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哈勒曼-施特雷夫综合征:无证据表明与核纤层蛋白病有关。

Hallermann-Streiff Syndrome: No Evidence for a Link to Laminopathies.

作者信息

Kortüm F, Chyrek M, Fuchs S, Albrecht B, Gillessen-Kaesbach G, Mütze U, Seemanova E, Tinschert S, Wieczorek D, Rosenberger G, Kutsche K

机构信息

Institut für Humangenetik, Universitätsklinikum Hamburg-Eppendorf, Hamburg, Germany.

出版信息

Mol Syndromol. 2011 Dec;2(1):27-34. doi: 10.1159/000334317. Epub 2011 Nov 12.

Abstract

Hallermann-Streiff syndrome (HSS) is a rare inherited disorder characterized by malformations of the cranium and facial bones, congenital cataracts, microphthalmia, skin atrophy, hypotrichosis, proportionate short stature, teeth abnormalities, and a typical facial appearance with prominent forehead, small pointed nose, and micrognathia. The genetic cause of this developmental disorder is presently unknown. Here we describe 8 new patients with a phenotype of HSS. Individuals with HSS present with clinical features overlapping with some progeroid syndromes that belong to the laminopathies, such as Hutchinson-Gilford progeria syndrome (HGPS) and mandibuloacral dysplasia (MAD). HGPS is caused by de novo point mutations in the LMNA gene, coding for the nuclear lamina proteins lamin A and C. MAD with type A and B lipodystrophy are recessive disorders resulting from mutations in LMNA and ZMPSTE24, respectively. ZMPSTE24 in addition to ICMT encode proteins involved in posttranslational processing of lamin A. We hypothesized that HSS is an allelic disorder to HGPS and MAD. As the nuclear shape is often irregular in patients with LMNA mutations, we first analyzed the nuclear morphology in skin fibroblasts of patients with HSS, but could not identify any abnormality. Sequencing of the genes LMNA, ZMPSTE24 and ICMT in the 8 patients with HSS revealed the heterozygous missense mutation c.1930C>T (p.R644C) in LMNA in 1 female. Extreme phenotypic diversity and low penetrance have been associated with the p.R644C mutation. In ZMPSTE24 and ICMT, no pathogenic sequence change was detected in patients with HSS. Together, we found no evidence that HSS is another laminopathy.

摘要

哈勒曼-施特雷夫综合征(HSS)是一种罕见的遗传性疾病,其特征为颅骨和面部骨骼畸形、先天性白内障、小眼症、皮肤萎缩、毛发稀少、身材比例矮小、牙齿异常,以及典型的面部外观,包括前额突出、小尖鼻和小颌畸形。这种发育障碍的遗传原因目前尚不清楚。在此,我们描述了8例具有HSS表型的新患者。HSS患者的临床特征与一些属于核纤层蛋白病的早老综合征有重叠,如哈钦森-吉尔福德早老综合征(HGPS)和下颌-肢端发育异常(MAD)。HGPS是由编码核纤层蛋白A和C的LMNA基因的新发点突变引起的。伴有A型和B型脂肪营养不良的MAD分别是由LMNA和ZMPSTE24基因突变导致的隐性疾病。ZMPSTE24与ICMT一起编码参与核纤层蛋白A翻译后加工的蛋白质。我们推测HSS是HGPS和MAD的等位基因疾病。由于LMNA突变患者的细胞核形状通常不规则,我们首先分析了HSS患者皮肤成纤维细胞的核形态,但未发现任何异常。对8例HSS患者的LMNA、ZMPSTE24和ICMT基因进行测序,发现1名女性患者的LMNA基因存在杂合错义突变c.1930C>T(p.R644C)。极端的表型多样性和低外显率与p.R644C突变有关。在HSS患者中,未在ZMPSTE24和ICMT中检测到致病序列变化。总之,我们没有发现证据表明HSS是另一种核纤层蛋白病。

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