Laboratoire de Génétique Moléculaire Humaine, Faculté de Médecine de Sfax, Université de Sfax, Avenue Magida Boulila, Sfax, Tunisia.
Biochem Biophys Res Commun. 2011 May 20;408(4):654-7. doi: 10.1016/j.bbrc.2011.04.079. Epub 2011 Apr 21.
Dravet syndrome (DS), previously known as severe myoclonic epilepsy of infancy, is one of the most severe forms of childhood epilepsy. DS is caused by a mutation in the neuronal voltage-gated sodium-channel alpha-subunit gene (SCN1A). However, 25-30% of patients with DS are negative for the SCN1A mutation screening, suggesting that other molecular mechanisms may account for these disorders. Recently, the first case of DS caused by a mutation in the neuronal voltage-gated sodium-channel beta-subunit gene (SCN1B) was also reported. In this report we aim to make the molecular analysis of the SCN1A and SCN1B genes in two Tunisian patients affected with DS. The SCN1A and SCN1B genes were tested for mutations by direct sequencing. No mutation was revealed in the SCN1A and SCN1B genes by sequencing analyses. On the other hand, 11 known single nucleotide polymorphisms were identified in the SCN1A gene and composed a putative disease-associated haplotype in patients with DS phenotype. One of the two patients with putative disease-associated haplotype in SCN1A had also one known single nucleotide polymorphism in the SCN1B gene. The sequencing analyses of the SCN1A gene revealed the presence of a putative disease-associated haplotype in two patients affected with Dravet syndrome.
德拉韦特综合征(DS),以前称为婴儿严重肌阵挛性癫痫,是儿童中最严重的癫痫形式之一。DS 是由神经元电压门控钠离子通道α亚单位基因(SCN1A)的突变引起的。然而,25-30%的 DS 患者 SCN1A 突变筛查呈阴性,这表明可能存在其他分子机制导致这些疾病。最近,也有报道称首次发现神经元电压门控钠离子通道β亚单位基因(SCN1B)突变可导致 DS。本研究旨在对两名患有 DS 的突尼斯患者的 SCN1A 和 SCN1B 基因进行分子分析。通过直接测序对 SCN1A 和 SCN1B 基因进行突变检测。测序分析未发现 SCN1A 和 SCN1B 基因的突变。另一方面,在 SCN1A 基因中鉴定出 11 个已知的单核苷酸多态性,这些多态性构成了 DS 表型患者的潜在疾病相关单倍型。具有潜在疾病相关单倍型的两名患者之一在 SCN1B 基因中也存在一个已知的单核苷酸多态性。SCN1A 基因的测序分析显示,两名患有德拉韦特综合征的患者存在潜在疾病相关的单倍型。