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并指畸形:表型、遗传学及现行分类。

Syndactyly: phenotypes, genetics and current classification.

机构信息

Human Genetics Program, Department of Animal Sciences, Quaid-i-Azam University Islamabad, Islamabad, Pakistan.

出版信息

Eur J Hum Genet. 2012 Aug;20(8):817-24. doi: 10.1038/ejhg.2012.14. Epub 2012 Feb 15.

Abstract

Syndactyly is one of the most common hereditary limb malformations depicting the fusion of certain fingers and/or toes. It may occur as an isolated entity or a component of more than 300 syndromic anomalies. Syndactylies exhibit great inter- and intra-familial clinical variability. Even within a subject, phenotype can be unilateral or bilateral and symmetrical or asymmetrical. At least nine non-syndromic syndactylies with additional sub-types have been characterized. Most of the syndactyly types are inherited as autosomal dominant but two autosomal recessive and an X-linked recessive entity have also been described. Whereas the underlying genes/mutations for types II-1, III, IV, V, and VII have been worked out, the etiology and molecular basis of the other syndactyly types remain unknown. In this communication, based on an overview of well-characterized isolated syndactylies, their cardinal phenotypes, inheritance patterns, and clinical and genetic heterogeneities, a 'current classification scheme' is presented. Despite considerable progress in the understanding of syndactyly at clinical and molecular levels, fundamental questions regarding the disturbed developmental mechanisms leading to fused digits, remain to be answered.

摘要

并指畸形是最常见的遗传性肢体畸形之一,表现为某些手指和/或脚趾融合。它可以作为一个孤立的实体或超过 300 种综合征异常的一个组成部分出现。并指畸形表现出很大的个体间和个体内临床变异性。即使在一个个体中,表型也可以是单侧或双侧、对称或不对称的。至少有 9 种非综合征性并指畸形具有额外的亚型特征。大多数并指畸形是常染色体显性遗传的,但也有两种常染色体隐性和一种 X 连锁隐性遗传的实体已被描述。虽然已经确定了 II-1、III、IV、V 和 VII 型的相关基因/突变,但其他并指畸形的病因和分子基础仍然未知。在本通讯中,基于对特征明确的孤立性并指畸形的概述,其主要表型、遗传模式以及临床和遗传异质性,提出了一个“当前分类方案”。尽管在临床和分子水平上对并指畸形的理解取得了相当大的进展,但关于导致融合指的发育机制紊乱的基本问题仍有待回答。

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