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阿尔及利亚队列中I型黏多糖贮积症患者的临床和分子特征

Clinical and Molecular Characterization of Patients with Mucopolysaccharidosis Type I in an Algerian Series.

作者信息

Tebani Abdellah, Zanoutene-Cheriet Lahouaria, Adjtoutah Zoubir, Abily-Donval Lenaig, Brasse-Lagnel Carole, Laquerrière Annie, Marret Stephane, Chalabi Benabdellah Abla, Bekri Soumeya

机构信息

Department of Metabolic Biochemistry, Rouen University Hospital, Rouen 76031, France.

Region-Inserm Team NeoVasc ERI28, Laboratory of Microvascular Endothelium and Neonatal Brain Lesions, Institute of Research for Innovation in Biomedicine, Normandy University, Rouen 76031, France.

出版信息

Int J Mol Sci. 2016 May 17;17(5):743. doi: 10.3390/ijms17050743.

Abstract

Mucopolysaccharidoses (MPS's) represent a subgroup of lysosomal storage diseases related to a deficiency of enzymes that catalyze glycosaminoglycans degradation. Mucopolysaccharidosis type I (MPS I) is a rare autosomal recessive disorder caused by a deficiency of α-l-iduronidase encoded by the IDUA gene. Partially degraded heparan sulfate and dermatan sulfate accumulate progressively and lead to multiorgan dysfunction and damage. The aim of this study is to describe the clinical, biochemical, and molecular characteristics of 13 Algerian patients from 11 distinct families. MPS I diagnosis was confirmed by molecular study of the patients' IDUA gene. Clinical features at the diagnosis and during the follow-up are reported. Eighty-four percent of the studied patients presented with a mild clinical phenotype. Molecular study of the IDUA gene allowed the characterization of four pathological variations at the homozygous or compound heterozygote status: IDUA NM_00203.4:c.1598C>G-p.(Pro533Arg) in 21/26 alleles, IDUA NM_00203.4:c.532G>A-p.(Glu178Lys) in 2/26 alleles, IDUA NM_00203.4:c.501C>G-p.(Tyr167*) in 2/26 alleles, and IDUA NM_00203. 4: c.1743C>G-p.(Tyr581*) in 1/26 alleles. This molecular study unveils the predominance of p.(Pro533Arg) variation in our MPS I patients. In this series, the occurrence of some clinical features linked to the Scheie syndrome is consistent with the literature, such as systematic valvulopathies, corneal opacity, and umbilical hernia; however, storage signs, facial dysmorphic features, and hepatomegaly were more frequent in our series. Screening measures for these debilitating diseases in highly consanguineous at-risk populations must be considered a priority health problem.

摘要

黏多糖贮积症(MPS)是溶酶体贮积病的一个亚组,与催化糖胺聚糖降解的酶缺乏有关。I型黏多糖贮积症(MPS I)是一种罕见的常染色体隐性疾病,由IDUA基因编码的α-L-艾杜糖醛酸酶缺乏引起。部分降解的硫酸乙酰肝素和硫酸皮肤素逐渐积累,导致多器官功能障碍和损害。本研究的目的是描述来自11个不同家庭的13例阿尔及利亚患者的临床、生化和分子特征。通过对患者IDUA基因的分子研究确诊为MPS I。报告了诊断时和随访期间的临床特征。84%的研究患者表现为轻度临床表型。对IDUA基因的分子研究确定了纯合子或复合杂合子状态下的四种病理变异:IDUA NM_00203.4:c.1598C>G-p.(Pro533Arg),占21/26个等位基因;IDUA NM_00203.4:c.532G>A-p.(Glu178Lys),占2/26个等位基因;IDUA NM_00203.4:c.501C>G-p.(Tyr167*),占2/26个等位基因;以及IDUA NM_00203.4:c.1743C>G-p.(Tyr581*),占1/26个等位基因。这项分子研究揭示了p.(Pro533Arg)变异在我们的MPS I患者中占主导地位。在本系列中,一些与谢伊综合征相关的临床特征的出现与文献一致,如系统性瓣膜病、角膜混浊和脐疝;然而,在我们的系列中,贮积体征、面部畸形特征和肝肿大更为常见。在高度近亲的高危人群中对这些致残性疾病的筛查措施必须被视为一个优先的健康问题。

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