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调查 ANKH 和 ENPP1 在斯洛伐克软骨钙质沉着症家系中的作用。

Investigating ANKH and ENPP1 in Slovakian families with chondrocalcinosis.

机构信息

Serviço Especializado de Epidemiologia e Biologia Molecular, Hospital de Santo Espírito, Angra do Heroísmo, Azores.

出版信息

Rheumatol Int. 2012 Sep;32(9):2745-51. doi: 10.1007/s00296-011-2022-8. Epub 2011 Aug 3.

Abstract

Familial articular chondrocalcinosis (CC) was first reported in 1963. It is characterised by multiple calcifications of hyaline and fibrous cartilage in the joints and intervertebral discs. Mutations in ANKH have been identified in several pedigrees as a monogenic cause for this disorder. ANKH is a key protein in pyrophosphate metabolism and is involved in pyrophosphate transport across the cell membrane. The objective of this work was to screen ANKH and ENPP1, two key genes in pyrophosphate metabolism, in Slovakian kindreds with familial CC. DNA samples from 25 individuals (10 affected, 15 unaffected) from 8 families were obtained. The promoter, coding regions and intron-exon boundaries of ANKH and ENPP1 were sequenced. Twelve DNA sequence variants, six in each gene, were identified. All the variants had been previously identified. None segregated with the disease. Our results suggest that neither ANKH nor ENPP1 mutations are the cause of CC in these families, indicating that possibly other major genes are involved in the aethiopathogenesis of this condition in these families.

摘要

家族性关节软骨钙质沉着症(CC)于 1963 年首次报道。其特征为关节和椎间盘透明软骨和纤维软骨的多处钙化。ANKH 的突变已在几个家系中被确定为该疾病的单基因原因。ANKH 是焦磷酸代谢中的关键蛋白,参与焦磷酸盐跨细胞膜的转运。本研究的目的是在斯洛伐克家族性 CC 家系中筛查焦磷酸盐代谢的两个关键基因 ANKH 和 ENPP1。从 8 个家系的 25 个人(10 个受影响,15 个未受影响)中获得 DNA 样本。对 ANKH 和 ENPP1 的启动子、编码区和内含子-外显子边界进行了测序。鉴定出 12 个 DNA 序列变异,每个基因 6 个。所有变异均已被先前鉴定。没有一个与疾病共分离。我们的结果表明,ANKH 和 ENPP1 突变均不是这些家系中 CC 的原因,这表明在这些家系中,可能涉及其他主要基因参与该病的发病机制。

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