Ghidoni Roberta, Paterlini Anna, Benussi Luisa
Proteomics Unit, IRCCS Istituto Centro San Giovanni di Dio Fatebenefratelli Brescia, Italy.
Am J Neurodegener Dis. 2012;1(2):180-90. Epub 2012 Aug 2.
Progranulin is a growth factor involved in the regulation of multiple processes including tumorigenesis, wound repair, development, and inflammation. The recent discovery that mutations in the gene encoding for progranulin (GRN) cause frontotemporal lobar degeneration (FTLD), and other neurodegenerative diseases leading to dementia, has brought renewed interest in progranulin and its functions in the central nervous system. GRN null mutations cause protein haploinsufficiency, leading to a significant decrease in progranulin levels that can be detected in plasma, serum and cerebrospinal fluid (CSF) of mutation carriers. The dosage of circulating progranulin sped up the identification of GRN mutations thus favoring genotype-phenotype correlation studies. Researchers demonstrated that, in GRN null mutation carriers, the shortage of progranulin invariably precedes clinical symptoms and thus mutation carriers are "captured" regardless of their disease status. GRN is a particularly appealing gene for drug targeting, in the way that boosting its expression may be beneficial for mutation carriers, preventing or delaying the onset of GRN-related neurodegenerative diseases. Physiological regulation of progranulin expression level is only partially known. Progranulin expression reflects mutation status and, intriguingly, its levels can be modulated by some additional factor (i.e. genetic background; drugs). Thus, factors increasing the production and secretion of progranulin from the normal gene are promising potential therapeutic avenues. In conclusion, peripheral progranulin is a nonintrusive highly accurate biomarker for early identification of mutation carriers and for monitoring future treatments that might boost the level of this protein.
颗粒蛋白前体是一种生长因子,参与包括肿瘤发生、伤口修复、发育和炎症在内的多种过程的调节。最近发现,编码颗粒蛋白前体(GRN)的基因突变会导致额颞叶痴呆(FTLD)以及其他导致痴呆的神经退行性疾病,这使得人们对颗粒蛋白前体及其在中枢神经系统中的功能重新产生了兴趣。GRN基因无效突变导致蛋白质单倍体不足,导致在突变携带者的血浆、血清和脑脊液(CSF)中可检测到的颗粒蛋白前体水平显著降低。循环颗粒蛋白前体的剂量加快了GRN突变的鉴定,从而有利于基因型-表型相关性研究。研究人员证明,在GRN基因无效突变携带者中,颗粒蛋白前体的缺乏总是先于临床症状出现,因此无论其疾病状态如何,突变携带者都能被“捕获”。GRN是一个特别有吸引力的药物靶点基因,因为提高其表达可能对突变携带者有益,预防或延缓GRN相关神经退行性疾病的发作。颗粒蛋白前体表达水平的生理调节仅部分为人所知。颗粒蛋白前体的表达反映了突变状态,有趣的是,其水平可以被一些其他因素(即遗传背景;药物)调节。因此,增加正常基因产生和分泌颗粒蛋白前体的因素是很有前景的潜在治疗途径。总之,外周颗粒蛋白前体是一种非侵入性的高度准确的生物标志物,可用于早期识别突变携带者以及监测未来可能提高这种蛋白质水平的治疗。