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鳞屑小鼠突变体具有先前未被认识到的血液学异常,并且类似于威斯科特-奥尔德里奇综合征。

The scurfy mouse mutant has previously unrecognized hematological abnormalities and resembles Wiskott-Aldrich syndrome.

作者信息

Lyon M F, Peters J, Glenister P H, Ball S, Wright E

机构信息

Medical Research Council Radiobiology Unit, Didcot, Oxon, United Kingdom.

出版信息

Proc Natl Acad Sci U S A. 1990 Apr;87(7):2433-7. doi: 10.1073/pnas.87.7.2433.

Abstract

The X chromosome-linked scurfy (sf) mutant of the mouse is recognized by the scaliness of the skin from which the name is derived and results in death of affected males at about 3-4 weeks of age. Consideration of known man-mouse homologies of the X chromosome prompted hematological studies, which have shown that the blood is highly abnormal. The platelet and erythrocyte counts are both reduced and become progressively lower relative to normal as the disease progresses. There is gastrointestinal bleeding, and most animals appear to die of severe anemia. By contrast, the leukocyte count is consistently raised. Some animals showed signs of infection but it is not yet clear whether there is immunodeficiency. Other features include the scaly skin and apparently reduced lateral growth of the skin, conjunctivitis, and diarrhea in some animals. The mutant resembles Wiskott-Aldrich syndrome in man, which is characterized by thrombocytopenia, eczema, diarrhea, and immunodeficiency. The loci of the human and mouse genes lie in homologous segments of the X chromosome, although apparently in somewhat different positions relative to other gene loci. Scurfy differs from Wiskott-Aldrich syndrome in that scurfy males are consistently hypogonadal.

摘要

小鼠的X染色体连锁的鳞屑(sf)突变体因其皮肤出现鳞屑而得名,患病雄性小鼠通常在3至4周龄时死亡。鉴于已知的人与小鼠X染色体同源性,研究人员开展了血液学研究,结果表明血液存在高度异常。随着病情发展,血小板和红细胞计数均降低,且相对于正常水平逐渐下降。存在胃肠道出血,大多数动物似乎死于严重贫血。相比之下,白细胞计数持续升高。一些动物表现出感染迹象,但目前尚不清楚是否存在免疫缺陷。其他特征包括皮肤鳞屑以及皮肤横向生长明显减缓、结膜炎,部分动物出现腹泻。该突变体类似于人类的威斯科特-奥尔德里奇综合征,其特征为血小板减少、湿疹、腹泻和免疫缺陷。人类和小鼠基因的位点位于X染色体的同源区段,尽管相对于其他基因位点,它们的位置显然略有不同。鳞屑突变体与威斯科特-奥尔德里奇综合征的不同之处在于,鳞屑突变体雄性小鼠始终性腺功能减退。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8a0e/53703/9ee27bae9538/pnas01032-0062-a.jpg

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