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21例意大利异染性脑白质营养不良患者的ARSA和PSAP基因分子分析:11个新的ARSA等位基因的鉴定与功能表征

Molecular analysis of ARSA and PSAP genes in twenty-one Italian patients with metachromatic leukodystrophy: identification and functional characterization of 11 novel ARSA alleles.

作者信息

Grossi Serena, Regis Stefano, Rosano Camillo, Corsolini Fabio, Uziel Graziella, Sessa Maria, Di Rocco Maja, Parenti Giancarlo, Deodato Federica, Leuzzi Vincenzo, Biancheri Roberta, Filocamo Mirella

机构信息

Laboratorio Diagnosi Pre-Postnatale Malattie Metaboliche, Genova, Italy.

出版信息

Hum Mutat. 2008 Nov;29(11):E220-30. doi: 10.1002/humu.20851.

Abstract

Metachromatic leukodystrophy (MLD), the demyelinating disorder resulting from impaired sulfatide catabolism, is caused by allelic mutations of the Arylsulfatase A (ARSA) locus except for extremely rare cases of Saposin-B (Sap-B) deficiency. We characterized twenty-one unrelated Italian patients among which seventeen were due to ARSA activity deficiency and 4 others resulted from Saposin-B defect. Overall, we found 20 different mutant ARSA alleles and 2 different Sap-B alleles. The eleven new ARSA alleles (c.53C>A; c.88G>C; c.372G>A; c.409_411delCCC; c.634G>C; [c.650G>A;c.1108C>T]; c.845A>G; c.906G>C; c.919G>T; c.1102-3C>G; c.1126T>A) were functionally characterized and the novel amino acid changes were also modelled into the three-dimensional structure. The present study is aimed at providing a broader picture of the molecular basis of MLD in the Italian population. It also emphasizes the importance of a comprehensive evaluation in MLD diagnosis including biochemical, enzymatic and molecular investigations.

摘要

异染性脑白质营养不良(MLD)是一种因硫脂分解代谢受损导致的脱髓鞘疾病,除极罕见的鞘脂激活蛋白B(Sap - B)缺乏病例外,均由芳基硫酸酯酶A(ARSA)基因座的等位基因突变引起。我们对21名无亲缘关系的意大利患者进行了特征分析,其中17名是由于ARSA活性缺乏,另外4名是由鞘脂激活蛋白B缺陷导致的。总体而言,我们发现了20种不同的突变ARSA等位基因和2种不同的鞘脂激活蛋白B等位基因。对11种新的ARSA等位基因(c.53C>A;c.88G>C;c.372G>A;c.409_411delCCC;c.634G>C;[c.650G>A;c.1108C>T];c.845A>G;c.906G>C;c.919G>T;c.1102 - 3C>G;c.1126T>A)进行了功能特征分析,并将新的氨基酸变化也构建到三维结构中。本研究旨在更全面地了解意大利人群中MLD的分子基础。它还强调了在MLD诊断中进行综合评估的重要性,包括生化、酶学和分子研究。

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