Ganesh S, Agarwala K L, Amano K, Suzuki T, Delgado-Escueta A V, Yamakawa K
Laboratory for Neurogenetics, RIKEN Brain Science Institute, Wako-shi, Saitama, 351-0198, Japan.
Biochem Biophys Res Commun. 2001 May 25;283(5):1046-53. doi: 10.1006/bbrc.2001.4914.
Lafora's disease, an autosomal recessive progressive myoclonus epilepsy, is caused by mutations in the EPM2A gene encoding a dual-specificity phosphatase (DSP) named laforin. Here, we analyzed the developmental and regional expression of murine Epm2a and discussed its evolutionary conservation. A phylogenetic analysis indicated that laforin is evolutionarily distant from other DSPs. Southern zoo blot analysis suggested that conservation of Epm2a gene is limited to mammals. Laforin orthologs (human, mouse, and rat) display more than 94% similarity. All missense mutations known in Lafora disease patients affect conserved residues, suggesting that they may be essential for laforin's function. Epm2a is expressed widely in various organs but not homogeneously in brain. The levels of Epm2a transcripts in mice brains increase postnatally, attaining its highest level in adults. The most intense signal was detected in the cerebellum, hippocampus, cerebral cortex, and the olfactory bulb. Our results suggest that Epm2a is functionally conserved in mammals and is involved in growth and maturation of neural networks.
拉福拉病是一种常染色体隐性进行性肌阵挛癫痫,由编码名为拉福林的双特异性磷酸酶(DSP)的EPM2A基因突变引起。在此,我们分析了小鼠Epm2a的发育和区域表达,并讨论了其进化保守性。系统发育分析表明,拉福林在进化上与其他DSPs距离较远。Southern杂交分析表明,Epm2a基因的保守性仅限于哺乳动物。拉福林直系同源物(人类、小鼠和大鼠)显示出超过94%的相似性。拉福拉病患者中已知的所有错义突变都影响保守残基,这表明它们可能对拉福林的功能至关重要。Epm2a在各种器官中广泛表达,但在大脑中表达不均匀。小鼠大脑中Epm2a转录本的水平在出生后增加,在成年时达到最高水平。在小脑、海马体、大脑皮层和嗅球中检测到最强的信号。我们的结果表明,Epm2a在哺乳动物中功能保守,并且参与神经网络的生长和成熟。