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威斯科特-奥尔德里奇综合征:不存在严格的基因型-表型相关性,但错义突变集中在WASP基因产物的氨基末端部分。

Wiskott-Aldrich syndrome: no strict genotype-phenotype correlations but clustering of missense mutations in the amino-terminal part of the WASP gene product.

作者信息

Schindelhauer D, Weiss M, Hellebrand H, Golla A, Hergersberg M, Seger R, Belohradsky B H, Meindl A

机构信息

Abteilung für Pädiatrische Genetik der Kinderpoliklinik, München, Germany.

出版信息

Hum Genet. 1996 Jul;98(1):68-76. doi: 10.1007/s004390050162.

Abstract

The Wiskott-Aldrich syndrome protein (WASP) gene was found to be mutated in patients presenting with WAS and in patients showing X-linked thrombocytopenia. Mutation analysis in 19 families of German, Swiss and Turkish descent by single-strand conformation polymorphism and sequencing resulted in the detection of seven novel and 10 known mutations. A striking clustering of missense mutations in the first four exons contrasted with a random distribution of nonsense mutations. More than 85% of all known missense mutations were localized in the amino-terminal stretch of the WASP gene product; this region contained a mutational hot spot at codon 86. No genotype-phenotype correlation emerged after a comparison of the identified mutations with the resulting clinical picture for a classical WAS phenotype. A substitution at codon 86 resulted in an extremely variable expression of the disease in a large Swiss family. An extended homology search revealed a distant relationship of this stretch to the vasodilator-stimulated phosphoprotein (VASP), which is involved in the maintenance of cyto-architecture by interacting with actin-like filaments.

摘要

威斯科特-奥尔德里奇综合征蛋白(WASP)基因在患有威斯科特-奥尔德里奇综合征(WAS)的患者以及表现为X连锁血小板减少症的患者中被发现发生了突变。通过单链构象多态性和测序对19个德裔、瑞士裔和土耳其裔家庭进行突变分析,结果检测到7个新突变和10个已知突变。前四个外显子中错义突变显著聚集,与无义突变的随机分布形成对比。所有已知错义突变中超过85%位于WASP基因产物的氨基末端区域;该区域在密码子86处存在一个突变热点。将鉴定出的突变与经典WAS表型的临床症状进行比较后,未发现基因型与表型之间的相关性。密码子86处的一个替换导致一个瑞士大家庭中该病的表达极具变异性。进一步的同源性搜索揭示了该区域与血管舒张刺激磷蛋白(VASP)存在远缘关系,VASP通过与肌动蛋白样细丝相互作用参与细胞结构的维持。

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