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骨发育异常系列。软骨发育不全、低软骨发育不全和致死性骨发育不全:综述与更新。

Bone dysplasia series. Achondroplasia, hypochondroplasia and thanatophoric dysplasia: review and update.

作者信息

Lemyre E, Azouz E M, Teebi A S, Glanc P, Chen M F

机构信息

Department of Medical Genetics, Ste. Justine Hospital, Montreal, Que.

出版信息

Can Assoc Radiol J. 1999 Jun;50(3):185-97.

Abstract

The authors summarize the clinical, genetic and histopathologic features, as well as the complications, and radiological diagnosis of 3 related generalized short-limb skeletal dysplasias: achondroplasia, hypochondroplasia and thanatophoric dysplasia. In all of these dysplasias, there is abnormal endochondral ossification, but periosteal ossification is not affected. These 3 relatively common entities are known to be allelic to the same gene: the fibroblast growth factor receptor 3 gene on chromosome 4p. Heterozygous achondroplasia is the most common nonlethal skeletal dysplasia. The distinctive clinical and radiological features allow a precise diagnosis, as there is little variability in the appearance of affected patients. There is also a very evident molecular homogeneity. On histopathology of the growth plate, there is a quantitative decrease in endochondral ossification. Precise prenatal ultrasonographic diagnosis is possible in the third trimester, and sometimes even in the second. Hypochondroplasia is a relatively common, milder form of achondroplasia, which varies within and between families and lacks the neurological complications often seen in achondroplasia of this group. An accurate prenatal ultrasonographic diagnosis is rare. There are milder changes on histology of the growth plate. Thanatophoric dysplasia is the lethal and most severe dysplasia. It has distinct features--mainly short tubular bones and short ribs with platyspondyly--allowing a precise radiologic and prenatal ultrasonographic diagnosis. On histopathology of the growth plate, there is disruption of endochondral ossification.

摘要

作者总结了三种相关的全身性短肢骨骼发育异常的临床、遗传和组织病理学特征,以及并发症和放射学诊断,这三种发育异常分别为:软骨发育不全、低软骨发育不全和致死性骨发育不全。在所有这些发育异常中,软骨内骨化异常,但骨膜骨化未受影响。已知这三种相对常见的病症是同一基因的等位基因:位于4号染色体短臂上的成纤维细胞生长因子受体3基因。杂合性软骨发育不全是最常见的非致死性骨骼发育异常。其独特的临床和放射学特征有助于精确诊断,因为受累患者的表现差异很小。分子同质性也非常明显。在生长板的组织病理学上,软骨内骨化数量减少。在孕晚期甚至有时在孕中期就可以进行精确的产前超声诊断。低软骨发育不全是软骨发育不全的一种相对常见、症状较轻的形式,在家族内部和家族之间存在差异,且没有该组软骨发育不全常见的神经并发症。准确的产前超声诊断很少见。生长板组织学上的变化较轻微。致死性骨发育不全是致死性且最严重的发育异常。它具有明显的特征——主要是短管状骨和短肋骨以及扁平椎——有助于进行精确的放射学和产前超声诊断。在生长板的组织病理学上,软骨内骨化遭到破坏。

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