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动物细胞突变体代表了过氧化物酶体缺陷型 Zellweger 综合征的两个互补群。

Animal cell mutants represent two complementation groups of peroxisome-defective Zellweger syndrome.

作者信息

Shimozawa N, Tsukamoto T, Suzuki Y, Orii T, Fujiki Y

机构信息

Meiji Institute of Health Science, Odawara, Japan.

出版信息

J Clin Invest. 1992 Nov;90(5):1864-70. doi: 10.1172/JCI116063.

Abstract

Generalized peroxisome-deficient disorders including cerebro-hepato-renal Zellweger syndrome, neonatal adrenoleukodystrophy, and infantile Refsum disease are autosomal recessive diseases, where catalase-containing particles (peroxisomes) are morphologically absent. We previously isolated two Chinese hamster ovary (CHO) cell mutants (Z24 and Z65) that resemble the fibroblasts from patients with such diseases, in their defective peroxisome assembly (Tsukamoto, T., S. Yokota, and Y. Fujiki. 1990. J. Cell Biol. 110:651-660). Here we report isolation by the P9OH/UV method of a peroxisome-deficient CHO mutant, ZP92, of the third complementation group distinct from those of Z24 and Z65. Peroxisomal membrane ghosts were noted by immunochemical staining in all of the CHO mutants. Complementation analysis by cell fusion of the CHO mutants with cultured fibroblasts from patients with generalized peroxisomal disorders revealed that two CHO mutants (Z24 and ZP92) represent the human complementation groups, E (the same as group 1 in the U.S.) and C (the same as group 4), respectively. These CHO cell mutants are an apparently relevant animal cell model for studies on the molecular bases and primary defects of human peroxisome-deficient diseases.

摘要

包括脑肝肾齐韦格综合征、新生儿肾上腺脑白质营养不良和婴儿型雷夫叙姆病在内的全身性过氧化物酶体缺乏症是常染色体隐性疾病,其形态学上缺乏含过氧化氢酶的颗粒(过氧化物酶体)。我们之前分离出了两个中国仓鼠卵巢(CHO)细胞突变体(Z24和Z65),它们在过氧化物酶体组装缺陷方面类似于此类疾病患者的成纤维细胞(冢本,T.,横田,S.,藤木,Y. 1990.《细胞生物学杂志》110:651 - 660)。在此,我们报告通过P9OH/紫外线方法分离出一个过氧化物酶体缺乏的CHO突变体ZP92,它属于与Z24和Z65不同的第三个互补群。通过免疫化学染色在所有CHO突变体中都观察到了过氧化物酶体膜空壳。CHO突变体与全身性过氧化物酶体疾病患者的培养成纤维细胞进行细胞融合的互补分析表明,两个CHO突变体(Z24和ZP92)分别代表人类互补群E(与美国的第1组相同)和C(与第4组相同)。这些CHO细胞突变体是用于研究人类过氧化物酶体缺乏症的分子基础和原发性缺陷的明显相关动物细胞模型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/31da/443247/2283ceb59d42/jcinvest00053-0240-a.jpg

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