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显性中肢发育不良、踝关节、腕骨和跗骨融合型:一种新的常染色体显性骨病。

Dominant mesomelic dysplasia, ankle, carpal, and tarsal synostosis type: a new autosomal dominant bone disorder.

作者信息

Kantaputra P N, Gorlin R J, Langer L O

机构信息

Department of Pediatric Dentistry, School of Dentistry, Chiangmai University, Thailand.

出版信息

Am J Med Genet. 1992 Dec 1;44(6):730-7. doi: 10.1002/ajmg.1320440606.

Abstract

A new type of mesomelic dysplasia was in 3 generations of a large Thai family. It is characterized by bilateral symmetrical marked shortening of the ulnae and shortening and bowing of the radii. The proximal fibula is usually short and synostoses are present between the tibia and fibula and the small malformed calcaneus and talus. The prominent calcanei on the ventral surfaces of the distal fibulae are a characteristic feature of the new type. Carpal and tarsal synostoses are present in some affected people. All affected individuals walk on the tips of their toes with the dorsal foot deviated laterally. The deformities of the radius and ulna somewhat resemble those of mesomelic dysplasia, Langer type, but otherwise the condition is distinctly different. This new mesomelic dysplasia is an autosomal dominant trait with complete penetrance and variable expressivity over 3 generations.

摘要

一个泰国大家庭的三代人中出现了一种新型的中肢发育不全。其特征是双侧尺骨明显对称缩短,桡骨缩短并弯曲。近端腓骨通常较短,胫骨和腓骨之间以及畸形的小跟骨和距骨之间存在骨性连接。远端腓骨腹面突出的跟骨是这种新型的一个特征性表现。一些患者存在腕骨和跗骨骨性连接。所有患者均以足背外侧偏斜、用脚尖行走。桡骨和尺骨的畸形在一定程度上类似于朗格型中肢发育不全,但在其他方面,这种情况明显不同。这种新型中肢发育不全是一种常染色体显性性状,在三代人中具有完全外显率和可变表达性。

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