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成纤维细胞生长因子受体2基因的突变会导致克鲁宗综合征。

Mutations in the fibroblast growth factor receptor 2 gene cause Crouzon syndrome.

作者信息

Reardon W, Winter R M, Rutland P, Pulleyn L J, Jones B M, Malcolm S

机构信息

Mothercare Unit of Clinical Genetics and Fetal Medicine, Institute of Child Health, London, UK.

出版信息

Nat Genet. 1994 Sep;8(1):98-103. doi: 10.1038/ng0994-98.

Abstract

Crouzon syndrome is an autosomal dominant condition causing premature fusion of the cranial sutures (craniosynostosis) and maps to chromosome 10q25-q26. We now present evidence that mutations in the fibroblast growth factor receptor 2 gene (FGFR2) cause Crouzon syndrome. We found SSCP variations in the B exon of FGFR2 in nine unrelated affected individuals as well as complete cosegregation between SSCP variation and disease in three unrelated multigenerational families. In four sporadic cases, the normal parents did not have SSCP variation. Finally, direct sequencing has revealed specific mutations in the B exon in all nine sporadic and familial cases, including replacement of a cysteine in an immunoglobulin-like domain in five patients.

摘要

克鲁宗综合征是一种常染色体显性疾病,可导致颅缝过早融合(颅缝早闭),定位于10q25 - q26染色体。我们现在提供证据表明,成纤维细胞生长因子受体2基因(FGFR2)的突变会导致克鲁宗综合征。我们在9名无亲缘关系的患病个体中发现了FGFR2基因B外显子的单链构象多态性(SSCP)变异,以及在3个无亲缘关系的多代家族中SSCP变异与疾病的完全共分离现象。在4例散发病例中,正常父母没有SSCP变异。最后,直接测序揭示了所有9例散发性和家族性病例的B外显子中存在特定突变,包括5例患者免疫球蛋白样结构域中的一个半胱氨酸被替换。

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