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骨骼发育异常的出生患病率

The birth prevalence rates for the skeletal dysplasias.

作者信息

Orioli I M, Castilla E E, Barbosa-Neto J G

出版信息

J Med Genet. 1986 Aug;23(4):328-32. doi: 10.1136/jmg.23.4.328.

Abstract

This study was undertaken to establish the prevalence rates at birth of the skeletal dysplasias that can be recognised in the perinatal period. Using the data base of the Latin-American Collaborative Study of Congenital Malformations (ECLAMC), for the years 1978 to 1983, on 349 470 births (live and stillbirths), a crude prevalence rate of 2.3/10 000 was observed. However, several indications of under-registration suggest that the real value is about twice that observed. The most frequent types of skeletal dysplasia were achondroplasia, with a prevalence rate between 0.5 and 1.5/10 000 births, the thanatophoric dysplasia/achondrogenesis group (0.2 and 0.5/10 000 births), and osteogenesis imperfecta (0.4/10 000 births). The mutation rate for autosomal dominant achondroplasia was estimated at between 1.72 and 5.57 X 10(-5) per gamete per generation.

摘要

本研究旨在确定围产期可识别的骨骼发育异常在出生时的患病率。利用拉丁美洲先天性畸形协作研究(ECLAMC)1978年至1983年期间349470例出生(活产和死产)的数据库,观察到粗患病率为2.3/10000。然而,一些登记不足的迹象表明,实际值约为观察值的两倍。最常见的骨骼发育异常类型是软骨发育不全,患病率在0.5至1.5/10000例出生之间,致死性发育异常/软骨发育不全组(0.2至0.5/10000例出生),以及成骨不全(0.4/10000例出生)。常染色体显性软骨发育不全的突变率估计为每代每个配子1.72至5.57×10⁻⁵。

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