Percy A K
Department of Pediatrics and Neurology, Baylor College of Medicine, Houston.
Brain Dev. 1992 May;14 Suppl:S57-62.
The current status of neurochemistry research with respect to the Rett syndrome (RS) is reviewed and correlations developed with the previously described neuropathologic changes. Despite reports of abnormalities in intermediary metabolism and the biogenic amine neurotransmitters, follow-up studies have failed to support any consistent abnormality in either area. In general, the levels of membrane lipid constituents in various brain regions have been similar to control values. New information suggests a disturbance in the ganglioside pattern leading to a reduction in GD1a and GT1b gangliosides in cerebrum and cerebellum. The corresponding reduction of these gangliosides in cerebrospinal fluid, if shown to be specific for RS, could represent an important clue in confirming the clinical diagnosis. Evidence of pervasive growth failure is presented and the current status of the role of beta-endorphins in RS is described. Considerations regarding future directions for research in RS are presented. These strategies include an approach to the fundamental question, namely, the identification of the primary molecular defect.
本文综述了雷特综合征(RS)神经化学研究的现状,并将其与先前描述的神经病理学变化建立了相关性。尽管有报道称中间代谢和生物胺神经递质存在异常,但后续研究未能证实这两个领域存在任何一致的异常情况。总体而言,不同脑区的膜脂成分水平与对照值相似。新信息表明神经节苷脂模式存在紊乱,导致大脑和小脑中GD1a和GT1b神经节苷脂减少。如果脑脊液中这些神经节苷脂的相应减少被证明是RS所特有的,那么这可能是确诊临床诊断的重要线索。文中还介绍了普遍生长发育迟缓的证据,并描述了β-内啡肽在RS中的作用现状。文中还提出了关于RS未来研究方向的思考。这些策略包括解决一个基本问题,即识别主要分子缺陷的方法。