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莱施-奈恩综合征中的微管解聚?(莱施-奈恩综合征与微管)

Disassembly of microtubules in the Lesch-Nyhan Syndrome? (Lesch-Nyhan syndrome and microtubules).

作者信息

Schneider W, Morgenstern E, Reimers H J

出版信息

Klin Wochenschr. 1979 Feb 15;57(4):181-6. doi: 10.1007/BF01477406.

Abstract

The Lesch-Nyhan syndrome is an unusual disease. It combines neurological disorders, behavioural disturbances, metabolic changes and haematological symptoms. The syndrome is caused by an X-chromosomal transmitted enzyme deficiency of the 'salvage pathway' in purine metabolism. The hitherto unexplained pathogenesis was the reason for investigations into metabolism and morphology of the blood cells of a patient suffering from the syndrome. Along with the defect in guanine nucleotide resynthesis there was a defect of microtubules in platelets and a sphaerocytosis in red cells, which could be the result of a disassembly of structural proteins. The development and maintenance of the highly heteromorphic structure of nerve cells and the neuronal function including axonal transport of cell organelles and transmitters is dependent on microtubules. Thus a disassembly of microtubules could be the mechanism in the pathogenesis of this complex syndrome.

摘要

莱施-奈恩综合征是一种罕见疾病。它兼具神经功能紊乱、行为障碍、代谢变化和血液学症状。该综合征由嘌呤代谢中“补救途径”的X染色体隐性遗传酶缺乏引起。此前无法解释的发病机制是对一名患有该综合征患者的血细胞代谢和形态进行研究的原因。除了鸟嘌呤核苷酸再合成缺陷外,血小板中还存在微管缺陷,红细胞中存在球形红细胞增多症,这可能是结构蛋白分解的结果。神经细胞高度异形结构的发育和维持以及包括细胞器和递质轴突运输在内的神经元功能依赖于微管。因此,微管的分解可能是这种复杂综合征发病机制中的一个环节。

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