Li Jianxue, Pelletier Marc R, Perez Velazquez Jose-Luis, Carlen Peter L
Division of Cellular and Molecular Biology, Toronto Western Research Institute, University of Toronto, Toronto, Ontario M5T 2S8, Canada.
Mol Cell Neurosci. 2002 Feb;19(2):138-51. doi: 10.1006/mcne.2001.1085.
Lack of expression of the fragile X mental retardation protein (FMRP), due to silencing of the FMR1 gene, causes the Fragile X syndrome. Although FMRP was characterized previously to be an RNA binding protein, little is known about its function or the mechanisms underlying the Fragile X syndrome. Here we report that the alpha-amino-3-hydroxy-5-methyl-4-isoxazole propionate receptor subunit, GluR1, was decreased in the cortical synapses, but not in the hippocampus or cerebellum, of FMR1 gene knockout mice. Reduced long-term potentiation (LTP) was also found in the cortex but not in the hippocampus. Another RNA binding protein, FXR; the N-methyl-D-aspartate receptor subunit, NR2; and other learning-related proteins including c-fos, synapsin, myelin proteolipid protein, and cAMP response element binding protein were not different between FMR1 gene knockout and wild-type mice. These findings suggest that the depressed cortical GluR1 expression and LTP associated with FMRP deficiency could contribute to the Fragile X phenotype.
由于FMR1基因沉默导致脆性X智力低下蛋白(FMRP)表达缺失,会引发脆性X综合征。尽管FMRP之前被鉴定为一种RNA结合蛋白,但其功能或脆性X综合征的潜在机制仍知之甚少。在此我们报告,在FMR1基因敲除小鼠的皮质突触中,α-氨基-3-羟基-5-甲基-4-异恶唑丙酸受体亚基GluR1减少,但在海马体或小脑中未减少。在皮质中也发现长时程增强(LTP)降低,但在海马体中未出现此现象。另一种RNA结合蛋白FXR、N-甲基-D-天冬氨酸受体亚基NR2以及其他与学习相关的蛋白,包括c-fos、突触素、髓磷脂蛋白脂蛋白和cAMP反应元件结合蛋白,在FMR1基因敲除小鼠和野生型小鼠之间并无差异。这些发现表明,与FMRP缺乏相关的皮质GluR1表达降低和LTP可能导致脆性X综合征的表型。