Shi Zhihong, Wang Ying, Liu Shuai, Liu Mengyuan, Liu Shuling, Zhou Yuying, Wang Jinhuan, Cai Li, Huo Ya Ruth, Gao Shuo, Ji Yong
Tianjin Key Laboratory of Cerebrovascular and Neurodegenerative Diseases, Tianjin Huanhu Hospital, Tianjin, China.
Dement Geriatr Cogn Disord. 2015;39(1-2):32-40. doi: 10.1159/000366272. Epub 2014 Oct 15.
Alzheimer's disease (AD) and frontotemporal dementia (FTD) are two common forms of primary neurodegenerative dementia. Mutations in 3 genes (PSEN1, PSEN2, and APP) have been identified in patients with early-onset AD.
We performed gene sequencing in PSEN1, PSEN2, and APP in 61 AD and 35 FTD Chinese patients. Amyloid load using (11)C-labeled Pittsburgh compound B ((11)C-PIB) positron emission tomography (PET) and cerebral glucose metabolism using (18)F-fludeoxyglucose PET were evaluated in patients carrying mutations.
We identified 1 known pathogenic PSEN1 (p.His163Arg, c.488A>G) mutation and 3 novel PSEN2 mutations in 6 patients. The novel mutation PSEN2 (p.His169Asn, c.505C>A) was identified in 1 patient with familial late-onset AD and in 1 sporadic FTD patient. The PSEN2 (p.Val214Leu, c.640G>T; p.Lys82Arg, c.245A>G) mutations were identified in 2 early-onset AD patients and 1 early-onset AD patient, respectively. Three patients with PSEN2 mutations were observed to have PIB retention on the cortex and striatum. One patient with the FTD phenotype was not observed to have PIB retention.
PSEN2 mutations are common in the Chinese Han population with a history of AD and FTD. Pathogenic mutations or risk variants in the PSEN2 gene can influence both FTD and AD phenotypic traits and show variations in neuroimaging characterization.
阿尔茨海默病(AD)和额颞叶痴呆(FTD)是原发性神经退行性痴呆的两种常见形式。早发性AD患者中已发现3个基因(PSEN1、PSEN2和APP)发生突变。
我们对61例AD中国患者和35例FTD中国患者的PSEN1、PSEN2和APP进行了基因测序。对携带突变的患者,使用(11)C标记的匹兹堡化合物B((11)C-PIB)正电子发射断层扫描(PET)评估淀粉样蛋白负荷,使用(18)F-氟脱氧葡萄糖PET评估脑葡萄糖代谢。
我们在6例患者中鉴定出1个已知的致病性PSEN1(p.His163Arg,c.488A>G)突变和3个新的PSEN2突变。新突变PSEN2(p.His169Asn,c.505C>A)在1例家族性晚发性AD患者和1例散发性FTD患者中被鉴定出。PSEN2(p.Val214Leu,c.640G>T;p.Lys82Arg,c.245A>G)突变分别在2例早发性AD患者和1例早发性AD患者中被鉴定出。观察到3例携带PSEN2突变的患者在皮质和纹状体有PIB滞留。1例具有FTD表型的患者未观察到PIB滞留。
PSEN2突变在有AD和FTD病史的中国汉族人群中很常见。PSEN2基因中的致病性突变或风险变异可影响FTD和AD的表型特征,并在神经影像学特征上表现出差异。