Montfort M, Vilageliu L, Garcia-Giralt N, Guidi S, Coll M J, Chabás A, Grinberg D
Departament de Genètica, Universitat de Barcelona, Spain.
Hum Mutat. 1998;12(4):274-9. doi: 10.1002/(SICI)1098-1004(1998)12:4<274::AID-HUMU9>3.0.CO;2-F.
The gene resposible for Sanfilippo syndrome type A, a lysosomal disorder caused by deficiency of sulfamidase, was recently cloned and more than 40 mutations were identified. This paper presents the mutation analysis and clinical findings in 11 Spanish patients in whom 19 of the 22 mutant alleles have been identified. This is the first report on mutations in Spanish Sanfilippo A patients. Seven different mutations were found, four of which (Q85R, R206P, A354P, and L386R) were not previously described. Mutation 1091delC was the most prevalent, accounting for nearly one-half of the mutated alleles, while mutations R245H and R74C were not found. Haplotype analysis suggests a founder effect as the cause of the high frequency of 1091delC in this population.
导致A型Sanfilippo综合征的基因,一种由硫酸酰胺酶缺乏引起的溶酶体疾病,最近已被克隆,并且鉴定出了40多种突变。本文介绍了11名西班牙患者的突变分析和临床发现,其中已鉴定出22个突变等位基因中的19个。这是关于西班牙Sanfilippo A型患者突变的首次报告。发现了7种不同的突变,其中4种(Q85R、R206P、A354P和L386R)以前未曾描述过。突变1091delC最为普遍,占突变等位基因的近一半,而未发现R245H和R74C突变。单倍型分析表明,奠基者效应是该人群中1091delC高频率出现的原因。