Rinaldi F, Botta A, Vallo L, Contino G, Morgante A, Iraci R, Catalli C, Silvestri G, Ventriglia V M, Politano L, Novelli G
Department of Biopathology, University of Rome Tor Vergata, Rome, Italy.
Acta Myol. 2008 Dec;27(3):82-9.
Myotonic dystrophy type 1 (DM1) is the most frequently inherited neuromuscular disease in adults. It is a multisystemic disorder with major cardiac involvement most commonly represented by first-degree atrioventricular heart block (AVB), followed by different degrees of bundle-branch and intraventricular blocks In search for candidate genes, modifiers of the AVB phenotype in DM1, the expression of the small-conductance calcium activated potassium channel (SK3) gene was analysed in muscle biopsies from DM1 patients. The association between SK3 polymorphisms and the AVB phenotype was then studied analyzing 40 DM1 patients with AVB and 40 age-matched DM1 affected individuals with no ECG abnormalities. [CTG]n repeat length and cardiac clinical picture were also assessed for correlation. QRT-PCR experiments showed an over-expression of the SK3 transcript in DM1 muscle biopsies compared to healthy controls. However, no statistical association between the AVB phenotype and either the [CTG]n expansion length or the presence of specific SNPs in the SK3 gene were detected. These findings suggest that modifier genes, other than SK3, should be identified in order to explain the cardiac phenotypic variability among DM1 patients.
1型强直性肌营养不良(DM1)是成人中最常见的遗传性神经肌肉疾病。它是一种多系统疾病,主要累及心脏,最常见的表现是一度房室传导阻滞(AVB),其次是不同程度的束支和室内阻滞。为了寻找DM1中AVB表型的候选基因和修饰基因,对DM1患者的肌肉活检样本进行了小电导钙激活钾通道(SK3)基因表达分析。然后,通过分析40例患有AVB的DM1患者和40例年龄匹配、无心电图异常的DM1患者,研究了SK3基因多态性与AVB表型之间的关联。同时评估了[CTG]n重复长度与心脏临床表现之间的相关性。定量逆转录聚合酶链反应(QRT-PCR)实验表明,与健康对照相比,DM1肌肉活检样本中SK3转录本表达上调。然而,未检测到AVB表型与SK3基因的[CTG]n扩增长度或特定单核苷酸多态性(SNP)之间存在统计学关联。这些发现表明,为了解释DM1患者心脏表型的变异性,应鉴定出SK3基因以外的修饰基因。