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黄斑突变小鼠的铜代谢:门克斯卷发疾病的动物模型。

Copper metabolism in the macular mutant mouse: an animal model of Menkes's kinky-hair disease.

作者信息

Shiraishi N, Aono K, Taguchi T

机构信息

Department of Radiation Medicine, Okayama University Medical School, Japan.

出版信息

Biol Neonate. 1988;54(3):173-80. doi: 10.1159/000242849.

DOI:10.1159/000242849
PMID:3219371
Abstract

The tissue copper contents were measured in mutant (hemizygous macular male and homozygous macular female), heterozygous macular female and normal mice. The copper content in kidney and small intestine from 7-day-old mutant and heterozygote were extremely high compared to normal mice, whereas the copper content in other tissues (liver, brain, spleen and serum) was low. Copper content in whole body of mutant mice was extremely low at three stages (18 days gestation, 1 day old, and 7 days old) compared to normal mice with the exception of the mutant fetus at 14 days of gestation. Renal copper contents in the mutant fetus at 18 days of gestation and in the 1-day-old mutant were not changed compared to normal mice, whereas that in 7- and 14-day-old mutant mice was significantly elevated. Hepatic copper content of the mutant mice was extremely low at all stages compared to normal mice. Copper therapy was applied to 7-day-old mutant mice. At 1 day after injection, hepatic copper content in the mutants had increased slightly in comparison with the normal control mice, whereas renal copper content was extremely increased. At 7 days after injection, hepatic copper content in the mutants was decreased greatly in comparison with normal control mice, whereas an increase in renal copper content had remained.

摘要

对突变体(半合子黄斑雄性和纯合子黄斑雌性)、杂合子黄斑雌性和正常小鼠的组织铜含量进行了测量。与正常小鼠相比,7日龄突变体和杂合子的肾脏和小肠中的铜含量极高,而其他组织(肝脏、大脑、脾脏和血清)中的铜含量较低。与正常小鼠相比,除妊娠14天的突变体胎儿外,突变体小鼠在三个阶段(妊娠18天、1日龄和7日龄)的全身铜含量极低。与正常小鼠相比,妊娠18天的突变体胎儿和1日龄突变体的肾脏铜含量没有变化,而7日龄和14日龄突变体小鼠的肾脏铜含量显著升高。与正常小鼠相比,突变体小鼠在所有阶段的肝脏铜含量都极低。对7日龄突变体小鼠进行了铜治疗。注射后1天,与正常对照小鼠相比,突变体的肝脏铜含量略有增加,而肾脏铜含量则极度增加。注射后7天,与正常对照小鼠相比,突变体的肝脏铜含量大幅下降,而肾脏铜含量仍保持增加。

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1
Copper metabolism in the macular mutant mouse: an animal model of Menkes's kinky-hair disease.黄斑突变小鼠的铜代谢:门克斯卷发疾病的动物模型。
Biol Neonate. 1988;54(3):173-80. doi: 10.1159/000242849.
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Autoradiographic demonstration of the copper-accumulating tissues in mice with a defect homologous to Menkes' Kinky Hair disease.与门克斯卷发疾病同源缺陷的小鼠体内铜蓄积组织的放射自显影证明
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Copper-induced toxicity in macular mutant mouse: an animal model for Menkes' kinky-hair disease.铜诱导的黄斑突变小鼠毒性:一种门克斯卷发综合征的动物模型。
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引用本文的文献

1
Mottled Mice and Non-Mammalian Models of Menkes Disease.斑驳小鼠与门克斯病的非哺乳动物模型
Front Mol Neurosci. 2015 Dec 18;8:72. doi: 10.3389/fnmol.2015.00072. eCollection 2015.
2
Prenatal treatment of mosaic mice (Atp7a mo-ms) mouse model for Menkes disease, with copper combined by dimethyldithiocarbamate (DMDTC).对 Menkes 病的 mosaic 小鼠(Atp7a mo-ms)模型进行产前治疗,使用二甲基二硫代氨基甲酸盐(DMDTC)结合铜。
PLoS One. 2012;7(7):e40400. doi: 10.1371/journal.pone.0040400. Epub 2012 Jul 18.
3
Effect of copper and diethyldithiocarbamate combination therapy on the macular mouse, an animal model of Menkes disease.
铜与二乙基二硫代氨基甲酸盐联合疗法对黄斑小鼠(一种门克斯病动物模型)的影响。
J Inherit Metab Dis. 2005;28(6):971-8. doi: 10.1007/s10545-005-0150-6.
4
Pathological structure of the kidney from adult mice with mosaic mutation.具有镶嵌突变的成年小鼠肾脏的病理结构
J Inherit Metab Dis. 2002 Dec;25(8):647-59. doi: 10.1023/a:1022877130344.
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Genes regulating copper metabolism.调节铜代谢的基因。
Mol Cell Biochem. 1998 Nov;188(1-2):57-62.
6
Neuropathological study on cerebellum of macular mutant mouse heterozygote.黄斑突变小鼠杂合子小脑的神经病理学研究
Acta Neuropathol. 1993;86(5):411-7. doi: 10.1007/BF00228573.
7
Intracellular distribution of hepatic copper in macular mutant mice. An animal model of Menkes' kinky-hair disease.黄斑突变小鼠肝脏铜的细胞内分布。一种门克斯卷发综合征的动物模型。
Biol Trace Elem Res. 1993 May-Jun;37(2-3):179-86. doi: 10.1007/BF02783793.
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Effect of age and sex on copper-induced toxicity in the macular mutant mouse. An animal model for Menkes' kinky-hair disease.年龄和性别对黄斑突变小鼠铜诱导毒性的影响。一种门克斯卷发疾病的动物模型。
Biol Trace Elem Res. 1993 Nov-Dec;39(2-3):129-37. doi: 10.1007/BF02783183.