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角膜改变、角化过度、身材矮小、短指畸形和早产:一种新的常染色体显性综合征。

Corneal changes, hyperkeratosis, short stature, brachydactyly, and premature birth: a new autosomal dominant syndrome.

作者信息

Stern J K, Lubinsky M S, Durrie D S, Luckasen J R

出版信息

Am J Med Genet. 1984 May;18(1):67-77. doi: 10.1002/ajmg.1320180111.

Abstract

We report on an autosomal dominant syndrome consisting of unique corneal epithelial changes, diffuse palmoplantar hyperkeratosis, distal onycholysis, brachydactyly, short stature, premature birth, and dental problems. This condition has been present in seven persons in three generations of one family. Corneal biopsies demonstrate mild dysplastic changes in the epithelium. Skin biopsies show hyperkeratosis and acanthosis. In both eye and skin specimens, results of stains for polysaccharides, amyloid, and tyrosine were unremarkable. Roentgenograms of the hands show short distal phalanges, short 4th metacarpals, and constriction of the heads of some of the metacarpals. In three of four affected relatives, a variable medullary narrowness is seen. In mode of inheritance, clinical appearance, and/or associated defects. This syndrome appears to differ from previously reported conditions that include palmoplantar hyperkeratosis and/or corneal changes.

摘要

我们报告了一种常染色体显性综合征,其特征包括独特的角膜上皮改变、弥漫性掌跖角化过度、甲远端分离、短指畸形、身材矮小、早产和牙齿问题。这种病症在一个家族的三代人中出现了7例。角膜活检显示上皮有轻度发育异常改变。皮肤活检显示角化过度和棘皮症。在眼部和皮肤标本中,多糖、淀粉样蛋白和酪氨酸染色结果均无异常。手部X线片显示远端指骨短、第四掌骨短以及一些掌骨头狭窄。在4名受影响的亲属中,有3人可见不同程度的骨髓狭窄。在遗传方式、临床表现和/或相关缺陷方面。该综合征似乎与先前报道的包括掌跖角化过度和/或角膜改变的病症不同。

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