Ługowska A, Berger J, Tylki-Szymańska A, Löschl B, Molzer B, Zobel M, Czartoryska B
Institute of Psychiatry and Neurology, Department of Genetics, Warsaw, Poland.
Clin Genet. 2005 Jul;68(1):48-54. doi: 10.1111/j.1399-0004.2005.00451.x.
The occurrence and genotype-phenotype correlations of the eight most common mutations in the arylsulfatase A (ARSA) gene were studied in 43 unrelated Polish patients suffering from different types of metachromatic leukodystrophy (MLD). Screening for mutations p.R84Q, p.S96F, c.459+1G>A, p.I179S, p.A212V, c.1204+1G>A, p.P426L, and c.1401-1411del allowed the identification of 53.5% of the mutant alleles. In the whole investigated group of patients, mutations c.459+1G>A and p.P426L were the most frequent, 19 and 17%, respectively. The prevalence of the third most frequent mutation, i.e. p.I179S (13%), seems to be higher than that in other populations. The incidence of c.1204+1G>A was 5%, which is higher than reported earlier (2%). It seems that p.I179S and c.1204+1G>A are more prevalent in MLD patients from Poland than from other countries. In the group examined by us, mutations p.R84Q, p.S96F, p.A212V, and c.1401-1411del were not detected; thus, 46.5% of MLD alleles remained unidentified. This indicates that other, novel or already described, but rare, mutations exist in Polish population. In late infantile homozygotes for c.459+1G>A and one homozygote for c.1204+1G>A, first clinical symptom was motor deterioration. In adult homozygotes for p.P426L, the disease onset manifested as gait disturbances, followed by choreoathetotic movements, difficulties in swallowing, dysarthria, tremor, and nystagmus. In the carriers of the p.I179S mutation, the hallmark of the clinical picture was psychotic disturbances.
对43名患有不同类型异染性脑白质营养不良(MLD)的不相关波兰患者,研究了芳基硫酸酯酶A(ARSA)基因中8种最常见突变的发生情况及其基因型与表型的相关性。对p.R84Q、p.S96F、c.459+1G>A、p.I179S、p.A212V、c.1204+1G>A、p.P426L和c.1401 - 1411del突变进行筛查,可识别出53.5%的突变等位基因。在整个研究患者组中,c.459+1G>A和p.P426L突变最为常见,分别为19%和17%。第三常见突变p.I179S的发生率为13%,似乎高于其他人群。c.1204+1G>A的发生率为5%,高于先前报道的2%。似乎p.I179S和c.1204+1G>A在波兰MLD患者中比在其他国家更普遍。在我们检查的组中,未检测到p.R84Q、p.S96F、p.A212V和c.1401 - 1411del突变;因此,46.5%的MLD等位基因仍未被识别。这表明波兰人群中存在其他新的或已描述但罕见的突变。在c.459+1G>A的晚婴型纯合子和一名c.1204+1G>A的纯合子中,首发临床症状为运动功能恶化。在p.P426L的成年纯合子中,疾病发作表现为步态障碍,随后出现舞蹈手足徐动症、吞咽困难、构音障碍、震颤和眼球震颤。在p.I179S突变携带者中,临床表现的特征是精神障碍。