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IV型高脂蛋白血症与雄激素受体基因中CAG三联体重复序列的中度不稳定性。对一个患有肯尼迪-奥尔特-宋氏病的中国家庭的脂质、性激素及分子研究。

Type IV hyperlipoproteinemia and moderate instability of CAG triplet expansion in the androgen-receptor gene. Lipid, sex hormone and molecular study in a Chinese family with Kennedy-Alter-Sung disease.

作者信息

Liu C S, Chang Y C, Chen D F, Huang C C, Pang C Y, Lee H C, Cheng C C, Horng C J, Wei Y H

机构信息

Department of Clinical Medicine, National Yang-Ming University, Taipei, Taiwan ROC.

出版信息

Acta Neurol Scand. 1995 Nov;92(5):398-404. doi: 10.1111/j.1600-0404.1995.tb00154.x.

Abstract

Kennedy-Alter-Sung (KAS) disease in a hereditary lower motor neuron disease. In this study, we investigate 2 KAS patients presenting with progressive muscle weakness and wasting, action tremor, perioral fasciculation and gynecomastia. Three carriers and 5 healthy members from this 3-generation KAS Chinese family and 60 normal Chinese controls were included in this study. Hormone studies revealed normal serum level in thyrotropin, prolactin, testosterone, leuteinizing hormone, follicle stimulating hormone, and estradiol. Lipid study disclosed type IV hyperlipoproteinemia in 2 KAS patients and 3 healthy members. Molecular studies revealed that the number of CAG triplet repeats in the first exon of androgen receptor gene of the normal allele is in the range of 15-19 and 12-25 in this family and normal controls, respectively. However, the number of CAG repeat of androgen receptor gene were unstable in the mutant alleles with a range of 41-45 and increased from generation to generation (genomic anticipation) in the 2 KAS patients and 3 female carriers. We conclude that the CAG triplet repeats in mutant allele were unstable in the family with the KAS disease. Furthermore, type IV hyperlipoproteinemia may be a co-transmitted syndrome in the family with KAS disease.

摘要

肯尼迪 - 奥尔特 - 宋氏(KAS)病是一种遗传性下运动神经元疾病。在本研究中,我们调查了2例表现为进行性肌肉无力和萎缩、动作性震颤、口周肌束震颤和男性乳房发育的KAS患者。本研究纳入了这个三代KAS中国家系的3名携带者和5名健康成员以及60名正常中国对照者。激素研究显示促甲状腺激素、催乳素、睾酮、黄体生成素、卵泡刺激素和雌二醇的血清水平正常。血脂研究发现2例KAS患者和3名健康成员存在IV型高脂蛋白血症。分子研究表明,该家系和正常对照者中,正常等位基因雄激素受体基因第一外显子中CAG三联体重复序列的数量分别在15 - 19和12 - 25范围内。然而,在2例KAS患者和3名女性携带者中,雄激素受体基因的CAG重复序列数量在突变等位基因中不稳定,范围为41 - 45,且代代增加(基因组早现)。我们得出结论,在患有KAS病的家系中,突变等位基因中的CAG三联体重复序列不稳定。此外,IV型高脂蛋白血症可能是KAS病家系中的一种共传递综合征。

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