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迟发性骨发育不全:一种新型软骨发育不全,主要累及手部骨骼和椎骨。

Opsismodysplasia: a new type of chondrodysplasia with predominant involvement of the bones of the hand and the vertebrae.

作者信息

Maroteaux P, Stanescu V, Stanescu R, Le Marec B, Moraine C, Lejarraga H

出版信息

Am J Med Genet. 1984 Sep;19(1):171-82. doi: 10.1002/ajmg.1320190117.

Abstract

The name opsismodysplasia is proposed for a new chondrodysplasia, which was studied in three patients. Clinically, the condition is recognized at birth on the basis of shortness, short hands, and facial abnormalities with a short nose and a depressed bridge of nose. The most characteristic radiographic signs are: very retarded bone maturation; marked shortness of the bones of the hands and of the feet with concave metaphyses; and thin, lamellar vertebral bodies. The growth cartilage studied in one case showed a wide hypertrophic area containing thick connective tissue septa, irregular provisional calcification, and vascular invasion. Type I collagen was detected in the hypertrophic area by immunohistochemical and microchemical tests. The transmission of opsismodysplasia is probably autosomal recessive.

摘要

我们为一种新的软骨发育不良提出了“迟发性发育异常”这一名称,该疾病已在三名患者身上进行了研究。临床上,这种病症在出生时即可根据身材矮小、手部短小以及面部异常(短鼻和鼻梁凹陷)得以识别。最具特征性的放射学表现为:骨成熟显著延迟;手部和足部骨骼明显短小,干骺端凹陷;椎体薄且呈板层状。在一例病例中研究的生长软骨显示出一个宽阔的肥大区域,其中包含粗大的结缔组织间隔、不规则的临时钙化和血管侵入。通过免疫组织化学和微量化学检测在肥大区域检测到了I型胶原蛋白。迟发性发育异常的遗传方式可能为常染色体隐性遗传。

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