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豹皮综合征中的遗传异质性:两个家系的PTPN11基因无突变

Genetic heterogeneity in LEOPARD syndrome: two families with no mutations in PTPN11.

作者信息

Kalidas Kamini, Shaw Adam C, Crosby Andrew H, Newbury-Ecob Ruth, Greenhalgh Lynn, Temple Isabel K, Law Caroline, Patel Amisha, Patton Michael A, Jeffery Steve

机构信息

Medical Genetics Unit, Department of Clinical Developmental Sciences, St. George's Hospital Medical School, London, SW17 0RE, UK.

Clinical Genetics Service, Bristol Royal Hospital for Sick Children, Bristol, UK.

出版信息

J Hum Genet. 2005;50(1):21-25. doi: 10.1007/s10038-004-0212-x. Epub 2004 Dec 10.

Abstract

LEOPARD syndrome (lentigines, electrocardiographic conduction abnormalities, ocular hypertelorism, pulmonary stenosis, abnormal genitalia, retardation of growth, and sensorineural deafness) is an autosomal dominant condition. The main clinical features include multiple lentigines, cardiovascular defects, and facial anomalies, some of which are shared with Noonan syndrome (NS). Recent reports have shown that LEOPARD syndrome can be caused by mutations in PTPN11, the gene in which mutations can produce NS. Here we report the findings of mutation screening and linkage analysis of PTPN11 in three families with LEOPARD syndrome. We identified a novel mutation in one family. The mutation (1529A>C) substitutes proline for glutamine at amino acid 510 (Gln510Pro). No variations in sequence were observed in the other two families, and negative LOD scores excluded linkage to the PTPN11 locus, showing that LEOPARD syndrome is genetically heterogeneous.

摘要

豹皮综合征(雀斑、心电图传导异常、眼距增宽、肺动脉狭窄、生殖器异常、生长发育迟缓及感觉神经性耳聋)是一种常染色体显性遗传病。其主要临床特征包括多发性雀斑、心血管缺陷及面部异常,其中一些与努南综合征(NS)相同。最近的报告显示,豹皮综合征可能由PTPN11基因突变引起,该基因突变可导致努南综合征。本文报告了对三个豹皮综合征家系进行PTPN11突变筛查及连锁分析的结果。我们在一个家系中鉴定出一个新的突变。该突变(1529A>C)导致第510位氨基酸由谷氨酰胺替换为脯氨酸(Gln510Pro)。在另外两个家系中未观察到序列变异,且阴性对数优势分数排除了与PTPN11基因座的连锁关系,表明豹皮综合征在遗传上具有异质性。

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