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指甲-髌骨综合征:55个家族的临床和分子数据,提出遗传异质性假说。

Nail-Patella Syndrome: clinical and molecular data in 55 families raising the hypothesis of a genetic heterogeneity.

作者信息

Ghoumid Jamal, Petit Florence, Holder-Espinasse Muriel, Jourdain Anne-Sophie, Guerra José, Dieux-Coeslier Anne, Figeac Martin, Porchet Nicole, Manouvrier-Hanu Sylvie, Escande Fabienne

机构信息

Service de Génétique Clinique, Hôpital Jeanne de Flandre, CHRU, Lille, France.

Faculté de Médecine, Université Lille Nord de France, Lille, France.

出版信息

Eur J Hum Genet. 2016 Jan;24(1):44-50. doi: 10.1038/ejhg.2015.77. Epub 2015 Apr 22.

Abstract

Nail-Patella Syndrome (NPS) is a rare autosomal dominant condition comprising nail and skeletal anomalies. Skeletal features include dysplastic patellae and iliac horns, as well as scapula and elbow dysplasia. Nephropathy and glaucoma or intra-ocular hypertension can sometimes be present. NPS is due to variants affecting function in LMX1B, which encodes a LIM-homeodomain protein critical for limb, kidney and eye development. We describe the phenotype and the molecular data of 55 index patients and their 39 relatives presenting with typical NPS. We identified 38 different LMX1B anomalies, 19 of which were not reported before. In our series, 9% of families are not carriers of a LMX1B genomic alteration after extensive study of the coding and non-coding regions of the gene. One of the families showed no linkage to the LMX1B locus, raising the hypothesis of a genetic heterogeneity.

摘要

指甲-髌骨综合征(NPS)是一种罕见的常染色体显性疾病,包括指甲和骨骼异常。骨骼特征包括发育不良的髌骨和髂角,以及肩胛骨和肘部发育不良。有时可能会出现肾病和青光眼或眼内高压。NPS是由于影响LMX1B功能的变异所致,LMX1B编码一种对肢体、肾脏和眼睛发育至关重要的LIM同源域蛋白。我们描述了55例典型NPS索引患者及其39名亲属的表型和分子数据。我们鉴定出38种不同的LMX1B异常,其中19种以前未曾报道过。在我们的系列研究中,经过对该基因编码区和非编码区的广泛研究,9%的家族不是LMX1B基因组改变的携带者。其中一个家族与LMX1B基因座无连锁关系,这提出了遗传异质性的假设。

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