Taylor H R, Hollows F C, Hopwood J J, Robertson E F
J Med Genet. 1978 Dec;15(6):455-61. doi: 10.1136/jmg.15.6.455.
The first 2 reported cases of a mucopolysaccharidosis occurring in an Australian aboriginal family are presented. Though these children had the characteristic morphological features of the Hurler syndrome, enzyme assay of cultured fibroblasts showed normal levels of alpha-L-iduronidase and decreased activity of arylsulphatase B. Thus, they represented the Hurler syndrome clinically, while they had the enzyme defect of the Maroteaux-Lamy syndrome, and they may represent a new severe form of the Maroteaux-Lamy syndrome. The parents of these children were first cousins. Though the children were not full blood aborigines, examination of the pedigree indicates that the gene originated in the common aboriginal family.
本文报道了澳大利亚一个原住民家庭中出现的首例黏多糖贮积症的2个病例。尽管这些患儿具有Hurler综合征的典型形态学特征,但培养的成纤维细胞酶分析显示α-L-艾杜糖醛酸酶水平正常,芳基硫酸酯酶B活性降低。因此,他们临床诊断为Hurler综合征,却存在Maroteaux-Lamy综合征的酶缺陷,可能代表Maroteaux-Lamy综合征的一种新的严重类型。这些患儿的父母是近亲。尽管这些患儿并非纯血统原住民,但系谱检查表明该基因源自同一个原住民家庭。