Barhmi R, Jacquemot C, el Rabiet G, Lasfargues P
Maternité, CHR, Versailles.
Rev Fr Gynecol Obstet. 1994 Dec;89(12):613-6.
The authors report a prenatal diagnosis of osteogenesis imperfecta or Porak and Durante disease, in which the child survived. The diagnosis was made by ultrasonography which revealed major abnormalities of osteogenesis more particularly affecting the lower limbs, which were short and deformed. Other essential features of this syndrome are osteoporosis, hyperrelaxation of ligaments and blue sclerae. This rare and genetic condition is due to type I collagen abnormalities. It is often governed by dominant transmission but manifestation of the gene is variable within a given family. Molecular biology and genetic studies offer new possibilities of prenatal diagnosis, but ultrasonography remains the investigation of choice, possibly helped by X-ray of the uterine contents.
作者报告了一例成骨不全症(又称波拉克和杜兰特病)的产前诊断病例,患儿存活。诊断通过超声检查做出,该检查显示成骨存在严重异常,尤其影响下肢,下肢短小且畸形。该综合征的其他基本特征包括骨质疏松、韧带过度松弛和巩膜呈蓝色。这种罕见的遗传病是由I型胶原蛋白异常所致。它通常由显性遗传控制,但在特定家族中基因表现存在差异。分子生物学和基因研究为产前诊断提供了新的可能性,但超声检查仍是首选的检查方法,子宫内容物的X线检查可能会辅助诊断。