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伴有先天性关节挛缩的成骨不全症(布鲁克综合征)。

Osteogenesis imperfecta with congenital joint contractures (Bruck syndrome).

作者信息

Viljoen D, Versfeld G, Beighton P

机构信息

Department of Human Genetics, Medical School, University of Cape Town, South Africa.

出版信息

Clin Genet. 1989 Aug;36(2):122-6. doi: 10.1111/j.1399-0004.1989.tb03174.x.

Abstract

Five children from three unrelated families were born with symmetrical contractures of the knees, ankles and feet. An initial diagnosis of arthrogryposis multiplex was made, but frequent fracturing occurred after walking commenced and it was then recognised that the children had osteogenesis imperfecta. The pathogenesis of the congenital contractures is unknown, but the symmetry and lack of evidence of prior fracturing is suggestive of articular immobility during early intra-uterine development. The consistency of the anatomical distribution of the contractures, in the setting of a uniform OI phenotype, is suggestive of syndromic identity. A similar case was documented by Alfred Bruck in 1897 and we propose that the eponymous designation "Bruck syndrome" should be applied to the disorder.

摘要

来自三个无亲缘关系家庭的五个孩子出生时膝盖、脚踝和足部出现对称性挛缩。最初诊断为多发性关节挛缩症,但开始行走后频繁发生骨折,随后发现这些孩子患有成骨不全症。先天性挛缩的发病机制尚不清楚,但挛缩的对称性以及缺乏先前骨折的证据提示在子宫内早期发育期间关节活动受限。在一致的成骨不全症表型背景下,挛缩的解剖分布具有一致性,提示存在综合征特征。1897年阿尔弗雷德·布鲁克记录了一个类似病例,我们建议将该病症命名为“布鲁克综合征”。

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