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假性软骨发育不全:不同年龄的临床诊断及常染色体显性与隐性类型的比较。32例患者(26个家族)的回顾分析

Pseudoachondroplasia: clinical diagnosis at different ages and comparison of autosomal dominant and recessive types. A review of 32 patients (26 kindreds).

作者信息

Wynne-Davies R, Hall C M, Young I D

出版信息

J Med Genet. 1986 Oct;23(5):425-34. doi: 10.1136/jmg.23.5.425.

Abstract

This survey reviews the diagnosis (predominantly radiological) of 32 cases of pseudoachondroplasia from 26 kindreds and illustrates the natural history and varying appearance of the disordered bone growth from infancy to adult life. In addition, an attempt has been made to detect phenotypic differences between autosomal dominant and recessive types (excluding isolated cases), analysing 10 kindreds of dominant inheritance (three in the current survey, seven from published reports) and six of recessive inheritance (three in the current survey, three from published reports). There appears to be no clinical or radiographical feature which clearly distinguishes them, but, using height as a criterion of severity, among those with autosomal recessive inheritance there was a disproportionate number of the most severely affected cases and there also appears to be very little intrafamilial variation. It is possible that pseudoachondroplasia can be subdivided into autosomal dominant mild and severe and autosomal recessive mild and severe, but full delineation must await elucidation of the basic defect at biochemical and molecular levels.

摘要

本调查回顾了来自26个家族的32例假软骨发育不全病例的诊断(主要是放射学诊断),并阐述了该疾病从婴儿期到成年期骨骼生长紊乱的自然病史及不同表现。此外,还尝试检测常染色体显性和隐性类型(不包括散发病例)之间的表型差异,分析了10个显性遗传家族(本次调查中有3个,已发表报告中有7个)和6个隐性遗传家族(本次调查中有3个,已发表报告中有3个)。似乎没有能明确区分它们的临床或影像学特征,但是,以身高作为严重程度的标准,在常染色体隐性遗传患者中,受影响最严重的病例比例过高,而且家族内差异似乎也很小。假软骨发育不全有可能可细分为常染色体显性轻度和重度以及常染色体隐性轻度和重度,但要进行全面分类,必须等待在生化和分子水平上阐明其基本缺陷。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/e0a1/1049780/d95a704e9abd/jmedgene00091-0043-a.jpg

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