进一步遗传异质性提供了支持。