Uhrhammer Nancy A, Lafarge Laurence, Dos Santos Laetitia, Domaszewska Anna, Lange Magdalena, Yang Yong, Aractingi Selim, Bessis Didier, Bignon Yves-Jean
Laboratoire Diagnostic Génétique et Moléculaire, Centre Jean Perrin, Clermont-Ferrand, France.
Hum Mutat. 2006 Jul;27(7):718-9. doi: 10.1002/humu.9435.
Werner syndrome (WS) is a pleiotropic disease of premature aging involving short stature, tight, atrophied, and/or ulcerated skin; a characteristic 'birdlike' facies and high, squeaky or hoarse voice; premature greying and thinning of the hair; and early onset cataracts. Additional common symptoms include diabetes mellitus, hypogonadism, osteoporosis, osteosclerosis of the digits, soft tissue calcification, premature atherosclerosis, rare or multiple neoplasms, malformed teeth, and flat feet. Diagnosis can be difficult due to the variable presentation and rarity of the disorder. Transmission is usually autosomal recessive. The WS gene, WRN, is member of the RecQ DNA helicase family. Biallelic mutations of WRN are responsible for most patients. Although heterozygous missense mutations in the LMNA gene have been observed in severely affected WS patients, this only accounts for a small fraction of non-WRN patients. Eighteen WS cases were referred to us for molecular analysis. Eleven had definite and three had probable WS according to the University of Washington Registry clinical criteria. All exons of the WRN gene and their splice junctions were sequenced. Of the fourteen definite or probable cases, 11 had one or more WRN mutation. Thirteen different mutations were found, and ten of these were previously undescribed. There were few phenotypic differences between patients with WRN mutation(s) and those who met clinical criteria though lacking WRN mutations. However, patients with mutations tended to have more symptoms overall, and mutations were not observed in the two cases with cardiomyopathy.
沃纳综合征(WS)是一种多效性早衰疾病,表现为身材矮小、皮肤紧绷、萎缩和/或溃疡;具有特征性的“鸟样”面容、声音高亢、尖锐或嘶哑;头发过早变白和稀疏;以及早发性白内障。其他常见症状包括糖尿病、性腺功能减退、骨质疏松、手指骨硬化、软组织钙化、过早发生动脉粥样硬化、罕见或多发肿瘤、牙齿畸形和平足。由于该疾病表现多样且罕见,诊断可能较为困难。其遗传方式通常为常染色体隐性遗传。WS基因WRN是RecQ DNA解旋酶家族成员。大多数患者由WRN双等位基因突变引起。尽管在严重受影响的WS患者中观察到LMNA基因杂合错义突变,但这仅占非WRN患者的一小部分。18例WS病例被转诊至我们处进行分子分析。根据华盛顿大学登记处临床标准,11例确诊为WS,3例可能为WS。对WRN基因的所有外显子及其剪接位点进行了测序。在14例确诊或可能的病例中,11例有一个或多个WRN突变。共发现13种不同突变,其中10种为先前未描述过的突变。有WRN突变的患者与符合临床标准但无WRN突变的患者之间在表型上差异不大。然而,有突变的患者总体症状往往更多,且在两例心肌病患者中未观察到突变。