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来自胡勒氏病、施艾氏病和胡勒/施艾氏综合征复合型患者的成纤维细胞杂交研究:对等位基因突变假说的支持

Hybridization studies of fibroblasts from Hurler, Scheie, and Hurler/Scheie compound patients: support for the hypothesis of allelic mutants.

作者信息

Fortuin J J, Kleijer W J

出版信息

Hum Genet. 1980 Feb;53(2):155-9. doi: 10.1007/BF00273487.

Abstract

Heterokaryons were made by fusion of alpha-L-iduronidase-deficient fibroblasts from patients with the Hurler, Scheie, or Hurler/Scheie compound syndrome. The fused cell populations remained alpha-L-iduronidase deficient and accumulated 35S-labeled glycosaminoglycans (35S-GAG) to the same extent as the parental cells strains. Also, when 35S-GAG accumulation was studied by autoradiography at the level of single bi- and multinuclear hybrid cells, no evidence was found for genetic complementation. The results support the hypothesis that the mutations in the Hurler and Scheie syndromes are allelic, and they are compatible with the view that patients with intermediate phenotypes represent genetic compounds.

摘要

通过将患有Hurler综合征、Scheie综合征或Hurler/Scheie复合综合征患者的α-L-艾杜糖醛酸酶缺陷成纤维细胞进行融合,制备了异核体。融合后的细胞群体仍然缺乏α-L-艾杜糖醛酸酶,并且积累35S标记的糖胺聚糖(35S-GAG)的程度与亲代细胞株相同。此外,当通过放射自显影在单个双核和多核杂交细胞水平上研究35S-GAG积累时,未发现基因互补的证据。这些结果支持以下假设:Hurler综合征和Scheie综合征中的突变是等位基因,并且它们与中间表型患者代表遗传复合物的观点一致。

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