Muñoz-Ballester Carmen, Berthier Arnaud, Viana Rosa, Sanz Pascual
Instituto de Biomedicina de Valencia (CSIC) and Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Valencia, Spain.
Instituto de Biomedicina de Valencia (CSIC) and Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Valencia, Spain.
Biochim Biophys Acta. 2016 Jun;1862(6):1074-83. doi: 10.1016/j.bbadis.2016.03.008. Epub 2016 Mar 11.
Lafora disease (LD, OMIM 254780) is a fatal rare disorder characterized by epilepsy and neurodegeneration. Although in recent years a lot of information has been gained on the molecular basis of the neurodegeneration that accompanies LD, the molecular basis of epilepsy is poorly understood. Here, we present evidence indicating that the homeostasis of glutamate transporter GLT-1 (EAAT2) is compromised in mouse models of LD. Our results indicate that primary astrocytes from LD mice have reduced capacity of glutamate transport, probably because they present a reduction in the levels of the glutamate transporter at the plasma membrane. On the other hand, the overexpression in cellular models of laforin and malin, the two proteins related to LD, results in an accumulation of GLT-1 (EAAT2) at the plasma membrane and in a severe reduction of the ubiquitination of the transporter. All these results suggest that the laforin/malin complex slows down the endocytic recycling of the GLT-1 (EAAT2) transporter. Since, defects in the function of this transporter lead to excitotoxicity and epilepsy, we suggest that the epilepsy that accompanies LD could be due, at least in part, to deficiencies in the function of the GLT-1 (EAAT2) transporter.
拉福拉病(LD,OMIM 254780)是一种致命的罕见疾病,其特征为癫痫和神经退行性变。尽管近年来已获得了许多关于伴随LD的神经退行性变分子基础的信息,但癫痫的分子基础仍知之甚少。在此,我们提供证据表明,在LD小鼠模型中,谷氨酸转运体GLT-1(EAAT2)的稳态受到破坏。我们的结果表明,来自LD小鼠的原代星形胶质细胞的谷氨酸转运能力降低,这可能是因为它们在质膜上的谷氨酸转运体水平降低。另一方面,与LD相关的两种蛋白质——拉福林和马啉在细胞模型中的过表达,导致GLT-1(EAAT2)在质膜上积累,并使该转运体的泛素化严重减少。所有这些结果表明,拉福林/马啉复合物减缓了GLT-1(EAAT2)转运体的内吞再循环。由于该转运体功能缺陷会导致兴奋性毒性和癫痫,我们认为伴随LD的癫痫可能至少部分归因于GLT-1(EAAT2)转运体功能的缺陷。