Molecular (Epi)Genetics Laboratory, Hospital Universitario Araba-Txagorritxu, BioAraba, Vitoria-Gasteiz 01009, Spain.
Orphanet J Rare Dis. 2013 Sep 12;8:141. doi: 10.1186/1750-1172-8-141.
Brachydactyly (BD) refers to the shortening of the hands, feet or both. There are different types of BD; among them, type E (BDE) is a rare type that can present as an isolated feature or as part of more complex syndromes, such as: pseudohypopthyroidism (PHP), hypertension with BD or Bilginturan BD (HTNB), BD with mental retardation (BDMR) or BDE with short stature, PTHLH type. Each syndrome has characteristic patterns of skeletal involvement. However, brachydactyly is not a constant feature and shows a high degree of phenotypic variability. In addition, there are other syndromes that can be misdiagnosed as brachydactyly type E, some of which will also be discussed. The objective of this review is to describe some of the syndromes in which BDE is present, focusing on clinical, biochemical and genetic characteristics as features of differential diagnoses, with the aim of establishing an algorithm for their differential diagnosis. As in our experience many of these patients are recruited at Endocrinology and/or Pediatric Endocrinology Services due to their short stature, we have focused the algorithm in those steps that could mainly help these professionals.
短指(BD)是指手、脚或两者的缩短。BD 有不同的类型;其中,E 型(BDE)是一种罕见的类型,可表现为孤立的特征或更复杂的综合征的一部分,如:假性垂体机能减退症(PHP)、BD 合并高血压或 Bilginturan BD(HTNB)、BD 合并智力障碍(BDMR)或 BDE 合并身材矮小、PTHLH 型。每种综合征都有其特征性的骨骼受累模式。然而,短指并不是一个恒定的特征,表现出高度的表型变异性。此外,还有其他一些综合征可能被误诊为 E 型短指,其中一些也将在本文中讨论。本综述的目的是描述一些存在 BDE 的综合征,重点介绍临床、生化和遗传特征作为鉴别诊断的特征,旨在为其鉴别诊断建立一个算法。根据我们的经验,由于身材矮小,许多这些患者在内分泌科和/或儿科内分泌科就诊,因此我们将算法集中在这些步骤上,这些步骤主要可以帮助这些专业人员。