Park Jiyun, An Seong Soo A, Giau Vo Van, Shim Kyuhwan, Youn Young Chul, Bagyinszky Eva, Kim SangYun
Department of Neurology, Ulsan University Medical School, Ulsan University Hospital, Ulsan, Republic of Korea.
Department of BioNano Technology and Gachon Medical Research Institute, Gachon University, Sungnam, Republic of Korea.
Neurobiol Aging. 2017 Aug;56:212.e11-212.e17. doi: 10.1016/j.neurobiolaging.2017.04.012. Epub 2017 Apr 26.
In the present study, a novel mutation in exon 7 of presenilin 1 (Leu232Pro) was discovered in a Korean patient with early-onset Alzheimer's disease, who represented memory decline at 37 years of age, followed by impairment in spatial activity and concentrations and personality changes. Imaging analyses with magnetic resonance scan showed diffuse atrophy in the frontoparietal regions. Targeted next generation sequencing and Sanger sequencing identified a heterozygous T to C transition at position 695 (c.695T>C) of in presenilin 1 gene (PSEN1), resulting in a novel missense mutation at codon 232 from leucine to proline (L232P). Several family members of the patient developed dementia, suggesting an autosomal dominant inheritance; however, we were unable to perform a segregation analysis to confirm this. Since the proline may play a role as a helix breaker, this mutation could significantly disturb the transmembrane helix domain-V of PSEN1 and perturb its protein functions. This hypothesis was supported by the results from the in silico analyses, predicted a major kink on this helix. Several leucine>proline substitutions in other PSEN1 transmembrane helices revealed aggressive AD phenotypes. Future functional studies would be needed to evaluate the pathogenicity of this mutation in AD.
在本研究中,在一名韩国早发性阿尔茨海默病患者中发现了早老素1第7外显子的一种新突变(Leu232Pro)。该患者37岁时出现记忆衰退,随后出现空间活动、注意力和性格改变。磁共振扫描成像分析显示额顶叶区域弥漫性萎缩。靶向二代测序和桑格测序在早老素1基因(PSEN1)第695位(c.695T>C)鉴定出一个杂合的T到C转换,导致第232密码子处由亮氨酸到脯氨酸的新错义突变(L232P)。该患者的几名家庭成员患上了痴呆症,提示常染色体显性遗传;然而,我们无法进行分离分析来证实这一点。由于脯氨酸可能起到螺旋破坏者的作用,这种突变可能会显著扰乱PSEN1的跨膜螺旋结构域-V并干扰其蛋白质功能。这一假设得到了计算机分析结果的支持,该分析预测该螺旋上有一个主要的扭结。PSEN1其他跨膜螺旋中的几个亮氨酸>脯氨酸替代显示出侵袭性的阿尔茨海默病表型。未来需要进行功能研究来评估这种突变在阿尔茨海默病中的致病性。