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ACTN3 R577X 多态性与墨西哥人群中的炎性肌病有关。

The ACTN3 R577X polymorphism is associated with inflammatory myopathies in a Mexican population.

机构信息

Research Institute of Rheumatology and the Musculoskeletal System, University Centre for Health Sciences, University of Guadalajara, Guadalajara, Jalisco, Mexico.

出版信息

Scand J Rheumatol. 2012 Oct;41(5):396-400. doi: 10.3109/03009742.2012.669495. Epub 2012 May 29.

Abstract

BACKGROUND

The ACTN3 gene encodes the fast muscle protein α-actinin-3. The ACTN3 R577X polymorphism is a premature stop codon and results in absence of α-actinin-3 in 577XX homozygotes. The aim of this study was to determine the ACTN3 genotype in idiopathic inflammatory myopathies (IIMs).

METHODS

We performed ACTN3 genotyping on 27 patients with dermatomyositis (DM), 10 with polymyositis (PM), and 85 healthy subjects. Muscle enzyme levels of creatine phosphokinase (CPK), lactic dehydrogenase (LDH), aspartate aminotransferase (AST), and alanine aminotransferase (ALT) were recorded at the time of diagnosis and recruitment. Genotyping was performed by polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) and the allele frequency was analysed.

RESULTS

A total of 36% of healthy subjects had the ACTN3 577XX polymorphism (α-actinin-3 deficiency), 18% had the 577RR (homozygous wild type) genotype, and 46% 577RX (heterozygous). In DM/PM, 70% had the ACTN3 577XX polymorphism, 6% RR, and 24% RX [odds ratio (OR) 4.12, 95% confidence interval (CI) 1.67-10.33, p < 0.001]. In healthy subjects, the R allele was present in 41% and the X allele in 59% compared to 18% and 82%, respectively, in the IIM group (OR 3.21, 95% CI 1.57-6.66, p < 0.001). Thus, the ACTN3 577X allele seemed to increase the risk of developing IIM, and DM in particular, although this was not related to severity of expression of the phenotype.

CONCLUSIONS

The ACTN3 577X allele appeared to increase the risk of developing IIM; 70% of IIM patients were deficient in α-actinin-3. By contrast, ACTN3 577XX patients seemed to have less severe disease as reflected in lower muscle enzyme levels.

摘要

背景

ACTN3 基因编码快肌蛋白 α-辅肌动蛋白-3。ACTN3 R577X 多态性是一个提前终止密码子,导致 577XX 纯合子中缺乏 α-辅肌动蛋白-3。本研究旨在确定特发性炎性肌病(IIM)中的 ACTN3 基因型。

方法

我们对 27 例皮肌炎(DM)、10 例多发性肌炎(PM)和 85 例健康受试者进行了 ACTN3 基因分型。在诊断和招募时记录肌酸磷酸激酶(CPK)、乳酸脱氢酶(LDH)、天门冬氨酸氨基转移酶(AST)和丙氨酸氨基转移酶(ALT)的肌肉酶水平。通过聚合酶链反应-限制性片段长度多态性(PCR-RFLP)进行基因分型,并分析等位基因频率。

结果

36%的健康受试者携带 ACTN3 577XX 多态性(α-辅肌动蛋白-3 缺乏),18%携带 577RR(纯合野生型)基因型,46%携带 577RX(杂合型)。在 DM/PM 中,70%的患者携带 ACTN3 577XX 多态性,6%携带 RR,24%携带 RX[比值比(OR)4.12,95%置信区间(CI)1.67-10.33,p<0.001]。在健康受试者中,R 等位基因的出现频率为 41%,X 等位基因的出现频率为 59%,而在 IIM 组中,R 等位基因的出现频率为 18%,X 等位基因的出现频率为 82%[OR 3.21,95%置信区间(CI)1.57-6.66,p<0.001]。因此,ACTN3 577X 等位基因似乎增加了发生 IIM 的风险,尤其是 DM,但这与表型表达的严重程度无关。

结论

ACTN3 577X 等位基因似乎增加了发生 IIM 的风险;70%的 IIM 患者缺乏 α-辅肌动蛋白-3。相比之下,ACTN3 577XX 患者的疾病似乎不那么严重,表现为肌肉酶水平较低。

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